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AJOMS Vol One 2024

Australasian Journal of

ORAL AND MAXILLOFACIAL SURGERY Official journal of the Australian and New Zealand Association of

Editor

Oral and Maxillofacial Surgeons and the Oceania Region

Professor A Goss editorajoms@anzaoms.org Deputy Editors Professor A Heggie AM Professor D Wiesenfeld

+61 2 8091 0535 ISSN: 2982-0065

ajoms@anzaoms.org

Level 13, 37 York Street, Sydney NSW 2000

AJOMS

CONTENTS 6

EDITORIAL

2024 APRIL Volume 1 | Issue 1

A RARE COMPLICATION OF SPHENOID SINUSITIS

PROTON BEAM THERAPY IN OROPHARYNGEAL CANCER A critical examination of efficacy and side effect

Septic arthritis of the temporomandibular joint

SIXTY YEARS ON Reflections of a major maxillofacial trauma centre

mitigation with this new treatment modality

107 THE EVIDENCE BASE FOR CONTEMPORARY 17

METASTATIC SQUAMOUS CELL

ORAL AND MAXILLOFACIAL SURGERY

CARCINOMA OF THE SALIVARY GLAND

RESEARCH IN DENTOALVEOLAR SURGERY

Treatment and prognostic factors related

IN AUSTRALIA

to overall survival and recurrence

THE EPIDEMIOLOGY OF TONGUE CANCER

OSTEONECROSIS OF THE JAW IN

A systematic review and meta-analysis

PATIENTS ON DENOSUMAB THERAPY

ORAL SQUAMOUS CELL CARCINOMA

DENTAL EXTRACTIONS

IN NON-SMOKING, NON-DRINKING ELDERLY FEMALES A follow-up study

PROGNOSTIC ASSOCIATION OF POST-OPERATIVE RADIOTHERAPY & DEPTH OF INVASION OF TONGUE CARCINOMAS

RECURRENT DESMOPLASTIC FIBROMA OF THE MAXILLA Case report and review of the literature

NEW ZEALAND PIONEERS & LEADERS Kiwi contributions to maxillofacial surgery from the early 20th century to the present day

SURVEY OF AUSTRALIAN & NEW ZEALAND ORAL AND MAXILLOFACIAL SURGEONS 2020 Forty years on monitoring scope and workforce

115 INCIDENCE RATE OF MEDICATION-RELATED

ALLOPLASTIC TEMPOROMANDIBULAR JOINT REPLACEMENT Past, present and future considerations

FOR OSTEOPOROSIS UNDERGOING

124 MAXILLARY PREMOLAR AUTOTRANSPLANTATION FOR A CHILD WITH BILATERAL CLEFT LIP AND PALATE

130 PUBLICATIONS BY AUSTRALIAN & NEW ZEALAND AUTHORS IN THREE MAJOR ORAL AND MAXILLOFACIAL SURGERY JOURNALS

136 A SURVEY OF ADELAIDE UNDERGRADUATE DENTAL STUDENTS’ CONFIDENCE IN DENTOALVEOLAR SURGERY

143 MYCOTIC ANEURYSM SECONDARY TO ODONTOGENIC INFECTION CAUSING BILATERAL CAVERNOUS SINUS THROMBOSIS A rare but important complication

148 TRENDS IN ORAL AND MAXILLOFACIAL SURGERY DURING THE COVID-19 PANDEMIC The Australian experience

159 UTILISATION OF ORAL AND MAXILLOFACIAL SPECIALIST TELEHEALTH SERVICES DURING COVID-19 IN AUSTRALIA A trend analysis ANZAOMS PREVIEW VERSION |

The Australasian Journal of Oral & Maxillofacial Surgery is the official scientific journal of the Australia & New Zealand Association of Oral &

F O U N D AT I O N E D I T O R

EDITORIAL BOARD

Alastair Goss

Business Manager Dieter Gebauer

• Emeritus Professor of Oral & Maxillofacial Surgery, The University of Adelaide • Emeritus Consultant Surgeon, The Royal Adelaide Hospital Adelaide, Australia

Perth, Australia

Maxillofacial Surgeons. DEPUTY EDITORS Aim & Scope The Australasian Journal of Oral & Maxillofacial Surgery is the premier forum for the exchange of information for new and significant research in oral and maxillofacial surgery, promoting the surgical discipline in the Oceanic region.

Oceania comprises 19 countries, spread over one sixth of the globe, but with Australia and New Zealand being the dominant developed countries.

The Journal comprises peer reviewed scientific reports, reviews, case reports of rare of unusual conditions, and perspective all of value for continuing professional development. Information for prospective authors, including author guidelines, publication ethics, malpractice statements and patient consent forms are available for download from the Australian & New Zealand Association Oral & Maxillofacial Surgery homepage. All correspondence with the Editor is via editorajoms@anzaoms.org.

Advertising Information

• Senior Consultant in Oral & Maxillofacial Surgery, Royal Perth Hospital • Clinical Associate Professor in Oral & Maxillofacial Surgery, The University of Western Australia

Andrew Heggie, AM

• Clinical Professor, Department of Paediatrics, The University of Melbourne • Senior Consultant Oral & Maxillofacial Surgeon, Royal Children’s Hospital of Melbourne Melbourne, Australia

David Wiesenfeld

• Honorary Clinical Professor, The University of Melbourne • Lead in Head & Neck Research & Education, The Victorian Comprehensive Cancer Centre Melbourne, Australia

Electronic Promotion Arun Chandu • Senior Consultant in Oral & Maxillofacial Surgery, Royal Dental Hospital of Melbourne • Clinical Associate Professor of Oral & Maxillofacial Surgery, The University of Melbourne Melbourne, Australia

Members Alexander Bobinskas • Consultant Oral & Maxillofacial Surgeon, The Canberra Hospital • Research Fellow, The John Curtin School of Medical Research Canberra, Australia

PRODUCTION MANAGEMENT Website and Distribution Belinda Mellowes • Executive Officer, Australian and New Zealand Association of Oral and Maxillofacial Surgeons Sydney, Australia

Production Editor Jac Taylor • Foundation Managing Director, Crucible Content Sydney, Australia

Nigel Johnson • Consultant Oral & Maxillofacial Surgeon, Princess Alexandra Hospital • Senior Lecturer in Oral & Maxillofacial Surgery, The University of Queensland Brisbane, Australia

Darryl Tong • Professor of Oral & Maxillofacial Surgery, The University of Otago • Senior Consultant in Oral & Maxillofacial Surgery, Dunedin Public Hospital Dunedin, New Zealand

The Australasian Journal of Oral & Maxillofacial Surgery accepts paid advertisements from companies involved with the surgical discipline.

Graphic Design Daniel Sim • Managing Director, Daniel Sim Design (DSD)

Jasvir Singh

For information on advertising guidelines and rates contact Ms Belinda Mellowes, Executive Officer, The Australian & New Zealand Association of Oral & Maxillofacial Surgeons: eo@anzaoms.org

Brisbane, Australia

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Catherine Offler

A D M I N I S T R AT I O N • Administrative Assistant to the Editor Adelaide, Australia

Disclaimer The Australasian Journal of Oral & Maxillofacial Surgery Editors and Editorial Board cannot be held responsible for error or consequence arising from the information contained in the Journal. The views and opinions expressed there do not necessarily reflect those of the Australian & New Zealand Association of Oral & Maxillofacial Surgeons, AJOMS Editor or Editorial Board. Neither does publication of advertisements constitute any endorsement of the products advertised. © Copyright 2024 ANZAOMS

Kathryn Steward • Advertising Sales and Administration - Membership and Administration Coordinator, Australian & New Zealand Association of Oral & Maxillofacial Surgeons Sydney, Australia

Ex Officio Members

Sydney Australia

Patrishia Bordbar • Immediate Past President, Australian & New New Zealand Association of Oral & Maxillofacial Surgeons • Consultant in Oral & Maxillofacial Surgery, The Royal Children’s Hospital of Melbourne • IAOMS Executive Representative – Oceania Region Melbourne, Australia

John Harrison • Regional Councillor for Oceania, International Association of Oral & Maxillofacial Surgery • Senior Consultant in Oral & Maxillofacial Surgery, Auckland City & Middlemore Hospital Auckland, New Zealand

Jocelyn Shand • Clinical Associate Professor, Department of Paediatrics, The University of Melbourne • Chair, Australian & New Zealand Association of Oral & Maxillofacial Surgeons Research & Education Foundation • Head of Section of Oral & Maxillofacial Surgery, The Royal Children’s Hospital of Melbourne • Vice President Elect, International Association of Oral & Maxillofacial Surgery Melbourne, Australia

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If ynotist, o a i a ed e ti (fo t. dCo y his es n not phle atio h a e be surg cern odo cti ■ M L m . Th eat ill, and our ad r is m ith lled pa an rom roo pra g on th L tly do oes pam form earc ut th ur p n) ll y r b Th am tr h A s tee o ca MS eth f o or c pros ical ferrin let in d t n o e i . x y o is e o ic e re o r b e d e v s is h O an ad th . Th ical re a er y, tion a med e re mp rec g t epti aspi e. T ergy icin -ray ibiot er als ZA m te lable eon) ed to t, i Re nce clin t su surg ques by l or to th is pa nly dru trac ing dicin y all med g X f, ant heth en t y AN isdo ava surg etain rt r no of ere e to ed enta po e th on. atm en l er en o ion we con clud r me d an other udin relie or w du u are ons ans ferr er d l re t. Us rge tre cons ou “W m”, facia f a r il y l in s, urg ertak scuss on. re th a or n r su ave a o ati to n o e c e s w i e h y o t ( l o n h l it h n n u d n . If e e t xil ova s , i pa ve nd di rge e lim ase bee t or eo eatm h yo to sign fully . s urg r tr it cou u hav iotic ent sia, dicin . is e le e ti lia ha y is u and al su not b ur . re ma rem p id s th to ca on w c t v on r ou stra to er ion aci ld on en yo antib reatm esthe r me layed ha hod you ut y tion u de you it rge yo Au ■ on ati 4, ort ner, abo sulta If yo ask read ur su ion urg rat llof ou ut ion to ect t , ana othe e de ati ur p r. 12 o rm d yo icke ,3 tio ctor con rm: will g, cis olar s nside maxi nd sh n abo opin b s y f f fo n e IC k d ld a tio n st i do ly in t fo on ignin , as ,V e in ou a the a gs an hou ell s e d oalve ul co and urs a erta cond s n sen rge th n n y o h rw n s n e re tio T nt ref ral yo t c se u in ind sig r y ou Co ur s Befo ues mb dru gery fill rem to Ca De er ca ur o n is re no ek a isto s y . yo . ny q 4, nt, ill nts sur l h em teeth x2 tie w tie form ve a aft th yo cisio ou a , se n. t Bo pa his pa ca bl ha ur T ir PO wi e de . 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orders@mitec.com.au ANZAOMS PREVIEW VERSION | 5

EDITORIAL One Small Step The Australian & New Zealand Society of Oral Surgery

to the meeting. So, with the various other speaker materials,

(ANZSOS) first considered the establishment of a Journal

we were able to photocopy and staple the Conference

around the time Neil Armstrong said those iconic words

booklet the night before commencement. Fortunately, there

in 1969. They came close in 1971, with the Organising

were only a few members then. At that meeting I became

Committee for the

new member number 31, and thus one could produce a

triennial meeting in

Conference booklet at short notice.

1971 (Figure 1). The lectures by the

Actually, my interest has always been related to intractable

principal guest Mr Ben

clinical problems. Although it feels good to report success,

Fickling, of England,

the real gain is when you develop methods to solve

were published in full,

previously unsolvable challenges. I was fortunate to have

as were the lectures

several first-class mentors plus others where, if one did the

by the Members. This

opposite, that put you on the right track! This all resulted

established the format

in many (300) peer-reviewed publications, editorial

for all future triennial,

experiences and a high personal H-score. I have always

then biennial, and finally

thought that research undertaken, but not peer reviewed, is

annual meetings. All

unethical. This includes Masters’ theses that are examined,

meetings produced

but not subsequently peer reviewed and published.

booklets of variable

Figure 1. 1971 Conference Booklet – note the Coat of Arms.

quantity and quality

In 2009, AZAOMS accorded me the privilege of taking on

depending on the skill

the role of Archivist from John Anker who had established

and enthusiasm of the

the existing collection. The plan was to produce a book

Organisers. Over time,

on the development of the speciality in our region. To do

they progressively

so, it was evident that professional help was needed, so I

became simpler abstract summaries, rather than full

approached Rob Linn, a Professional Historian. Together,

presentations of scientific articles.

we produced the coffee table book “Extractions to Reconstruction: The Development of Oral & Maxillofacial

The Australia and New Zealand Association of Oral and

Surgery in Australia and New Zealand” (Figure 2). Copies

Maxillofacial Surgeons (ANZAOMS), as ANZSOS has

were given to all members of ANZAOMS and a CD version

become, took a different approach in the late 1990’s, when

was given to trainees and all delegates at the International

it appointed John Anker as its archivist. John commenced working at gathering photos of ANZAOMS members and collecting all the above Conference booklets which displayed the ever increasing scope of Oral and Maxillofacial Surgery. Later, these booklets became part of the core collection held in the National Library of Australia in Canberra. I first became involved in the development of this scientific collection documenting our scope in 1973. At that time I had just been appointed Lecturer in Oral Surgery & Pathology at the University of Adelaide. At short notice I was asked to edit the 1973 ANZSOS Conference booklet. The Organisers had well set up the social program, but had paid less attention to the scientific program! Fortunately, the invited guest lecturer, Mr Norman Rowe, of England, was well organised and provided me the printed summaries of his four lectures prior

| ANZAOMS PREVIEW VERSION

Figure 2. “Extractions to Reconstruction”

Conference in Oral and Maxillofacial Surgery in Melbourne,

year plan. We then approached the ANZAOMS

2015. It was jointly funded by ANZAOMS and an

Council, under the leadership of Patrishia Bordbar, who

anonymous donor. The records collected are archived in the

courageously agreed to support a one year trial of the

National Library of Australia in Canberra, the only surgical

Australasian Journal of Oral & Maxillofacial Surgery. When

discipline to be so honoured.

successful it will continue annually.

Concurrent with these developments, the Australian

Please enjoy the product of our labours. I thank those who

Dental Journal commenced production of a series of

have submitted articles for consideration, and the Editorial

supplements based on

Board for their hard work and kind support. It is pleasing

the various disciplines

that the majority of papers presented are co-authored by

in Dentistry. In 2019,

trainees.

the supplement was “Contemporary Oral &

So, half a century after it was first conceived, the speciality

Maxillofacial Surgery”,

has its own journal.

edited by myself, with deputy editors Andrew

“One small step… becomes a great step for oral and maxillofacial surgery!”

Heggie and Paul Sambrook (Figure 3). Alastair Goss Research has always

Founding Editor

been a significant component of the Figure 3. Australian Dental Journal Special Edition 2019: “Contemporary Oral and Maxillofacial Surgery”

Training Units in Oral and Maxillofacial Surgery, with all

trainees being expected to show evidence of research experience. To complete the Fellowship in Oral and Maxillofacial Surgery at the Royal Australasian College of Dental Surgeons (FRACDS[OMS]), candidates are required to either complete a Masters degree thesis, or to have a peer review article published in a professional journal. In the last few years, candidates have been having increasing difficulty in having articles accepted for publication in international journals as they all have a high rejection rate.

Andrew Heggie and David Wiesenfeld Deputy Editors

Hence the need for a regional journal in the speciality arose once again. In 2020, Arun Chandu and I approached publishing houses regarding a print journal. They either failed to reply, or quoted a figure of $100k plus, recurrent per annum. So, we decided on a peer reviewed electronic closed-access journal. In 2022 I approached the Royal Australasian College of Dental Surgeons to resurrect the Annals of the College as a specific oral and maxillofacial surgery journal. After careful consideration, the College Directors declined as it was not part of their current five

ANZAOMS PREVIEW VERSION |

O N C O L O G Y INVITED REVIEW

PROTON BEAM THERAPY IN OROPHARYNGEAL CANCER A critical examination of efficacy and side effect mitigation with this new treatment modality

Marden N (BSc (Hons), MSc)†‡ Robinson A (BSc (Hons), MA)‡ Gorayski P (BSc (Hons), BMBS, FRACGP, FRANZCR)† §¶††

ABSTRACT The incidence of oropharyngeal squamous cell carcinomas (OPSCC) has increased, prompting a need for treatments that are effective yet minimally toxic. This paper examines proton beam therapy (PBT)

† ICON Cancer Centre Noarlunga, Noarlunga, South Australia, Australia ‡ Department of Allied Health Professions, Sheffield Hallam University, Sheffield, England §

within this context, assessing its targeted approach for treating OPSCC and its potential to spare adjacent critical structures, known as organs at risk (OAR). Addressing the persistent issue of long-term toxicities such as

Australian Bragg Centre for Proton Therapy

xerostomia and dysphagia associated with radical radiotherapy

Research, Adelaide, South Australia, Australia

remains a priority, as these conditions significantly impact survivors’

Royal Adelaide Hospital,

quality of life and present ongoing treatment challenges. Although

Adelaide, South Australia, Australia †† University of South Australia, Adelaide, South Australia, Australia

Corresponding Author:

progress has been made with Intensity-Modulated Radiation Therapy (IMRT) and Volumetric Modulated Arc Therapy (VMAT) in toxicity reduction, the comparative advantage of PBT in this regard warrants investigation. This review synthesises current research and emerging practices, providing evidence that PBT may offer a reduced risk of toxicity in

NICOLA MARDEN

sensitive areas like the salivary glands and oral cavity for certain

ICON Cancer Centre Noarlunga

patient groups. It also suggests employing normal tissue complication

Noarlunga, South Australia, Australia

probability (NTCP) models to quantify and potentially maximise this

Email: nikki.marden@icon.team

advantage. In our pursuit of balancing effective cancer eradication with maintenance of quality of life, this paper presents proton therapy as a promising option in managing OPSCC. It contributes to the field by promoting a nuanced discussion on optimising cancer care and supports a treatment paradigm that favours long-term health and patient wellbeing.

Keywords: Proton | Radiotherapy | Oropharynx | Squamous cell

| ANZAOMS PREVIEW VERSION

METHODS The review was conducted in accordance with the

The PRISMA method was used during data collection. A

‘Preferred Reporting Items for Systematic Reviews and

total of 224 articles were identified for screening, 114 were

Meta-Analyses’ (PRISMA) statement¹. While the project is a

removed as duplicates and 110 articles were screened

critical review rather than a systematic review, adapting the

via an abstract appraisal. An additional 50 articles were

PRISMA method allows for organised and structured data

excluded and four were unavailable as full papers. The

collection. As such, a systematic search took place then the

remaining 56 articles were read in full and the appropriate

information collated was critically analysed.

CASP checklist was applied to critically assess each article.

A literature search was conducted across multiple

Full text articles were read and screened, and any that did

databases, including PubMed, ScienceDirect, and the

not meet the inclusion criteria were excluded. Eligibility

Cochrane Library in November 2022 and subsequent

assessment was performed by a single reviewer. Following

forward citation tracing utilised Google Scholar and the

the segregated methodology, articles were categorised to

Sheffield Hallam University Library resources. The search

assist with the data synthesis. These included randomised

strategy incorporated the use of Boolean logic to construct

control trials (RCTs), comparative planning studies, quality

comprehensive search strings, with appropriate truncation

of life (QoL) research and NTCP modelling.

applied to select terms and phrase searching employed for queries consisting of multiple words. Given the uniform spelling of search terms in British, Australian, and American English, the use of wildcards was deemed unnecessary. The outputs of each search were systematically compiled and exported as CSV files to facilitate subsequent screening processes. The following is an example of the electronic search utilised for PubMed:

Randomised controlled trials As of the completion of this review, no randomised controlled trials for proton beam therapy and oropharyngeal cancer have been reported. Two ongoing trials are currently exploring this area: TORPEdO (Toxicity Reduction using Proton bEam therapy for Oropharyngeal cancer) and Trial NCT01893307, which compares intensitymodulated proton beam therapy (IMPT) with intensity-

(“Oropharynx SCC” or “oropharyngeal cancer*” or “head and neck SCC”)

modulated photon therapy (IMRT) for the treatment of oropharyngeal cancer of the head and neck. Both trials share the primary objective of assessing the late treatment toxicities of IMPT versus IMRT in patients with locally advanced OPSCC.

AND

TORPEdO, the UK’s inaugural proton clinical trial, is a multi-centre Phase III trial. Its focus is to evaluate the late

(proton or PBT or IMPT)

treatment toxicities of IMPT versus IMRT in patients with locally advanced OPSCC requiring concurrent radiotherapy and chemotherapy. The assessment of primary objectives

AND (radiotherapy or photon or IMRT or VMAT)

will utilise a combination of patient-reported outcome measures, specifically the University of Washington physical toxicity score, along with feeding tube dependence and severe weight loss, 12 months post-treatment. Patients in the IMPT arm will receive treatment at either the Christies Hospital in Manchester or UCL in London,

AND

while those in the IMRT arm will be treated at the referring hospital. Recruitment for this trial began in January 2020, with completion expected by October 2023. Secondary

(xerostomia or dysphagia or QoL*)

objectives include validating a biomarker (NTCP model) as a predictor of benefit from IMPT versus IMRT and estimating the cost-effectiveness of IMPT versus IMRT for OPSCC in the UK. ANZAOMS PREVIEW VERSION |

Trial NCT01893307 is a multi-centre Phase II/III study

While lower OAR doses were reported by Holliday et al.

currently being led by the University of Texas MD

(2016) the parotid and submandibular glands did not show

Anderson Cancer Centre which compares the incidence

significant differences between the three groups potentially

and severity of chronic grade 3–5 toxicity for patients with

suggesting that xerostomia occurrence and severity may

advanced oropharyngeal cancer treated with IMPT or

not change between IMPT and IMRT cohorts. 5 Oral cavity

IMRT. Completion is estimated for 2025 with 22 recruiting

dose was reduced significantly for the IMPT/IMPT cohort

hospitals within the USA. Patients are randomised to either

suggesting increased normal tissue sparing with IMPT.

the IMRT arm or IMPT arm with both undergoing 6.5 weeks

Wright et al. (2021) also reported reduced oral cavity dose

of treatment, five days per week (Monday to Friday). After

with proton therapy as well as reduced dose to the organs

completion of treatment patients will be followed up every

associated with both xerostomia and dysphagia for the

three months for one year, every four months for the second

proton therapy patient group.6 Figure 1 demonstrates the

year, and then every six months for a further five years. It is

general dose distribution differences between PBT and

anticipated that the results of the trial will help clarify the

IMRT plans. With proton beams it is possible to modulate

extent to which IMPT can reduce toxicity for patients with

the beam to deposit the dose at the required depth using

oropharyngeal cancer. The final phase of the study will be

the Bragg peak, achieving significant dose reduction and

to compare the rate of tree-year progression-free survival

allowing for greater normal tissue sparing, in this instance

between concurrent chemo-radiation strategies with

reducing dose to the oral cavity which affects both

IMRT and IMPT following the treatment of oropharyngeal

xerostomia and dysphagia.

tumours. The same group of patients were followed up post

Comparative planning studies In the absence of published RCTs, comparative studies serve as the primary source to draw inferences regarding

treatment (mean follow time of 20.4 months, range 6-50 months) and the high rates of local control were demonstrated (97%) suggesting that the increased tissue sparing did not affect the disease outcomes.6

treatment efficacy. Table 1 presents a summary of the reviewed comparative planning studies, which collectively suggest that PBT has the capacity to minimise radiation dose to non-targeted tissues while maintaining adequate coverage of the targeted oncologic volume. Simone et al. (2011) demonstrated a more favourable dosimetric profile with IMPT and there was a larger potential for OAR dose reduction while maintaining the PTV dose.2 Both the IMPT and adaptive IMPT proton plans had superior conformity than either IMRT plans or adaptive IMRT plans and delivered less dose outside of the target volumes. This would indicate that an IMPT plan would be most appropriate at reducing xerostomia by ensuring that the contralateral parotid dose is below 20 Gy and the larynx dose below 44 Gy as per the dose constraint recommendations. 3 Romesser et al. (2016) found whilst there is a reduction in dose with PBT, the IMRT plans are also well within the dose guidelines for parotid sparing and laryngeal dose reduction and should be low enough to reduce any radiation induced xerostomia and dysphagia; however, lower Grade I acute dysphagia was reported by the PBT cohort whilst xerostomia was not reported on.4

Figure 1 Dose distributions comparing PRT (A) with the treatment-approved rapid arc plan (B) of a Seventy-year-old male with a stage I (pT1pN1cM0) HPV-associated right palatine tonsil squamous cell carcinoma. Mean dose to the oral cavity was 8.5 Gy and 23.5 Gy for PRT and rapid arc plans, respectively. Reproduced from Wright et al. (2022)6

10 | ANZAOMS PREVIEW VERSION

Year

Author

Method

Sample size

Results

2016

Apinorasethkul, et al.7

Comparative planning study

7 – stage Iva, HPV+ oropharyngeal SCC

OAR

Photon

Proton

Constrictor (meancGy)

3661

3683

Larynx (meancGy)

2390

2637

Mandible-PTV (maxcGy)

6049

6081

Oral cavity (meancGy)

1771

293

Contra parotid (meancGy)

1796

1358

Contra submandibular (mean cGy)

3608

3251

OAR

VMAT

MFO3

MF05

MF07

SFO

Ipsilateral parotid (mean Gy)

25.0

17.5

16.9

16.7

21.3

Cont parotid (mean Gy)

19.2

10.3

9.7

9.2

14.0

Cont SM gland (mean Gy)

28.9

21.9

20.5

20.1

34.9

Composite salivary (mean Gy)

23.0

14.6

13.8

13.5

20.0

Composite swallowing (mean Gy)

25.3

16.8

16.5

16.4

23.7

OAR

Photon

Proton

Mandible Max dose Gy (D0.1 %)

71.2

70.1

Larynx V50Gy (%)

30.6

22.3

Parotid glands Mean dose Gy

27.1

25.9

SMG Mean dose Gy

58.0

61.6

OAR

IMPT/IMPT

IMPT/IMRT

IMRT/IMRT

Anterior oral cavity (mean Gy)

8.3

31.0

30.5

Posterior oral cavity (mean Gy)

40.5

54.3

50.6

Lt. parotid gland (mean Gy)

32.0

25.8

39.6

Lt. SMG (mean Gy)

43.9

53.7

61.0

Rt. parotid gland (mean Gy)

31.6

31.7

25.0

Rt. SMG (mean Gy)

51.3

58.7

50.5

IPC (mean Gy)

32.8

45.6

28.8

MPC (mean Gy)

48.2

57.0

54.6

SPC (mean Gy)

55.3

58.1

58.0

OAR

IMRT

Proton

Cont SMG Dmean (cGy)

638.7

4.3

Cont parotid gland Dmean (cGy)

533.3

48.5

Oral cavity Dmean (cGy)

1760.4

458.9

Larynx Dmean (cGy)

2605.3

1642.2

23 – IMRT

OAR

PRT: Mean Gy

IMRT: Mean Gy

18 – PBRT

Larynx

10.3

21.4

Oral Cavity

0.94

20.6

Contralateral Parotid

0.00

1.4

Contralateral SMG

0.00

4.1

OAR

IMPT

aIMPT

IMRT

aIMRT

Ips parotid (meanGy)

32.9

29.8

43.1

39.0

Cont parotid (meanGy)

19.5

18.3

26.8

25.3

Larynx (meanGy)

35.3

31.0

45.4

41.8

OAR

PRT: Mean dose Gy

IMRT: Mean dose Gy

Constrictors

29.6

37.3

Larynx

21.6

27.4

Mandible maximum

60.5

62.6

Oral Cavity

5.0

20.5

Ipsilateral parotid

24.8

30.8

Cont Parotid

12.2

18.1

Cont Submandibular

28.7

31.0

2015

2020

2016

2013

2016

2011

2022

Barten, et al.27

Håkansson, et al.10

Holliday, et al.5

Kandula, et al.8

Romesser, et al.4

Simone, et al.2

Wright, et al.6

Comparative planning study

Retrospective planning study

Case matched control analysis

Comparative planning study

Retrospective comparative planning study

4-pronged comparative planning study

Retrospective comparative study

25 pair matched (50 pts)

Table 1 Summary of the reviewed comparative planning studies

ANZAOMS PREVIEW VERSION |

Apinorasethkul et al. (2016) noted that the mean of the

Long term toxicities And QoL

average integral dose was 9.1% lower for the proton plans compared to the photon plans. Although not all OAR doses

While it is accepted that patient reported outcomes are the

were reduced, the lower integral dose and xerostomia

best way to measure QoL and treatment side effects, there

related organs dose support proton therapy as a method to

are currently no universally accepted measures.11

improve QOL and decrease short and long term morbidity.7 When using the rate of feeding tube dependency and/or Kandula et al. (2013) reported dose reduction with proton

severe weight loss as a measure Blanchard et al. (2016)

therapy for all reported on OAR; however, the extent of the

examined 100 patients treated with IMRT and 50 patients

dose reduction varied greatly between patients, with the

treated with IMPT and concluded that IMPT shows lowers

contralateral parotid gland receiving a dose range of 358.9

rates of morbidity than IMRT while providing similar rates

to 707.1 cGy for IMRT and 0.4 to 192.3 cGy with protons

of tumour control.12 In a study by Cao et al. (2021) of

and the larynx receiving a dose range of 1950.1 to 3505.2

103 IMPT and 429 IMRT oropharyngeal cancer patients,

cGy for IMRT and 162.0 to 2366.9 cGy with protons. 8 These

similar rates of moderate-severe xerostomia were reported

results suggest that the benefits of proton therapy OAR

up to 18 months after treatment via an eight-item self-

dose reduction are patient specific and not all patients

reported xerostomia-specific questionnaire.13 At 18-24

would have the same dosimetric gains.

months the rates reported in the IMPT cohort dropped to 6% compared to 20% for the IMRT cohort and these

However, what needs to be considered with PBT is the

results were maintained at the 24-36 month follow up

increased level of uncertainty, they are not as robust as

questionnaire (6% and 20% respectively). It was noted that

photon radiotherapy plans. The main aim of radiotherapy

parotid gland dose/volume exposure was similar for both

treatment is to deliver the desired dose to the target volume

IMRT and IMPT patients; however, fewer IMPT patients

while minimising the dose to surrounding healthy tissue.

reported late xerostomia compared to the IMRT cohort. The

Treatment plan robustness comes from checking the

dose delivered to the oral cavity and submandibular glands

dosimetric effects of various uncertainties in a logical way.

was lower in the IMPT cohort, supporting the idea that late

Barten et al. (2015) examined three planning techniques

xerostomia is linked to the oral cavity and submandibular

(MFO, SFO and VMAT), and reported the MFO beam

gland dose not just the parotid dose.

arrangements would be superior in reducing dosage to the swallowing organs compared

These results were supported by Sharma et al. (2018) in

to SFO and VMAT; however, the MFO PBT plans were

a study of 33 patients treated with VMAT and 31 treated

accepted by the author to be the least robust of the three

with PBT. Patients treated with PBT had significantly

planning techniques.

less radiation to normal structures than the VMAT

cohort and better scores in QoL questionnaires.14 Both Proton beams are known to be more susceptible to tissue

groups consisted of patients of similar age, site, stage

changes in the beam path and the MFO 3f plans produced

and treatment dose. The European Organization for

CTVb 95% coverage ranging from 79-99%, highlighting

Research and Therapy of Cancer QLQ-30 Version 3 was

these issues. When reviewing just the VMAT and SFO

used and patient-reported QoL questionnaires were

plans, due to the similar reported robustness, the OAR

prospectively collected at initial treatment consultation

mean doses reported are not significantly different and

and at three, six, and 12 month follow up intervals.

possibly not clinically significant. Both contralateral parotid

Significantly less xerostomia was reported at both six

doses were below 20 Gy and the composite swallowing

and 12 month timepoints which could be attributed to

organ doses are 25.3 Gy and 23.7 Gy, significantly below

the dose differences. It should also be noted that at the 3

the 44 Gy QUANTEC guidelines. Hakansson et al. (2020)

month follow up xerostomia was nearly equally reported

also found the photon plans to be more robust than proton

by both patient cohorts. The mean contralateral parotid

plans and, while maintaining to CTV coverage, little

dose reported was 20.77 Gy (VMAT) and 11.90 Gy (PBT),

difference was recorded in the dose to the OAR structures

the mean contralateral submandibular doses were 30.01

in close proximity to the target volume, however proton

Gy and 28.75 Gy respectively and the oral cavity mean

plans demonstrated generally lower dose to remaining OAR

dose was 35.11 Gy and 21.23 Gy respectively. This again

and normal tissue when compared with the photon plans.10

supports the theory that overall oral cavity dose may have an impact on long term radiation induced toxicities.

12 | ANZAOMS PREVIEW VERSION

Additionally, Soi et al. (2016) noted that IMPT patients may

then an estimation of radiation toxicities is compiled from

have a more rapid return to normal function during the first

the dose distribution and OAR data for each plan. These

three months after treatment.15 No differences in acute and

are compared and a ∆NTCP is computed to establish if a

chronic phase symptom burden were detected between

PBT is advantageous for an individual. Modelling systems

both groups of 35 IMPT and 46 IMRT patients, and

allow patients who will benefit most to proceed with PBT

subacute recovery phase (three months post treatment)

and patients who would not continue with traditional IMRT

patient reported symptom burden was significantly lower

treatment. As a plan is assessed there are

with IMPT. This study utilised MD Anderson Symptom

multiple triggers for a comparative proton plan and if

Inventory-Head and Neck Module Survey which reports

expected toxicities are below the calculated thresholds

distress and changes in a patient’s condition.

the patient proceeds with a photon plan. Xerostomia, dysphagia, and tube dependency were the three main measuring tools for model-based selection and the CTCAE

Normal tissue complication probability models

guidelines. Figure 2 is an example of the flowchart used in the proton selection process.

RCTs are widely recognised as the gold standard for evaluating the efficacy of various radiotherapy modalities.16

According to the prevailing clinical commissioning

However, the implementation of RCTs must consider

guidelines in the United Kingdom, adults diagnosed with

both financial considerations and ethical constraints.

squamous cell carcinoma of the oropharynx are currently

Central to the ethical framework of RCTs is the concept

precluded from receiving PBT as specified in the clinical

of clinical equipoise, defined as a state of uncertainty

commissioning policy statement for PBT in the treatment

or disagreement among medical professionals about

of adult head and neck cancers.21 Before the inauguration

the optimal treatment choice for a particular patient or treatment site.17 This principle of clinical equipoise serves as the ethical foundation for RCTs, reinforcing the notion that no single treatment method should be presumed superior, thereby justifying the conduct of clinical trials. Sheehan et al. (2014) have posited that a substantive debate exists regarding the research necessary for PBT to become a preferred treatment option in various cancer

Patient meets general eligibility criteria for proton therapy: Primary tumor originating in the pharynx, larynx or oral cavity No distant metastases Curative intent Plan comparison indicated

YES

Generate PROTON plan

Generate PHOTON plan

care scenarios.18 Unlike a placebo, PBT represents an evolving technology within the field of radiotherapy. It is well-established that excessive doses to normal tissue can lead to significant patient side effects, and therefore, a practicable effort to minimise the dose to OAR during the planning process should be considered. Research has shown that PBT, through the utilisation of the Bragg peak,

NTCP max -photons for dysphagia grade ≥ II >10%

YES

NO NTCP max -photons for moderateto-severe xerostomia >10%

ΔNTCP for dysphagia graad ≥ II ≥10%

YES

ΔNTCP for moderate-to-severe xerostomia ≥10%

YES

possesses the capability to reduce normal tissue dose.1,10,12 Consequently, the question arises: ‘If there is a consensus among medical professionals about the best treatment option, is there still a need for RCTs before administering the treatment?’ In the Netherlands a National Indication Protocol Proton therapy for head and neck cancer patients (NIPP-HNC)

ΣNTCP max -photons for dyshagia + xerostomia >10%

YES

NO NTCP max -photons for tube feeding dependence >5%

ΣΔNTCP for dyshagia + xerostomia ≥15%

YES

ΔNTCP for tube feeding dependence ≥5%

PHOTON THERAPY

PROTON THERAPY

YES

has been created to identify which head and neck patients would benefit most from proton therapy over photon therapy using a model-based approach. NTCP models are used to describe the relationship between the dose delivered to a particular OAR and the anticipated risk of a radiation-induced side effect. All new treatment modalities require level I-II scientific evidence for reimbursement by healthcare insurance companies. RCTs are required for this level of data. An NTCP model has been created as an alternative but not to replace RCTs.19 A comparison

Figure 2 Flowchart for selecting patients for a plan comparison and PBT, respectively. Reproduced from Langendijk et al. (2021)20

between the optimal IMRT and PBT plans is completed and ANZAOMS PREVIEW VERSION |

of two proton facilities, the Christie NHS Foundation Trust

There is no single generic NTCP model followed by all

in 2018 and University College London Hospital in 2020,

departments. Rwigema et al. (2019) created models and

the NHS provided funding for suitable patients to receive

applied them to the patient cohort who had received PBT

PBT overseas. Once both centres are fully operational, it

and the treatment approved photon back up plans.26

is projected that each will have the capacity to treat up to

Comparisons were made between the predicted and the

750 patients annually.22 This increase in available resources

recorded outcomes with the PBT cohort serving as an

raises the question of whether probability models could

internal control group. The results demonstrated a reduced

be employed to ensure that patients who would derive the greatest benefit are prioritised for PBT treatment. Conversely, the provision of proton therapy in Australia, representative of the southern hemisphere’s healthcare landscape, entails distinct logistical complexities. Australian candidates for proton therapy are necessitated to seek treatment internationally via the Medical Treatment Overseas Program (MTOP), with funding facilitated by Medicare. Opting for private proton therapy outside domestic borders remains a possibility, albeit with considerable financial implications for the patients. Between 1998 and 2016, a total of 68 Australian residents,

risk of complications for the PBT cohort with the greatest differences being reported for grade 2 dysphagia and grade 2 xerostomia. It was found that the model overpredicted the rate of xerostomia for the PBT group compared to the recorded outcomes suggesting that the results need to be validated, perhaps with a large patient cohort.

LIMITATIONS OF THE STUDY

predominantly paediatric cases (72% of approvals), received authorisation for such overseas interventions through MTOP.23 It is of particular note that during this

Researcher bias

period no approvals were granted for patients with

The entire search and screening process was conducted by

oropharyngeal carcinoma.

a single researcher, which may have introduced bias into the selection of studies for inclusion. Ideally, the involvement

Tambas et al. (2020) used the thresholds defined in Dutch

of more than one researcher would have enhanced the

National Indication Protocol to complete a prospective

impartiality and robustness of the selection process..

cohort study of 277 patients. Optimal VMAT plans were 24

created for all patients and compared with a model based

Geographical concentration

optimised IMPT plan using NTCP models of NIPP-HNC. Of

The limited number and geographical distribution of proton

the 277 patients, 141 qualified for comparative planning. 80

therapy centres worldwide have resulted in a concentration

patients qualified for IMPT and the remaining patients were

of studies from only two regions. Specifically, six out of 18

treated with the VMAT plans created for the comparison.

studies had a contributor from MD Anderson Cancer Centre

In the group who qualified for IMPT dose to the oral cavity,

in Houston, Texas, and overall, 12 out of 18 studies were

PCM inferior, supraglottic area and cricopharyngeal muscle

based in the USA, while the remaining 6 were based in

was lower when compared to the non-qualifying group

Europe. This geographical limitation may have introduced

which are connected to dysphagia-related toxicities.

bias, and a more diverse range of data from various global sources would have been preferable to ensure a more

It was noted by Tambas et al. (2020) that the selection

comprehensive and unbiased perspective. As of January

rate was higher among patients with advanced disease,

2023, all operational PBT facilities are in the Northern

pharyngeal tumours, and/or baseline complaints.24 This

Hemisphere, split between Europe, North America and

study deduced that it was clinically feasible to use model-

Asia.

based selection for head and neck cancers. This was also supported by Behrends et al. (2021) who using the same

Small patient cohorts

NTCP model and deduced that one third of the sampled

Some of the included studies were characterised by

patients would qualify for PBT, with the most notable

small patient cohorts, which raises concern about the

advantage being in the mid-to-low doses, especially for

representativeness of these samples for the entire patient

dysphagia and tube feeding.25 A follow-up clinical study is

group. The limited size of these cohorts makes it challenging

planned to further investigate the comparison of proton and

to generalise the findings, and further research with larger

photon RT and to validate the NTCP-models for VMAT and

and more diverse patient populations is necessary to

IMPT.

validate the conclusions drawn from these studies.

14 | ANZAOMS PREVIEW VERSION

CONCLUSION PBT offers an optimistic advance in radiotherapy by optimising the balance between effective tumour control

At the forefront of advancing PBT in Australia is the

and minimising exposure to adjacent OAR. Current

construction of The Australian Bragg Centre for Proton

evidence indicates that a comprehensive reduction in

Therapy and Research in Adelaide. The centre is poised

radiation dosage to the oral cavity, rather than targeting

to navigate the intricate eligibility criteria for state-funded

only specific OARs, may alleviate persistent side effects

proton therapy in Australia and is proposed to focus on

like xerostomia and dysphagia. However, broader and more

the needs of paediatric and selected adult patient groups,

inclusive research, particularly with larger cohorts, is pivotal

with an exclusion of oropharyngeal cancer cases in the

to substantiate these findings across the broader spectrum

initial phase. Mandatory comparative planning for all

of patients with OPSCC.

cases is envisioned to verify the role of PBT in toxicity reduction across various patient demographics. This

With PBT still in the nascent stages of availability as of

prudent approach reflects a commitment to leveraging PBT

2023, its integration into routine clinical practice is cautious

capabilities to their fullest within the calculated constraints

and selective. The deployment of NTCP models emerges

of current healthcare frameworks.

as a significant stride towards refining patient selection, highlighting those who stand to gain the most from PBT

Conflict of interest statement

in a landscape where it remains an exclusive treatment The author reports no potential conflicts of interest.

modality.

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Behrends C, Haussmann J, Kramer P-, et al. Model-based comparison of organ at risk protection between VMAT and robustly optimised IMPT plans. Zeitschrift fur Medizinische Physik 2021; 31(1): 5-15.

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Rwigema JM, Langendijk JA, Paul van der Laan H, et al. A model-based approach to predict short-term toxicity benefits with proton therapy for oropharyngeal cancer. Int J Radiat Oncol Biol Phys 2019; 104(3): 553-562.

27.

Barten DLJ, Tol JP, Dahele MR, Slotman BJ, Verbakel WFAR. Comparison of organ-at-risk sparing and plan robustness for spot-scanning proton therapy and volumetric modulated arc photon therapy in head-and-neck cancer. Med Phys 2015; 42(11): 6589-6598.

16 | ANZAOMS PREVIEW VERSION

O N C O L O G Y SCIENTIFIC ARTICLE

METASTATIC SQUAMOUS CELL CARCINOMA OF THE SALIVARY GLAND Treatment and prognostic factors related to overall survival and recurrence ABSTRACT

McKenzie J (BSc, BDS)† Simpson E (BDS)‡ Maher H (BDS)†

Purpose:

Lockyer J (BDS, MBChB)§ Singh T (BDS, MBChB, MPhil, FRACDS (OMS))

Nguyen E (BDSc (Hons), MBBS (Hons), FRACDS (OMS))††

† U niversity of Otago, Christchurch, New Zealand ‡ C hristchurch Hospital, Christchurch, New Zealand §

estmead Hospital, W Sydney, Australia

aikato Hospital, W Hamilton, New Zealand

†† Austin and Monash Health, Melbourne, Australia

This study aims to address treatment and prognostic factors related to metastatic salivary gland squamous cell carcinoma (SCC) overall survival and recurrence in a New Zealand hospital. Methods: 10-year retrospective case series of patients with surgical management of metastatic salivary gland SCC. Data was collected on patient demographics, treatment and outcomes. Results: 101 patients were diagnosed with metastatic SCC of the salivary glands, occurring primarily in the parotid (94%), in elderly (median 84 years) Caucasian (92%) males (72%). All patients were treated with parotidectomy with high rates of neck dissection (80%) and adjuvant radiation therapy (83%). Histology showed perineural invasion (PNI)

Corresponding Author: JAMIE McKENZIE University of Otago, Christchurch, New Zealand Email: jamie.w.mckenzie@gmail.com

(28%), cervical metastasis (53%) and extranodal extension (ENE) (28%) were common. Overall survival was reduced in patients with cervical metastasis, lymphovascular invasion (LVI) and PNI, while PNI was associated with disease recurrent and cervical node metastasis. Conclusion: This research provides insight into the high rates of metastatic salivary gland SCC in New Zealand. SCC of the salivary glands is an aggressive entity, with lower rates of survival related to PNI, LVI and cervical metastasis. Conservative surgical margins and adjuvant radiation therapy provide adequate oncological management, with reduced morbidity. While primary radiation therapy in cN0 patients may provide a potential alternative treatment modality.

Keywords: salivary gland | squamous cell carcinoma ANZAOMS PREVIEW VERSION |

INTRODUCTION

Squamous cell carcinoma (SCC) of the salivary gland is rare, accounting for 1 to 5% of all salivary gland tumours.1-3 SCC of the salivary glands is primarily metastatic involving intraparotid and periparotid lymph nodes, secondary to non-melanoma skin cancer (NMSC).1,4,5 Five percent of NMSC metastasise, and in the head and neck the parotid gland acts as a metastatic basin providing first echelon lymph node drainage from the scalp, forehead and midface, which experience high rates of ultraviolet radiation.6 In advanced disease, metastasis spreads to the cervical lymph nodes.7,8 Australasia has the highest rates of NMSC in the world, with SCC being the most common metastatic disease of the salivary glands, occurring primarily in Caucasian males in their eighth decade. 9,10 SCC of the submandibular gland account for around 20% of patients, with a heterogeneous etiology from lymph nodes draining both cutaneously and intraorally.10,11 Primary SCC of the salivary glands is rare (<1%), with non-specific histological variation, absence of definitive pathogenesis, and often misdiagnosed metastatic SCC, or a diagnosis of clinical exclusion.2,3 Metastatic SCC of the salivary gland is aggressive, with 30-50% having cervical node involvement, a five-year survival of 54-64% and a recurrence rate of 2060%.2,3,6,8,9 There is a paucity of information relating to the management and prognostic factors of metastatic SCC of the salivary glands. Current guidelines do not address resection margins in metastatic disease, and it remains unknown whether margins surrounding lymph node disease influence survival. However, the National Comprehensive Cancer Network (NCCN) and National Institute of Health Care Excellence (NICE) recommend primary surgery with neck dissection, and adjuvant radiation therapy.12,13 Following our previous paper Mckenzie et al. (2022), which identified high rates of metastatic SCC in salivary glands in New Zealand, this paper aims to document the experience in the management of metastatic salivary gland SCC and identify prognostic factors in a New Zealand population via a retrospective case series.10

18 | ANZAOMS PREVIEW VERSION

METHODS Ethics consent was obtained by the New Zealand Health

Analysis of primary cutaneous tumour characteristics were

and Disability Commission and approved by the Waikato

not undertaken as this information is not readily available

District Health Board. Data was collected between January

in New Zealand. This is due to treatment with topical

2010 to December 2020. Patients were included if they had

chemotherapeutic agents, and the absence of mandatory

a histological diagnosis of metastatic SCC of the salivary

histological reporting for cutaneous SCC. Furthermore,

glands (WHO classification) that was primarily surgically

histological features such as primary tumour depth of

managed.14 All patients were presented at a specialist

invasion, grade, PNI or LVI are not routinely reported for

head and neck multidisciplinary meeting (MDM) for shared

cutaneous SCC, limiting prognostication of salivary gland

treatment planning.

metastasis from primary cutaneous SCC. Within our study cohort histological reporting did not indicate any cases of

The definition of margins, beyond nodal disease is not

primary SCC of the salivary glands.

widely reported in the literature. We defined an involved margin as less than 1mm, a clear margin as greater than

Statistical analysis was completed with SPSS (Version 26.0,

5mm and a close margin as greater than 1mm and less

IBM, Somers, USA). Medians and interquartile ranges were

than 5mm, as suggested by other authors.15,16 Tumour

used for non-parametric data. Chi-squared (Chi-sq) tests

excision was based on clinical assessment of facial

were used to assess disease characteristics. Kaplan Meier

nerve, pre-operative imaging, intra-operative findings

analysis and log-rank tests were used to assess univariate

and surgeon experience with preservation of the facial

factors for survival. Close and clear margins were combined

nerve if indicated. These margin definitions were selected

for Kaplan-Meier analysis due to the small number of

to achieve consistency with reporting, and aligns with

patients with clear margins. Kaplan Meier analysis and

literature findings that more radical surgery does not

log-rank tests were used to assess univariate factors for

improve disease free survival.

survival. Cox regression was used for multivariate analysis.

7,8,17,18

A p-value of 0.05 was statistically significant.

RESULTS A total of 101 patients were diagnosed with metastatic

Eleven (11/101, 11%) patients underwent surgery

SCC of the salivary glands. The median age of patients

alone, with six (6/101, 6%) patients receiving adjuvant

was 84 years (IQR: 71-97 years). Eighty-three (83/101,

chemoradiation therapy, and 84 (84/101, 83%) patients

72%) patients were male with a median age of 82 years

receiving adjuvant radiation therapy. Patients who

(IQR 70-94 years), with 18 (18/101, 18%) being female

underwent parotidectomy but did not undergo neck

with a median age of 91 years (IQR 79-103 years) (p=0.

dissection, were treated with adjuvant radiation therapy,

04, Chi-sq). Ninety-three (93/101, 92%) patients were

none had recurrence. Seventy-five percent (15/20) of

Caucasian, with three (3/101, 3%) being Māori and five

patients who did not undergo neck dissection were

(5/101, 5%) other European. Ninety-five (95/101, 94%)

misdiagnosed on fine needle aspirate, and were cN0.

tumours occurred in the parotid gland, with six (6/101, 6%)

Histologically the majority of patients had positive cervical

occurring in the submandibular gland. Of the patients with

lymph nodes (54/101, 53%). Occult nodal disease was

SCC of the submandibular gland three (3/6, 50%) of these

identified in 28 (28/101, 28%) patients. Twenty-eight

patients were female, and two (2/6, 33%) were Māori, with

(28/101, 28%) patients had extranodal extension (ENE),

a median age of 63 years (IQR = 59-68 years), which was

while 15 (15%) and 28 (28%) patients were positive for

statistically younger (p=0.001) than patients with SCC of

lymphovascular infiltration (LVI) and perineural invasion

the parotid gland (median = 83 years, IQR = 83-86 years).

(PNI), respectively. For patients with submandibular SCC, six (6/6, 100%) had neck dissection with four (4/6, 67%) having positive cervical lymph nodes, with two (2/6, 33%) patients having ENE and PNI, respectively.

ANZAOMS PREVIEW VERSION |

Surgery Surgery + PORT

Involved margins were found in 51 (51/101, 51%) Treatment Modality

patients, 41 (41/101, 41%) patients had close margins 1.0

and nine (9/101, 9%) had clear margins. Of the patients with involved margins 40 (40/51, 78%) had adjuvant chemoradiation therapy. Of patients with involved margins 40 (40/51, 78%) had neck dissection, with 28 (28/51, 55%) having positive nodal disease and 15 (15/28, 54%) having ENE.

0.8

Cumulative Survival

radiation therapy and four (4/51, eight%) had adjuvant

0.6

0.4

0.2

Eight (8/101, 8%) patients had recurrence. Of patients

0.0

with recurrence, five (7/8, 88%) had involved margins, five

(5/8, 63%) had PNI and four (4/8, 50%) had ENE and nodal

100

150

200

Overall Survival Months

Graph 1 Treatment modality Kaplan-Meier survival. Surgery median survival 14 months (IQR = 3-83 and surgery with radiation therapy median survival 75 months (IQR = 20-130 months). (p=0.02).

positivity, respectively. The median time of survival for all patients was 35 months (IQR = 9-68 months), this was reduced in patients with recurrence (25 months, IQR 4-45 months), but was not

Ipsilateral

statistically significant (p=0.24). While patients had an

None

overall five-year survival of 53%. Parotid gland SCC

Neck Dissection

median survival was 32 months (IQR = 19-48 months)

1.0

compared to the submandibular gland of 44 months (IQR = 39-114), but this was not statistically significant (p=0.16,

Kaplan-Meier analysis showed overall survival was reduced in patients with cervical metastasis (p=0.04) (Graph 4), LVI (p<0.001) (Graph 6) and PNI (p<0.001)

0.8

Cumulative Survival

Chi-sq).

(Graph 7). Survival benefit was found in patients treated

0.6

0.4

0.2

with adjuvant radiation therapy (Graph 1). No statistically 0.0

significant differences were identified for margins, neck

dissection or ENE (Graphs 2-3, 5).

100

150

200

Overall Survival Months

Graph 2 Neck dissection Kaplan-Meier survival. Ipsilateral neck dissection median survival 35 months (IQR = 11-72 months) and no neck dissection median survival 21 months (IQR = 5-58) (p=0.33).

Cox regression for overall survival showed worse outcomes for patients with PNI (HR = 2.5, p=<0.01) and LVI (HR = 2.3, p=0.04), but not ENE (HR = 1.0, p=0.9), margins (involved HR = 1.1 p=0.3; close HR = 1.2 p=0. 8), or cervical metastasis (HR = 1.1, p=0.7). For disease recurrence only

Close/Clear Involved

PNI was statistically significant (HR 2.1, p=0.03), with Margins

margins (involved HR = 0.8, p=0.6; close = 0.58, p=0.1), 1.0

ENE (HR = 1.1, p=0.7) LVI (HR = 1.3, p=0.5), and nodal positivity (HR = 1.1, p=0.8) not reaching significance. While only factor statistically significant.

0.8

Cumulative Survival

for cervical node metastasis PNI (HR 1.9, p=0.04) was the

0.6

0.4

0.2

0.0 0

100

150

200

Overall Survival Months

Graph 3 Margins Kaplan-Meier survival. Involved margin median survival was 23 months (IQR = 7-65 months) with close/clear margins having a median survival of 83 months (IQR = 29-136 months) (p=0.12).

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Negative

Positive

Negative

Lymph Vascular Infiltration 1.0

0.8

Cumulative Survival

LymphNodes 1.0

0.6

0.4

0.6

0.4

0.2

0.0

0.0 0

100

150

200

100

150

200

Overall Survival Months

Graph 4 Cervical lymph node positivity Kaplan-Meier survival. Node positive median survival 22 months (IQR = 5-65) vs. node negative median survival 46 months (IQR = 15-83) (p=0.04).

Graph 6 Lymphovascular infiltration (LVI) positivity Kaplan-Meier survival. LVI positive median survival 11 months (IQR = 4 -19 months), vs LVI negative median survival 45 months (IQR = 14-75) (p=<0.001).

Positive

Negative

Perineural Invasion 1.0

0.8

Cumulative Survival

Extranodal Extension 1.0

0.6

0.4

0.2

0.6

0.4

0.2

0.0

0.0 0

100

150

200

Overall Survival Months

100

150

200

Overall Survival Months

Graph 5 Extranodal extension (ENE) positivity Kaplan-Meier survival. ENE positive median survival 24 months (IQR = 6-47 months) vs extranodal extension negative median survival 44 months (IQR = 11-75 months) (p=0.11).

Graph 7 Perineural invasion (PNI) positivity Kaplan-Meier survival. PNI positive median survival 16 months (IQR = 4-35 months), vs PNI negative median survival 46 months (IQR = 15-75) (p=<0.001).

DISCUSSION Consistent with previous findings, metastatic SCC of

Involved margins were found in 51% of patients (51/101)

the salivary glands occurred predominantly in older

with no differences in survival found compared to close or

Caucasian males within the parotid gland.

clear margins. Positive margin rates of 16-29% have been

7,8,16,19,20

The

majority of patients were treated with surgery involving a neck dissection and adjuvant radiation therapy. Occult nodal disease was identified in 28% of patients, and the majority (53%) of patients had positive cervical nodes,

found in the literature related to the presence of LVI, PNI and proximity to the facial nerve.16,19 However, definitions of margins vary significantly, making comparison difficult.17,23 Principles of oncological resection in parotid tumours are compromised by preservation of the facial nerve.19 Radical

with high rates of ENE (28%). Metastatic SCC of the

parotidectomy with facial nerve transection is indicated in

salivary glands demonstrated high rates of LVI (15%) and

patients with pre-operative facial nerve palsy, but in the

PNI (28%), but low rates (8%) of recurrence compared to

absence of this, guidelines recommend the preservation

other studies.

of the facial nerve, often resulting in close or involved

2,3,6,8,9

Overall, there was a median survival

of 35 months (IQR = 9-68 months) and five-year absolute survival of 53%, consistent with existing literature. 8,20–22 Overall survival was improved in patients without LVI or PNI, with PNI also implicated in recurrence (Graphs 4, 6-7). Patients had improved outcomes if treated with adjuvant

margins.19,24 Our research supports the stance of preserving the facial nerve, as the presence of close or involved margins did not reduce overall survival. We hypothesis marginal status doesn’t influence overall survival due to disease encapsulation within the node (72%) and high rates of adjuvant radiation therapy, which in our study

radiation therapy, but margins, staging and neck dissection

improved overall survival and is supported by the wider

were not found to influence overall survival (Graphs 2-3, 5).

literature.19,20,25

ANZAOMS PREVIEW VERSION |

Within our study the majority of patients had positive

SCC of the submandibular gland is a rare entity. In our

cervical lymph nodes (53%), with a rate of occult nodal

study patients with SCC of the submandibular gland

disease (28%), which is consistent with previous findings

were younger and more likely to be Māori compared to

(20-30%, and 16-35%, respectively). 8,22,26 Metastasis to the cervical lymph nodes has been associated with PNI and LVI, which were highly prevalent in our patient cohort. Other factors such as differentiation, tumour size (>2 cm) and an immunocompromised host have been shown to increase the risk of nodal metastasis.

20,21

Given the high rates of cervical

patients with parotid SCC. This potentially correlates with oral metastatic SCC to the submandibular gland, but no primary lesions were identified. Case reports of SCC of the submandibular gland have high rates of cervical metastasis and ENE as found in our study.29,30 Five-year disease

metastasis authors and guidelines have recommend

specific survival for submandibular SCC is 25-30%, with

selective neck dissection for all patients with salivary gland

high rates of recurrence of up to 60%, however with in our

SCC.13,22,27 Interestingly neck dissection within our study

study, no cases of recurrence were found.29,30 Given the

was not statistically significant regarding overall survival,

aggressiveness of the tumour, authors have advocated for

however few patients were treated with primary radiation therapy (20%). Patients who underwent primary radiation therapy were often misdiagnosed initially (75%) and cN0. Following MDM, primary RT was undertaken based on

surgery, adjuvant radiation and chemotherapy.13,27,29,30 Summarising, these findings, this research demonstrates

patient preference. Given the absence of recurrence, and

the demographics of metastatic salivary gland SCC in a

improved overall in patients treated with neck dissection,

New Zealand hospital. Marginal status should preserve the

primary RT may provide appropriate management of the

facial nerve if clinically able, and adjuvant radiation therapy

cN0 neck, however further research is required.

is indicated, especially for patients with PNI and LVI. Neck dissection was not found to improve survival within our

Within our study 8% of patients had disease recurrence,

study cohort, but has categorically been shown to improve

lower than what has been reported (20-60%). 8,20,25,28

patient survival previously, and is currently indicated

Recurrences commonly occur within the parotid bed, in skin

by head and neck cancer guidelines.12,13,22,27 However,

pre-auricularly or in the ipsilateral neck. 9,20 Overall survival

interestingly within our study cohort primary radiation

is reduced in patients with recurrence as seen within

therapy of the neck in patients who were cN0 did not

our study, however this was not statistically significant.

have reduced overall survival and no patients developed

Recurrence rates have been associated with PNI as found

recurrence. Therefore, primary radiation therapy to the neck

in our study, along with positive margins, increased nodal

may provide an alternative treatment modality, however

burden, ENE, staging and differentiation.

further research is required.

8,28

Our low rates of

recurrence maybe associated with population factors such as age of diagnosis and death from other factors or sample size.

Conflict of interest statement The author(s) declare that there is no conflict of interest. The author(s) received no financial support for the research, authorship, and/ or publication of this article.

Metastatic squamous cell carcinoma of the salivary gland: treatment and prognostic factors related to overall survival and recurrence Referen ce s 1.

Franzen A, Lieder A, Guenzel T, et al. The heterogenicity of parotid gland squamous cell carcinoma: A study of 49 patients. In Vivo 2019; 33; 2001–2006.

Pfisterer MJ, Vazquez A, Mady LJ, et al. Squamous cell carcinoma of the parotid gland: A population-based analysis of 2545 cases. Am J Otolaryngol 2014; 35; 469–475.

Chen MM, Roman SA, Sosa JA, et al. Prognostic factors for squamous cell cancer of the parotid gland: An analysis of 2104 patients. Head Neck 2015; 37; 1–7.

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Pondicherry A, Martin R, Meredith I, et al. The burden of non-melanoma skin cancers in Auckland, New Zealand. Austral J Dermatol 2018; 59; 210–213.

Hinerman RW, Indelicato DJ, Amdur RJ, et al. Cutaneous squamous cell carcinoma Metastatic to parotid-area lymph nodes. Laryngoscope 2008; 118; 1989–1996.

O’Brien CJ. The parotid gland as a metastatic basin for cutaneous cancer. Arch Otolaryngol Head Neck Surg 2005; 131; 551.

Hong TS, Kriesel KJ, Hartig GK, et al. Parotid area lymph node metastases from cutaneous squamous cell carcinoma: Implications for diagnosis, treatment, and prognosis. Head Neck 2005; 27; 851–856.

Dona E, Veness MJ, Cakir B, et al. Metastatic cutaneous squamous cell carcinoma to the parotid: the role of surgery and adjuvant radiotherapy to achieve best outcome. ANZ J Surg 2003; 73; 692–696.

O’Brien CJ, McNeil EB, McMahon JD, et al. Significance of clinical stage, extent of surgery, and pathologic findings in metastatic cutaneous squamous carcinoma of the parotid gland. Head Neck 2002; 24; 417–422.

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Mckenzie J, Lockyer J, Singh T, et al. Salivary gland tumours: an epidemiological review of non-neoplastic and neoplastic pathology. Br J Oral Maxill Surg 2022; 61; 12–18.

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Tartaglione G, Potenza C, Caggiati A, et al. Lymphatic mapping and sentinel node identification in squamous cell carcinoma and melanoma of the head and neck. Tumori Journal 2002; 88; S39–S41.

12.

Sood S, McGurk M, Vaz F. Management of salivary gland tumours: United Kingdom national multidisciplinary guidelines. J Laryngol Otol 2016; 130; S142–S149.

13.

Pfister DG, Spencer S, Adelstein D, et al. Head and Neck Cancers, Version 2.2020, NCCN Clinical Practice Guidelines in Oncology. Journal of the National Comprehensive Cancer Network 2020; 18; 873–898.

14.

El-Naggar AK, Chan JKC, Rubin Grandis J, et al. WHO classification of head and neck tumours. 347.

15.

Goh RYH, Bova R, Fogarty GB. Cutaneous squamous cell carcinoma metastatic to parotid - analysis of prognostic factors and treatment outcome. World J Surg Oncol 2012; 10; 117.

16.

Makki FM, Mendez AI, Taylor SM, et al. Prognostic factors for metastatic cutaneous squamous cell carcinoma of the parotid. J Otolaryngol Head Neck Surg 2013; 42; 14.

17.

Ord RA, Ghazali N. Margin Analysis. Oral Maxillofac Surg Clin North Am 2017; 29; 315–324.

18.

O’Brien CJ, Ka VBM, Mijailovic M. Evaluation of 242 consecutive parotidectomies performed for benign and malignant disease. Awl NZ J Sutg 1993; 63; 870–877.

19.

Iyer NG, Clark JR, Murali R, et al. Outcomes following parotidectomy for metastatic squamous cell carcinoma with microscopic residual disease: Implications for facial nerve preservation. Head Neck 2009; 31; 21–27.

20.

Veness MJ, Morgan GJ, Palme CE, et al. Surgery and adjuvant radiotherapy in patients with cutaneous head and neck squamous cell carcinoma metastatic to lymph nodes: combined treatment should be considered best practice. Laryngoscope 2005; 115; 870–875.

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Hinerman RW, Indelicato DJ, Amdur RJ, et al. Cutaneous squamous cell carcinoma metastatic to parotid-area lymph nodes. Laryngoscope 2008; 118; 1989–1996.

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O’Brien CJ, McNeil EB, McMahon JD, et al. Incidence of cervical node involvement in metastatic cutaneous malignancy involving the parotid gland. Head Neck 2001; 23; 744–748.

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Hanson M, McGill M, Mimica X, et al. Evaluation of surgical margin status in patients with salivary gland cancer. JAMA Otolaryngo Head Neck Surg 2022; 148; 128.

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Geiger JL, Ismaila N, Beadle B, et al. Management of Salivary Gland Malignancy: ASCO Guideline. J Clin Oncol 2021; 39; 1909–1941.

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Andruchow JL, Veness MJ, Morgan GJ, et al. Implications for clinical staging of metastatic cutaneous squamous carcinoma of the head and neck based on a multicenter study of treatment outcomes. Cancer 2006; 106; 1078–1083.

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Jackson GL, Ballantyne AJ. Role of parotidectomy for skin cancer of the head and neck. Am J Surg 1981; 142; 464–469.

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van Herpen C, Vander Poorten V, Skalova A, et al. Salivary gland cancer: ESMO–European Reference Network on Rare Adult Solid Cancers (EURACAN) Clinical Practice Guideline for diagnosis, treatment and follow-up. ESMO Open 2022; 7; 100602.

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Chua MS-T, Veness MJ, Morgan G, et al. Parotid lymph-node metastases from cutaneous squamous-cell carcinomas: Treatment outcome and prognostic factors following surgery and adjuvant radiotherapy. Australas Radiol 2002; 46; 174–179.

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Tahiri I, El Houari O, Loubna T, et al. Squamous cell carcinoma of the submandibular gland with cutaneous fistula: A case report and literature review. Cureus 2022; 14(8); e27785.

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Agarwal M, Agarwal L, Saxena R. Primary squamous cell carcinoma of submandibular salivary gland: A case report. J Clin Diagn Res 2017; 11: XD01–XD02.

ANZAOMS PREVIEW VERSION |

O N C O L O G Y SYSTEMATIC REVIEW

THE EPIDEMIOLOGY OF TONGUE CANCER: A systematic review and meta-analysis

Thanh LP (PhD)† Lopez DJ (PhD)† Wiesenfeld D(MDSc, FDSRCPS , FRACDS(OMS))‡ § Singh A (BDS, MSc, PhD)† §

† C entre for Epidemiology and Biostatistics, Melbourne School of Population and Global Health, University of Melbourne, Parkville, Victoria, Australia ‡ H ead and Neck Tumour Stream, Royal Melbourne Hospital, Parkville, Victoria, Australia §

elbourne Dental School, University of Melbourne, M Parkville, Victoria, Australia

ABSTRACT Objectives: To summarise the epidemiology (incidence rate, prevalence and survival rates) of oral tongue cancer worldwide. Methods: A comprehensive search of relevant literature was conducted in April 2022. English papers that were non-interventional studies, included participants aged 18 years or above, and that investigated the incidence rate, prevalence and morbidity of oral tongue cancer, were selected. A random-effect model was chosen for the meta-analysis, and a sub-group analysis of overall survival and disease-specific

Corresponding Author:

survival between regions was also performed.

ANKUR SINGH Melbourne School of Population and Global Health, University of Melbourne, Parkville, Victoria, Australia Email: ankur.singh@unimelb.edu.au

Results: Of 6,521 articles retrieved, 21 eligible studies were selected for inclusion. Within the study period, the annual percent change in the incidence rates of oral tongue cancer among men was unchanged, whereas that of women increased. The incidence rate of oral tongue cancer among those over 50 was higher than among those under 50. Sub-group analysis indicated that the survival rate after five years of patients from the West Pacific region was higher than that of the Americas and Europe. Conclusion: There is a need for further research on the histological grade of tongue cancers, smoking and alcohol consumption in sufferers, and studies on different regions worldwide to provide a more comprehensive epidemiologic picture. Specific population sub-groups, including women, showed an increase in the oral tongue cancer incidence rate. Abbreviations: GDP, Gross Domestic product; QOL, Quality of life; PRISMA, Preferred Reporting Items for Systematic Reviews and Meta-Analyses; MESH,

Keywords: oral tongue cancer | epidemiology | systematic review

24 | ANZAOMS PREVIEW VERSION

Medical subject headings; WBIC, World Bank Income Classification; IR, Incidence rate; APC, Annual percentage changes; OS, Overall survival; DSS, Disease-specific survival

INTRODUCTION

Tongue cancer is an uncontrolled development of abnormal cells of the tongue and is the most common among head and neck cancer subsites.1,2 It is the most prevalent type of oral squamous cell carcinoma, the eighth most prevalent of all cancers worldwide. 3 A study showed that patients diagnosed with tongue cancer at advanced stages in Brazil only have 30 to 50% 5-year survival.4 Meanwhile, another study showed an improvement in oral tongue cancer survival in Finland from 47% in their previous published series (1995-1999) to 65% in 2017. 5 Squamous cell carcinoma is the most common of lingual malignancies.6 In Australia, oral cancer, which commonly arose from tongue sites (49%), is a lethal disease with a 4.49-fold increase in the number of cases from 2006 to 2018.7 In addition, there is a steep increase in the incidence of tongue cancer in many regions of the world, mostly from 0.4 to 3.3% per year. 8 North and South America and Europe were reported as regions with high rates of tongue cancer cases, which comprised a significant proportion of oral cancer cases. 9 Asian countries, such as Japan and Thailand, have also witnessed a high incidence of tongue cancer, and Taiwan recorded an increasing trend of tongue cancer both in men and women.10,11 Despite having better overall survival, younger patients with tongue cancer have a greater risk of recurrence.12 In young persons, the tongue is the most frequent subsite for oral cavity squamous cell carcinoma, and epidemiological studies have shown a trend of increasing tongue cancer in people under 21 years old.13 The standard treatment for oral tongue cancer is primary surgery with adjuvant radiation and also chemotherapy with strict clinical and pathological criteria for treatment selection.14 Treatment approaches for oral tongue cancer can vary based on several factors, including geography and the stage of the disease.15 In general, single-modality therapy, such as surgery or radiation, can provide a cure for earlystage tongue cancer (T1 or T2).14 Surgery and adjuvant therapy is the optimal treatment plan for patients with advanced disease (T3 or T4).16 Poor locoregional control and adverse prognosis is more common with presentation at these stages.14 Geographic factors may affect access to the diagnosis and treatment of oral tongue cancer patients, leading to different survival outcomes.17,18 Cutting-edge treatment or tailored therapies might not be widely available and accessible in some regions of the world.19,20 There is a significant difference in the disease prognosis across various racial and ethnic groups.21 A two-year follow-up study in 2022 indicated that the treatment cost for recurrent tongue squamous cell carcinoma was more expensive by 51% than primary tumours.22 The treatment of an oral tongue cancer patient in Australia might have an average two-year total cost of $92,671, roughly twice Australia’s Gross Domestic Product (GDP) per capita in 2020, and 95 days of hospital contact.23,24 This is a high cost compared with another common type of cancer, lung cancer, where observed costs for patients aged 45 to 59 and 60 to 69 were $67,700 and $63,500, respectively.25

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INTRODUCTION

In addition, tongue cancer causes severe disability and

A new systematic review to provide updated epidemiology

negatively impacts quality of life. Tongue cancer patients

of tongue cancer is warranted given that current public

are prone to psychological problems due to the limitations

health strategies for preventing oral tongue cancer may not

in their social interaction and emotional expression. As a

address current challenges due to epidemiological changes

result, they usually encounter difficulties eating, swallowing

over the last two decades. 30 Furthermore, the oral tongue,

or speaking.26 A study in Taiwan in 2021 also mentioned the

which is the anterior two-thirds of the tongue, is the most

correlation between depression and difficulty swallowing

common subsite of tongue cancer, has the worst prognosis,

among these patients.

and is less affected by the Human Papillomavirus (HPV)

than the posterior one-third of the tongue. 8,31,32 For that Despite the adverse effects of tongue cancer on the

reason, we aimed to investigate the updated data and

health, economy and quality of life, the last systematic

information regarding the epidemiology of oral tongue

review on the epidemiology of tongue cancer was the

cancer.

study by Moore et al. (2000), which did not include any meta-analysis, and only reviewed the incidence of tongue cancer.6 Meta-analysis provides a quantitative summary of epidemiological measures and effect estimates by pooling data from multiple studies.28 Reviews should ideally be updated every two years or when new evidence exists.29

METHODS Data sources and search progress

Eligibility

A research protocol describing the study objectives, search strategy and analysis plan, was developed for this

A. Inclusion criteria

systematic review and meta-analysis. This systematic search was undertaken in accordance with the Preferred

•

18 years or above.

Reporting Items for Systematic Reviews and MetaAnalyses (PRISMA) guidelines. 33 A comprehensive search of relevant literature was conducted in April 2022 using

Population: those with participants aged

•

Studies investigating the prevalence or incidence rate or mortality rate or survival

online databases of Ovid Medline and Ovid Embase. The

rate of oral tongue cancer/oral tongue

databases were systematically searched using Medical

squamous cell carcinoma.

Subject Headings (MESH) terms and keywords related to oral tongue cancer, epidemiology, and adults. As only

•

cross-sectional study, case-control study

a few cases were reported in the literature, oral tongue

and cohort study.

cancer is a sporadic disease in children.26 Hence, this study focused on people over 18 years old. The search terms used were as follows: (“Oral tongue cancer*” OR “Oral squamous cell carcinoma” OR “Oral tongue tumour*” OR “Oral tongue neoplasm*”) AND (Epidemiology OR Incidence OR Prevalence OR Morbidity OR Mortality) AND (Adult* OR aged OR “over 18 years”).

26 | ANZAOMS PREVIEW VERSION

Studies with epidemiological design:

•

Only English language papers were included.

Risk of bias assessment B. Exclusion criteria •

Population: those with participants aged under 18 years.

•

Case reports, studies and articles that did not investigate the prevalence, incidence rate, mortality rate, or survival rate of oral tongue cancer/oral tongue squamous cell

•

JBI Critical Appraisal Tool for reviews of prevalence/ incidence was used to assess the methodological quality and determine the possibility of bias in its design, conduct and analysis. 35-37 This appraisal tool included nine questions evaluating the (1) sample frame (2) sample selection

carcinoma.

(3) sample size

Study design: interventional studies.

(4) study subjects (5) coverage bias (6) classification bias (7) condition measurement (8) statistical analysis and (9) response rate

Study selection and data extraction After searching online databases, research papers were imported to Covidence for abstract and full-text screening. 34 Firstly, the abstract and title were reviewed, and then, the full-text versions of articles selected from the round of abstract screening were reviewed for the final selection. Studies that met the requirements and eligibility for the synthesis were selected. The following information was extracted from each eligible article to an Excel file for further data analysis: first author’s name, year of publication, country, region, World Bank Income Classification (WBIC), study period, population group, sample size, number of oral tongue cancer patients, study design, cancer classification system, mean age of study population and oral tongue cancer patients, proportion of female participants, incidence rate (IR), annual percentage changes (APC) in the incidence rate, prevalence of oral tongue cancer at stage III and IV, overall survival (OS), disease-specific survival (DSS), mortality rate and risk factors.

Data synthesis and analysis The full text of the selected studies was examined, and information on these was extracted into tables as mentioned above. In addition, the studies were divided by study design groups, and study characteristics within each group were reported to articulate broader similarities and differences among and between the groups. A random-effect meta-analysis was performed as there was an assumption that the effect estimates differed among studies (heterogeneity) due to differences in countries and how the outcome was defined. Each study has its distribution of study effects. Then, the KnappHartung method was used to calculate the confidence interval. In addition to quantitatively assessing the heterogeneity of the pooled estimates, I2 (25, 50, and 75% were considered low, moderate and high heterogeneity, respectively) was applied. 38 The subgroup analysis was performed based on different regions where studies were located as it was expected to be a source of heterogeneity. As there were papers that did not provide the value of 95% confidence intervals (95% CI), a formula for imputing missing standard deviations in meta-analyses from a study in 2006 was used to impute the 95% CI for values of the OS and DSS after five years. 39 Stata version 17.0 software (Stata Corp, College Station, TX) was used for all analyses in this study.

ANZAOMS PREVIEW VERSION |

RESULTS Characteristics of included studies

studies from upper-middle-income countries. In addition, 13 articles were studied within 10 years or more, at which

Figure 1 shows the study selection using a PRISMA flow

the study with the most prolonged period of being followed

chart. Thorough electronic searches identified 6,521

was Ng et al. (2017), with 40 years reported. 8 Samples

studies. After excluding 2,584 duplicates and reviewing

mostly came from oral squamous cell cancer patients and

titles and abstracts, 47 publications were evaluated in full

oral tongue cancer. The retrospective study was the most

text. Of those, 21 studies from 14 countries across five

popular study design among 21 articles in this systematic

regions and 329,177 participants were included in this

review (Table 1).

systematic review. Using the JBI critical assessment tool for prevalence studies, there was one study attaining a full

The proportion of females in articles varied to a great

score of 9, five studies with a score of 8, eight studies with

extent. Whilst Chen et al. (1999) witnessed the lowest

a score of 7, three studies with a score of 6, and four studies with a score of 5.

proportion of females, at 6.25%, 60% of the study sample of Sun et al. (2015) was female, which was the highest figure compared to other articles included in this study. 50 Among 21 studies, Mukdad et al. (2019) had the oldest

Most of the articles included were conducted in the

mean age of participants, 61.6 years old, whereas Farhat

Americas (7/21 studies), Europe (5/21 studies) and Western

et al. (2022) was the youngest, at 37.54 years old. 54,57 There

Pacific (4/21 studies). As per the World Health Organization

were eight articles examining the risk factors of oral tongue

definition, Western Pacific includes South East Asia (Vietnam, Phillipines, Malaysia), Oceania and East Asia (Japan, China, Mongolia). Eleven out of 21 studies were done in countries with high income, and there were five

cancer. In terms of epidemiological outcomes, various measures were used by studies. The geographical sites of these studies and countries were not always reported in the text.

6521 references imported for screening as 6521 studies

2584 duplicates removed

3937 studies screened against title and abstract

3866 studies irrelevant

71 studies assessed for full-text eligibility

50 studies excluded 35 Wrong outcomes 8 Cannot ﬁnd the full-text 6 Wrong exposure 1 Non-English paper

Figure 1

21 studies included

28 | ANZAOMS PREVIEW VERSION

Study selection PRISMA flow chart

ANZAOMS PREVIEW VERSION |

Australia

USA

Daniell 2021⁵⁵

Farhat 2022⁵⁶

France

Brazil

Souto 2021⁴

Deneuve 2022⁵⁷

India

Subramaniam 2020⁵⁴

Serbia

Lazic 2018⁵¹

Global

USA

Tota 2017⁵⁰

Mukdad 2019⁵³

Global

Ng 2017⁸

Brazil

China

Sun 2015⁴⁹

Goldemberg 2018⁵²

China

Fan 2014⁴⁸

Germany

Sri Lanka

Siriwardena 2006⁴⁴

Mucke 2014⁴⁷

Israel

Popovtzer 2004⁴³

USA

Japan

Tateda 2000⁴²

Saba 2011⁴⁶

Taiwan

Chen 1999⁴¹

Italy

USA

SiegelmannDanieli 1998⁴⁰

Garavello 2007⁴⁵

USA

Country

Oliver 1996³⁹

Article

199 0-201 8

199 2-201 7

2007-2016

2007-2009

2004- 2015

1973-2012

2000-2014

1999- 2010

1973-2012

1970-2010

2005-2012

2001-2010

1992-2008

1973-1975

1981-1998

1996-2001

1983-2001

1993- 1998

1985- 1996

1985-1996

1985-1992

Time Period

Table 1. Descriptive characteristics of the investigated articles in the study

Upper middle income

Western Pacific

Europe

High income

N/A

346

Retrospective cohort study

Western Pacific

Americas

High income

Americas

425

243

Retrospective cohort study

Upper middle income

16423

28029

638

140613

89212

430

100

327

51092

138

112

703

Sample Size

Retrospective study

Lower middle income

South-East Asia

N/A

Upper middle income Retrospective study

N/A

Upper middle income

N/A

Retrospective study

Prospective study

Clinical study

Case-control study

Comparative study

Retrospective study

N/A

Retrospective study

Study Design

High income

Global

Americas

Europe

Americas

N/A

Upper middle income

Western Pacific

Global

High income

Europe

Americas

High income

Lower middle income

South-East Asia

Europe

High income

Europe

N/A

High income

WBIC

Western Pacific

Americas

WHO Region

*N/A: Not available WBIC: World Bank Income Classification OS: Overall survival

N/A

45.05%

49.00%

22.50%

28.24%

41.54%

20.00%

24%

30.48%

N/A

60.00%

34.00%

32.10%

29.40%

32.61%

20.54%

58.33%

36%

6.25%

36.36%

N/A

Proportion of Female Participants

N/A

37.54

60.7

N/A

61.6

58.7

N/A

58.7

60.6

N/A

Mean Age of the Participants

N/A

243

346

425

16423

6172

376

16206

89212

176

327

11825

138

147

Number of Tongue Cancer Patients

ICD

AJCC

ICD

AJCC

TNM

ICD

N/A

AJCC

TNM

ICD

AJCC

N/A

AJCC

UICC

ICD

AJCC

ICD

System of Classifying Oral Tongue Cancer

DSS: Disease-specific survival APC: Annual percentage change

Incidence rate, APC

Prevalence, OS, DSS

Mortality rate, DSS

OS, DSS

Prevalence, OS, DSS

Prevalence, Incidence rate, OS, Mortality rate, APC

Incidence rate, APC

APC

DSS

OS, DSS

Mortality rate, OS

OS, APC

Prevalence, OS, DSS

Mortality rate

Prevalence, OS, DSS, mortality rate

Mortality rate

Prevalence

Epidemiological Outcomes

Epidemiology of Oral Tongue Cancer Table 2 shows that the mean age of oral tongue

Table 2 Epidemiological outcomes of investigated articles in the

Table outcomes of of Table 2. 2. Epidemiological outcomes study investigated investigatedarticles articlesininthe thestudy study

cancer participants reported in articles varied from 59 to 61.6 years old, with standard deviations from 12.45 to 13.5. In terms of incidence rate, Deneuve et al.

Study period

Mean age of oral tongue cancer patients

Number of oral tongue cancer patients

Oliver 1996³⁹

1985-1992

N/A

SiegelmannDanieli 1998⁴⁰

1985-1996

N/A

Chen 1999⁴¹

1985-1996

N/A

147

Tateda 2000⁴²

1993-1998

N/A

Popovtzer 2004⁴³

1983-2001

N/A

Siriwardena 2006⁴⁴

1996-2001

N/A

Garavello 2007⁴⁵

1981-1998

N/A

138

Saba 2011⁴⁶

1973-1975

N/A

11825

Mucke 2014⁴⁷

1992-2008

60.6

327

Fan 2014⁴⁸

2001-2010

N/A

Sun 2015⁴⁹

2005-2012

N/A

176

Ng 2017⁸

1970-2010

N/A

89212

Tota 2017⁵⁰

1973-2012

60.2

16206

Lazie 2018⁵¹

1992-2010

N/A

376

Article

(2022) indicated that, in France, whilst the incidence rate among men decreased from 3.9 per 100,000 person-years in 1990 to 1.8 per 100,000 personyears in 2018, that of women went up slightly from 0.6 per 100,000 person-years in 1990 to 0.9 per 100,000 person-years in 2018. However, Lazic et al. (2018) 58

showed that the incidence rate in Serbia increased over the study period from 1999 to 2010 for both genders. 52 In addition, according to Tota et al. (2017), incidence rates among 50 years old or over in the USA were higher than those under 50 years old. 51 Whilst Goldemberg et al. (2018) and Deneuve et al. (2022) reported a decrease in the overall annual percentage change (APC) in the incidence rate of oral tongue cancer among males as well as a rise in that of females in Brazil and France, Lazic et al. (2018) and Tota et al. (2017) showed an increasing trend in this figure at both genders in Serbia and the USA. 51-53,58 Regarding the prevalence of oral tongue cancer at stage III or over, there was a significant variation from 26.09% to 71.70%. This was a considerable aspect as the survival proportion of oral tongue cancer patients at stage III, or over was reported as 50% or under. Regarding the OS, except for the result of Mukdad et al. (2019), the proportion of oral tongue cancer survivors after the first two years was over 80.0%. 54 According to Garavello et al. (2007), this proportion went down remarkably after five years in Italy, as Italy’s lowest OS reported at this time was 39.13%.46 The results of other studies mainly ranged from 47.50% to 69.39%. On the other hand, 79.90% was the highest OS after five years, which Fan et al. (2014) reported in China.49 The DSS proportion after five years was slightly higher than the OS proportion after five years in overall. The overall mortality rate after two years due to oral tongue cancer was up to 20%, and this figure after five years was 37.50%, as reported by Popovtzer et al. (2014) in Israel.44

30 | ANZAOMS PREVIEW VERSION

*N/A: Not available

DSS: Disease-specific survival

WBIC: World Bank Income Classification

APC: Annual percentage change

IR: Incidence rate

95% CI: 95% Confidence interval

OS: Overall survival

Incidence rate

APC in the incidence rate

Prevalence of oral tongue cancer at stage III or over

DSS

Mortality rate

N/A

OS after 2 years=80.00%

N/A

40.43%

OS after 5 years=51.00%

N/A

Overall mortality rate=27.20%

DSS after 5 years=64.00%

Overall mortality rate=30.77%

OS after 5 years=60.00% N/A

N/A

35.90%

OS after 5 years at those ≤40=80.30% OS after 5 years at those>40=61.50%

N/A

N/A OS after 3 years among those≤40=78.26% OS after 3 years among those≥50=100.00%

N/A

Overall mortality rate after 2 years=20.00% Overall mortality rate after5 years =37.50%

N/A

OS after 5 years=39.13% N/A

N/A

26.09%

OS after 5 years at those ≤40=50.00% OS after 5 years at those>40=34.00%

N/A

OS after 5 years=53.10%

N/A

OS after 2 years=87.50%

N/A

OS after 5 years=79.90%

DSS after 5 years=66.00%

N/A

DSS after 5 years=63.00%

N/A

Male APC minimum= -3.00%(95%CI:-7.00%; -0.20%) Male APC maximum= 3.70%(95%CI:2.80%;4.60%) Female APC minimum= -0.60%(95%CI:-1.00%;0.1) Female APC Maximum= 9.10%(95%CI:6.10%;12.00%)

N/A

Overall male APC=5.10% Overall female APC=0.60%

N/A

Male≥50 IR=3.4 Male<50 IR=2.3 Female≥50 IR=4.8 Female<50 IR=1.8 Overall male IR=1.97 per 100,000 persons a year Overall female IR=0.54 per 100,000 persons a year Male IR in 1999=1.55 per 100,000 persons a year Male IR in 2010=3.02 per 100,000 persons a year Female IR in 1999= 0.63 per 100,000 persons a year Female IR in 2010=0.76 per 100,000 persons a year

Overall male APC=4.10% (95%CI:-3.10%; 11.80%) Overall female APC=1.70% (95%CI:-11.50%;16.90%)

Overall mortality rate=39.45%

ANZAOMS PREVIEW VERSION |

Table 2. Epidemiological outcomes of investigated articles in the study

Article

Study period

Mean age of oral tongue cancer patients

Number of oral tongue cancer patients

Incidence rate

APC in the incidence rate

Male IR min=1.62

Goldemberg 2018(52)

2000-2014

6172

Male IR max=2.80 Female IR min=1.62

Overall male APC=-5.60% Overall female APC=0.40%

Female IR max=2.81

Mukdad 2019(53)

1973-2012

61.6

16423

N/A

Subramaniam 2020(54)

2004-2015

N/A

425

N/A

Souto 2021(4)

2007-2009

N/A

346

N/A

Daniell 2021(55)

2007-2016

60.7

243

N/A

Farhat 2022(56)

1992-2017

N/A

Male IR in 1990=3.9 per 100,000 person-years

Deneuve 2022(57)

1990-2018

N/A

Male IR in 2018=1.8 per 100,000 person-years

Overall male APC= -2.80% (95%CI:-3.20%; -2.30%)

Female IR in 1990= 0.6 per 100,000 person-years

Overall female APC =1.70%(95%CI:1.00%;2.30%)

Female IR in 2018=0.9 per 100,000 person-years

32 | ANZAOMS PREVIEW VERSION

*N/A: Not available WBIC: World Bank Income Classification IR: Incidence rate

DSS: Disease-specific survival APC: Annual percentage change 95% CI: 95% Confidence interval

OS: Overall survival

Prevalence of oral tongue cancer at stage III or over

65.40%

OS after 2 years=81.60%

OS after 2 years=68.20% OS after 5 years=53.70% OS after 10 years=38.40% Men≤40: OS after 2 years=74.70% OS after 5 years=66.80% OS after 10 years=61.1% Men>40: OS after 2 years=65.40%

34.90%

OS after 5 years=48.90% OS after 10 years=33.20% Women≤40: OS after 2 years=80.20% OS after 5 years=74.70% OS after 10 years=69.40% Women>40: OS after 2 years=68.20% OS after 5 years=53.70% OS after 10 years=38.40%

N/A

71.70%

DSS after 10 years=66.90% Men≤40: DSS after 2 years=78.60% DSS after 5 years=71.90% OS after 10 years=68.1% Men>40: DSS after 2 years=77.90% DSS after 5 years=69.70% DSS after 10 years=63.10% DSS after 2 years=81.80% DSS after 5 years=76.70% DSS after 10 years=73.80% Women>40: DSS after 2 years=79.80% DSS after 5 years=73.30% DSS after 10 years=66.90%

DSS after 5 years at those ≤45=67.00% DSS after 5 years at those>45=74.00%

OS after 5 years at stage 2=67.00%(95%CI:50.00%;80.00%)

OS after 5 years at stage 4=41.00%(95%CI:29.00%;54.00%)

OS after 2 years=80.30% OS after 5 years=47.50%

N/A

Women≤40:

OS after 5 years at those>45=71.00%

OS after 5 years at stage 3=50.00%(95%CI:32.00%;65.00%)

N/A

DSS after 5 years=73.20%

DSS after 5 years=72.12%

OS after 5 years at stage 1=84.00%(95%CI:75.00%;90.00%)

N/A

DSS after 2 years=79.80%

OS after 5 years at those ≤45=65.00%

N/A

Mortality rate

Overall mortality rate after 2 years= 18.40%

N/A

OS after 5 years=69.39%

OS after 5 years=65.00% (95%CI:59.00%;71.00%)

39.00%

DSS

DSS after 2 years=40.60% DSS after 5 years=31.20%

N/A

Overall mortality rate=72.50%

DSS after 5 years=69.00% (95%CI:62.00%;7.00%) DSS after 5 years at stage 1=92.00%(95%CI:84.00%;97.00%) DSS after 5 years at stage 2=71.00%(95%CI:53.00%;83.00%)

N/A

DSS after 5 years at stage 3=51.00%(95%CI:30.00%;68.00%) DSS after 5 years at stage 4=41.00%(95%CI:28.00%;53.00%)

N/A

ANZAOMS PREVIEW VERSION |

Meta-analysis

The pooled APC in the incidence rate of oral tongue cancer among women was 1.00%, with a 95%CI from 0.00 to

The studies by Oliver et al. 1996, Chen et al. 1999,

2.00%, which means that the APC in the incidence rate

Popovtzer et al. 2004, Siriwardena et al. 2006, and Ng et

of oral tongue cancer among women increased by 1.00%

al. 2017 were not included in the meta-analysis as they did

over the study period (Figure 2). The level of heterogeneity

not provide any of the expected epidemiological outcomes

in the forest plot measuring the APC in the incidence rate

for the meta-analysis (i.e. overall survival after five years,

of oral tongue cancer among men was 99.44% (Figure 2),

disease-specific survival after five years, and overall

significantly higher than that of the forest plot measuring

annual percentage change). 8,40,42,44,45 Among the other 16

the APC in the incidence rate of oral tongue cancer among

studies, those provided relevant epidemiological outcomes

women, at 38.25% (Figure 2).

will be conducted meta-analysis according to different epidemiological measures.

The pooled OS after five years was 59.00%, with a 95%CI from 51.00% to 66.00% (Figure 3). The pooled DSS was

The pooled APC in the incidence rate of oral tongue cancer

higher, at 63.00%, with a 95%CI from 51.00% to 74.00%

among men showed no change, with a 95%CI from -5.00

(Figure 3). Forest plots measuring OS and DSS after five

to 5.00% (Figure 2). As this was a very wide CI that crossed

years show models with high heterogeneity.

the 0, and the I2 for the pooled estimated is substantial, it indicated that further information was required or the sample was too small.

Male APC with 95% CI

Study

Weight (%)

Goldemberg, 2018

-0.06 [ -0.07, -0.04] 27.21

Deneuve, 2022

-0.03 [ -0.03, -0.02] 27.51

Lazic, 2018

0.04 [ -0.03, 0.12] 17.79

Tota, 2017

0.05 [ 0.05, 0.06] 27.49

Overall

-0.00 [ -0.05, 0.05]

Heterogeneity: 2 = 0.00, I 2 = 99.44%, H 2 = 178.96 Test of 0i = 0 j : Q(3) = 608.56, p = 0.00 Test of 0 = 0:z = -0.06, p = 0.95

-.05

Random-effects REML model

.05

Female APC with 95% CI

Study

Weight (%)

Goldemberg, 2018

0.00 [ -0.02, 0.03] 12.80

Deneuve, 2022

0.02 [ 0.01, 0.02] 49.58

Lazic, 2018

0.02 [ -0.12, 0.16]

Tota, 2017

0.01 [ 0.00, 0.02] 37.21 0.01 [ -0.00, 0.02]

Overall Heterogeneity:

0.40

= 0.00, I = 38.25%, H = 1.62 2

Test of 0i = 0 j : Q(3) = 3.66, p = 0.30 Test of 0 = 0:z = 2.43, p = 0.02 Random-effects REML model

-.1

Figure 2 Forest plot using the random-effect REML model with the Knapp-Hartung adjustment for the measure of APC in the incidence rate of oral tongue cancer among men and women

34 | ANZAOMS PREVIEW VERSION

Study

OS after 5 years with 95% CI

Weight (%)

Daniel, 2019

0.65 [ 0.59, 0.71] 10.63

Fan, 2014

0.80 [ 0.68, 0.92]

8.80

Farhat, 2022

0.47 [ 0.35, 0.60]

8.74

Mukdad, 2019

0.54 [ 0.53, 0.54] 11.44

Saba, 2011

0.53 [ 0.52, 0.54] 11.43

Siegelmann-Danieli, 1988

0.51 [ 0.41, 0.61]

Subramaniam, 2020

0.69 [ 0.65, 0.74] 10.96

Tateda, 2000

0.60 [ 0.45, 0.75]

Mucke, 2014

0.68 [ 0.63, 0.74] 10.82

Garavello, 2007

0.39 [ 0.31, 0.47] 10.07

Overall Heterogeneity:

9.40 7.71

0.59 [ 0.51, 0.66] 2

= 0.01, I = 98.93%, H = 93.59 2

Test of 0i = 0 j : Q(9) = 124.46, p = 0.00 Test of 0 = 0:z = 15.72, p = 0.00

Random-effects REML model

Study

DSS after 5 years Weight with 95% CI (%)

Daniel, 2019

0.69 [ 0.62, 0.75] 14.69

Souto, 2021

0.31 [ 0.26, 0.36] 14.98

Fan, 2014

0.66 [ 0.54, 0.78] 13.18

Mukdad, 2019

0.73 [ 0.72, 0.74] 15.44

Sabramaniam, 2020

0.72 [ 0.68, 0.77] 15.07

Tateda, 2000

0.64 [ 0.49, 0.79] 12.08

Sun, 2015

0.63 [ 0.56, 0.70] 14.55

Overall

0.63 [ 0.51, 0.74]

Heterogeneity: 2 = 0.02, I 2 = 97.36%, H 2 = 37.84 Test of 0i = 0 j : Q(6) = 269.00, p = 0.00 Test of 0 = 0:z = 10.95, p = 0.00

Random-effects REML model Figure 3 Forest plot using the random-effect REML model with the Knapp-Hartung adjustment for the measure of OS and DSS after five years

The forest plot measuring the OS after five years by regions

The forest plot measuring the DSS after five years between

shows that the pooled OS after five years in Europe and the

regions shows significant differences in the DSS after five

Americas were similar, at 54.00% and 53.00% (Figure 4).

years between different regions worldwide, especially the

Whilst the heterogeneity in Europe was high, at 97.23%,

difference between the Americas and other regions (Figure

that in the Americas was remarkably low, at 0.03% (Figure

5). Whilst the pooled DSS after five years in other regions

4). In Western Pacific, the pooled OS after five years was

varied from 66.00% to 73.00%, that of the Americas was

68.00%, with moderate heterogeneity, at 67.37% (Figure

31.00% (Figure 5).

4).

ANZAOMS PREVIEW VERSION |

Study

OS after 5 years with 95% CI

Weight (%)

0.47 [ 0.35, 0.60]

8.74

Americas Farhat, 2022 Saba, 2011

0.53 [ 0.52, 0.54] 11.43

Siegelmann-Danieli, 1988

0.51 [ 0.41, 0.61]

Heterogeneity:

9.40

0.53 [ 0.52, 0.54]

= 0.00, I = 0.03%, H = 1.00 2

Test of 0i = 0 j : Q(2) = 0.99, p = 0.61 Western Pacific Daniel, 2019

0.65 [ 0.59, 0.71] 10.63

Fan, 2014

0.80 [ 0.68, 0.92]

8.80

Tateda, 2000

0.60 [ 0.45, 0.75]

7.71

Heterogeneity: 2 = 0.01, I 2 = 67.37%, H 2 = 3.07

0.68 [ 0.58, 0.79]

Test of 0i = 0 j : Q(2) = 5.81, p = 0.05 Europe Mucke, 2014

0.68 [ 0.63, 0.74] 10.82

Garavello, 2007 Heterogeneity:

0.39 [ 0.31, 0.47] 10.07 0.54 [ 0.25, 0.83]

= 0.04, I = 97.23%, H = 36.09 2

Test of 0i = 0 j : Q(1) = 36.09, p = 0.00 Global Mukdad, 2019 Heterogeneity:

0.54 [ 0.53, 0.54] 11.44 2

0.54 [ 0.53, 0.54]

= 0.00, I = . %, H = . 2

Test of 0i = 0 j : Q(0) = 0.00, p = . South-East Asia Subramaniam, 2020

0.69 [ 0.65, 0.74] 10.96

Heterogeneity: 2 = 0.00, I 2 = .%, H 2 = .

0.69 [ 0.65, 0.74]

Test of 0i = 0 j : Q(0) = 0.00, p = . Overall Heterogeneity:

0.59 [ 0.51, 0.66] 2

= 0.01, I = 98.93%, H = 93.59 2

Test of 0i = 0 j : Q(9) = 124.46, p = 0.00

Test of group differences: Q(4) = 54.71, p = 0.00 Random-effects REML model

Figure 4 Forest plot using the random-effect REML model with the Knapp-Hartung adjustment for the measure of OS after five years between different regions worldwide

36 | ANZAOMS PREVIEW VERSION

DSS after 5 years Weight with 95% CI (%)

Study Americas Souto, 2021

0.31 [ 0.26, 0.36] 14.98

Heterogeneity: 2 = 0.00, I 2 = .%, H 2 = .

0.31 [ 0.26, 0.36]

Test of 0i = 0 j : Q(0) = 0.00, p = . Western Pacific Daniel, 2019

0.69 [ 0.62, 0.75] 14.69

Fan, 2014

0.66 [ 0.54, 0.78] 13.18

Tateda, 2000

0.64 [ 0.49, 0.79] 12.08

Sun, 2015

0.63 [ 0.56, 0.70] 14.55

Heterogeneity: 2 = 0.00, I 2 = 0.00%, H 2 = 1.00

0.66 [ 0.62, 0.70]

Test of 0i = 0 j : Q(3) = 1.57, p = 0.07 Global Mukdad, 2019 Heterogeneity:

0.73 [ 0.72, 0.74] 15.44 2

0.73 [ 0.72, 0.74]

= 0.00, I = . %, H = . 2

Test of 0i = 0 j : Q(0) = 0.00, p = . South-East Asia Subramaniam, 2020 Heterogeneity:

0.72 [ 0.68, 0.77] 15.07 0.72 [ 0.68, 0.77]

= 0.00, I = . %, H = . 2

Test of 0i = 0 j : Q(0) = 0.00, p = . Overall

0.63 [ 0.51, 0.74]

Heterogeneity: 2 = 0.02, I 2 = 97.36%, H 2 = 37.84 Test of 0i = 0 j : Q(6) = 269.00, p = 0.00

Test of group differences: Q(3) = 267.43, p = 0.00 Random-effects REML model

Figure 5 Forest plot using the random-effect REML model with the Knapp-Hartung adjustment for the measure of DSS after five years between different regions worldwide

DISCUSSION Studies recorded increased oral tongue cancer incidence

The sub-group analysis also demonstrated the differences

rates among women from 1990 to 2018. In addition, many

of OS and DSS after five years between regions in the

patients diagnosed with oral tongue cancer at advanced

world, where these figures of the Western Pacific were

stages reported a 50% or less survival rate. The trend of

higher than in other regions, such as the Americas and

increasing APC in the incidence rate of oral tongue cancer

Europe. However, the number of papers per region involved

was also found among women, whereas that of men was

in the sub-group analysis was relatively small, especially

inconclusive due to different outcomes between articles.

in South-East Asia, Europe and America, making less

The meta-analysis indicated that whilst there was a slight

heterogeneity.

increase of APC in the incidence rate of oral tongue cancer among women, that of men seemed to be unchanged over

This study has the advantage of including a wide range

time.

of epidemiological measures and a significant number of studies over different countries and regions, providing a comprehensive insight into the epidemiology of oral ANZAOMS PREVIEW VERSION |

DISCUSSION

tongue cancer. Another strength is that meta-analysis was

III and IV were 35.90% and 18.95%, respectively.66 Hence,

performed, summarising and quantifying the results from

it is probably a need for a better screening technique for this

different studies. There was a lack of 95% CI or standard

cancer.

deviations for some epidemiological measures within selected studies. To overcome this limitation, we used a

Regarding survival, a study in South Korea in 2017 reported

formula for imputing missing standard deviations in meta-

that the OS after five years among the older and younger

analyses from a study in 2006 that was used to impute the

patients were 70% and 42%, respectively, and the DSS

95% CI for values of the OS and DSS after five years.

after years was 73% for the former and 40% for the latter.67

spite of conducting sub-group analysis of the OS and DSS

In comparison with other studies involved in this systematic

after five years by different regions worldwide, there was a

review, whilst outcomes from Subramaniam et al. 2020

low number of studies involved in each area.

showed a relatively similar trend, results from Mukdak et

al. (2019), Garavello et al. (2007) and Tateda et al. (2000) The lack of cancer registry results is another limitation

were remarkably different, in which the survival rate of

as many global disease databases (e.g. Global Burden of

older patients was lower than the younger ones.43,46,54,55,67

Disease study and International Agency for Research on

These differences might be due to the difference in

Cancer) do not present data by oral sites including oral

socioeconomic and other relevant factors of different

tongue. Therefore, it is not possible to include data within

regions where research was conducted. Previous studies

meta-analysis from cancer registries across the world

indicate that those from the higher social class tend to have

in this review. Also, including only English articles might

better awareness and participate in cancer screening more

exclude other relevant studies written in other languages.

than their counterparts.68,69 Further studies are needed to

Not including grey literature, including government reports

investigate these differences.

and data from cancer registries, is a key limitation of this Findings showed that whilst the APC of the incidence rate

study.

of oral tongue cancer increased among women, that of men Compared with the latest systematic review on the

was constant over time. This might be explained by the

epidemiology of tongue cancer published in 2000,

increased exposure to alcohol and tobacco, which are the

this study considered a wide range of epidemiological

risk factors for oral tongue cancer, among women.70 Women

measurements. In terms of the prevalence trend, whilst

seem to be more susceptible to the effect of smoking

the study conducted by Moore et al. (2000) mentioned

than men.70 For that reason, different policies reducing

the decreasing and increasing trend in different regions,

the impact of oral tongue cancer’s risk factors on women

the study of Kim et al. (2020) and Satgunaseelan et al.

are necessary. Prevention solutions such as screening for

(2020) showed a significant increasing trend from 1999 to

tongue cancer might also be essential as this solution not

2015.6,59,60 Kim et al. (2020) also showed a rising prevalence

only protect people against the risk of severe diseases

of oral tongue cancer by 2.36%, similar to many studies

but also might save a substantial cost of treatment as the

in this systematic review.

treatment expenses for early stages were five times lower

This prevalence trend can also

be found in the study of Patel et al. (2011) and Cassidy et al. (2014).

61,62

Regarding risk factors, in addition to alcohol

than the advanced one.71 Regarding major risk factors, smoking and alcohol consumption were mentioned the most

and smoking, HPV has been identified as the cause of the

by studies included in this systematic review. However,

increasing oropharyngeal cancer (including the tonsil, base

this research did not investigate their effect magnitudes on

of the tongue, and other parts of the pharynx) incidence

oral tongue cancer. Hence, there is a need for research to

in some countries.63 However, a case-control study in the

examine the reasons behind the increasing trend of tongue

US showed that tongue cancer is less frequently linked

cancer among non-smokers and non-drinkers, as results

to HPV-related cancers than oropharyngeal cancer (the

from these studies might affect the policy of preventing

back of the throat).

Another paper in Sweden discovered

risk factors of tongue cancer. As many papers in this study

that just a small percentage of tongue cancer cases were

indicated that many patients were diagnosed at advanced

HPV-positive, indicating that HPV is not a significant

stages, which observed a lower survival rate, better

factor in causing tongue cancer. The meta-analysis of

screening techniques and programs encouraging people to

this systematic review revealed different outcomes, in

do screening are necessary. Stages when the disease was

which the pooled APC in the incidence rate just increased

diagnosed also affected the treatment and prognosis.14

to a small extent in women, and there was no change in this figure among men over time. Many articles included

On the other hand, there is a contradiction of findings

in this systematic review showed a significant proportion

regarding the differences in the survival rate between

of patients diagnosed at advanced stages. This is a point

older and younger groups in the literature. This might be

to pay attention to as another study in Thailand in 2021

due to the histologic grade, which affects the difference in

reported remarkably more detrimental results in which the

survival rate in both groups of patients.67 Furthermore, the

OS after five years of oral tongue cancer patients at stages

extent of invasion is the most critical histological finding in

38 | ANZAOMS PREVIEW VERSION

tongue squamous cell carcinoma for assessment of survival, as it significantly impacts the prognosis and treatment selection.72 Therefore, further studies are recommended

CONCLUSIONS

to examine specific causes of survival in tongue cancer patients. This systematic review and meta-analysis provide a In addition, as for the outcomes from the subgroup analysis

comprehensive picture of the epidemiology of oral tongue

of OS and DSS after five years between different regions

cancer. Whilst the APC in the incidence rate of oral tongue

worldwide, the low heterogeneity of studies in some

cancer among men was found to be unchanged over

regions (e.g. there was only one study from South East

time, that of women had a slight increase. This difference

Asia included in the analysis) might affect the final results.

might come from the increased exposure and the more

The changing epidemiology of tongue cancer, including

susceptibility to risk factors among women. The incidence

incidence, prevalence and survival can directly impact

rate of oral tongue cancer among women increased,

clinicians and health systems in planning resources and

whereas there was a variation in this result among men

health services. Importantly, as tongue cancer management

between different studies. The incidence rate of oral

requires a multidisciplinary team, it is critical to plan future

tongue cancer among those who are 50 years old or over

resources based on the increasing trends of oral tongue

was higher than among those under 50 years old. There

cancer.

was a significantly high number of patients diagnosed at advanced stages of the disease than those diagnosed at

The increase in tongue cancer among women also

earlier stages. The OS and DSS after five years were at

highlights important shifts and a better understanding of

a low level, and those of female was higher than male.

the aetiology of tongue cancer as, comparatively, there is

People living in the Western Pacific seemed to have higher

lesser smoking and alcohol use in women than men across

OS and DSS than those from other regions. However, this

most populations. Finally, this systematic review stresses

is not strong evidence as the number of articles per region

the need to consistently monitor the epidemiological trends

involved in the meta-analysis is relatively small.

of tongue cancer internationally.

Conflict of interest statement The authors report no conflicts of interests or funding related to the subject matter in this article.

Acknowledgements This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

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O N C O L O G Y SCIENTIFIC ARTICLE

ORAL SQUAMOUS CELL CARCINOMA IN NON-SMOKING, NONDRINKING ELDERLY FEMALES A follow-up study McKenzie J (BDS)‡

ABSTRACT

DeAngelis A (BDSc (Hons), PGDipOMS, MBBS, MPhil, FRACDS (OMS)†

Objectives:

Heo J (BDS, MBChB)†

Wiesenfeld D (FDSRCPS (Glasg), FRACDS (OMS), MDSc, FICD, FADI)† † O ral & Maxillofacial Surgery, Royal Melbourne Hospital, Parkville, Victoria, Australia ‡ M BChB Student, Faculty of Medicine, University of Otago, Otago, New Zealand

To extend previous findings of poorer disease specific survival outcomes in non-smoking, non-drinking elderly females with oral squamous cell carcinoma, characterising risk factors in disease-free survival. Methods: A total of 508 patients with oral SCC were included in a retrospective cohort study between January 2007 to December 2016 at the Royal Melbourne Hospital. Analysis of patient demographics, disease characteristics and

Corresponding Author:

treatment outcomes were completed for patients categorised by age

JAEWON HEO

(elderly >70 years), gender, alcohol and cigarette smoking status (smoker

Oral & Maxillofacial Surgery Registrar

and/or drinkers SD or non-smoker, non-drinker NSND).

Royal Melbourne Hospital Parkville, Victoria, Australia

Results:

Email: jaewon727@gmail.com

One hundred and twenty (24%) patients identified as NSND and 9.8% (50) were NSND elderly females. Disease specific survival was significantly worse in NSND elderly females compared to all others (126 months vs 159 months, p=0.004). NSND elderly females showed significantly different tumour subsite distribution compared to others with higher proportion of alveolus tumour and higher proportion of persistent disease post treatment. Conclusion: NSND elderly females have poorer disease specific survival outcome with difference in the disease subsite and more persistent disease post treatment.

INTRODUCTION

Smoking and alcohol consumption are known independent risk factors for the development of oral squamous cell carcinoma (SCC). Together they have a strong synergistic impact on oral SCC carcinogenesis.1 With the overall reduction in smoking and alcohol consumption in recent years, many studies have noted an increasing trend of non-smoking, non-drinking (NSND) patients presenting with oral SCC. Although smokers and drinkers still make up the majority of oral SCC patients, the percentage of NSND patients can be as high as up to 55% in some study populations.2-6 There has been a growing interest in NSND cohort of oral SCC patients as it appears that the prognosis and survival outcome are different to those smoking and/or drinking (SD) populations, likely due to different biology of carcinogenesis.7,8 The NSND population are largely female with a bimodal age distribution (<40 or >70 years of age) and high tendency for oral tongue subsites as compared to the SD population who are largely males aged 50-60 years. 3,4,9-11 Carcinogenesis in the NSND population is poorly understood. Unlike the SD population where there is a clear carcinogen involved with risk reduction upon cessation of exposure, the NSND population does not appear to have an obvious causative agent responsible for their risk profile.12 Various studies have shown that HPV and other viruses are unlikely to be the aetiological agents responsible for carcinogenesis in the NSND population, and to date no other candidate agents have been identified.13,14 Recently there have been various molecular biomarker studies to better understand the pathogenesis of oral cancer in NSND group.7,13-16 This will be a key to understanding the reasons why NSND oral SCC behaves differently, and also to tailor management in this particular subgroup of oral SCC. The aim of this study was to update previous research at our institution identifying poorer outcomes for OSCC in NSND elderly females and to further characterise their risk factors for overall survival. 9,17

44 | ANZAOMS PREVIEW VERSION

METHODS Five hundred and eight patients were included in the

standard drinks/week with no previous history of heavy

study between 1 January 2007 and 31 December 2016

drinking. Elderly was defined as aged over 70 years old at

who were managed within the Multidisciplinary Head and

presentation. The TNM staging from previous years was

Neck Tumour Stream at Royal Melbourne Hospital. Only

converted from the AJCC (American Joint Committee on

patients with oral SCC were included (base of tongue,

Cancer) 7th edition to the AJCC 8 th criteria based on the

oropharyngeal and lip sub-sites were excluded using the

clinical, radiological, and histopathological records. Margins

International Classification of Disease 10th revision, ICD-

were classified as involved (<1mm), close (1-5mm) or clear

10).

(5-10mm and >10mm).

Ethics approval was obtained from the Royal Melbourne

Statistical analysis was completed with SPSS statistical

Hospital Human Research and Ethics Committee

package (Version 26.0, IBM, Somers, NY, USA). Non-

(Reference: QA2021093). Data was obtained via the

parametric data was presented with a median and

BioGRID Australian Comprehensive Cancer Outcomes

interquartile ranges (IQR). Chi-squared (Chi-Sq) tests and

and Research Database (ACCORD) and hospital records.

students’ t-tests were used for disease characteristics and

Survival data was obtained from the Victorian Cancer

demographic data. Kaplan Meier analysis and log-rank

Council Registry and hospital records.

tests were used to assess univariates factors for disease specific survival (DSS). Cox proportional hazards survival

Patients were classified as SD (smokers and/or drinkers)

regression was used for multivariate survival analysis. A

or NSND (Non-Smoker and Non-Drinker). Non-smoking

p-value of <0.05 was taken to be statistically significant.

was defined as <5 cigarettes/week with no previous history of heavy smoking. Non-drinking was defined as <3

RESULTS In total 508 patients were diagnosed with oral SCC

Four hundred and sixty-two (91%) patients underwent

between 2007 and 2016. The median age of patients was

surgical intervention with 314 (62%) achieving clear

66 years (IQR 55 to 75 years). Three hundred and eighty-

margins. Three hundred and twenty-five (70%) underwent

eight patients (76%) identified as SD (smoking and/or

neck dissection, and 252 (55%) had free flap reconstruction.

drinking), with 256/388 (66%) being male. 120 patients

Two hundred and thirteen patients (42%) underwent

(24%) identified as NSND (non-smoking and non-drinking)

adjuvant radiation therapy with 65 (13%) undergoing

with 96/120 (80%) being female. Fifty three percent of

adjuvant chemotherapy. Thirty-seven (7%) patients

NSND patients were elderly (63/120) and 83% of NSND

were managed with palliative intent due to inability to

elderly were females (50/63), while 80/93 (86%) of elderly

tolerate treatment or due to inoperable disease following

SD patients were male (Table 1).

assessment (Table 2).

The most common tumour subsite was the oral tongue

Total follow-up duration varied from 60 months to 187

(218; 43%), followed by the alveolus/gingiva (106, 21%,

months with median of 118 months. At the end of the study

maxilla and mandible combined) and the floor of mouth

duration, 304 (60%) patients were alive, with 138 (27%)

(91; 17%) (Table 1). The majority of patients had early-

having died from their disease. The overall median all-cause

stage oral SCC (T1 159, 31%; T2 119, 23%) without nodal

mortality duration was 82 months (IQR = 75-85 months),

involvement (N0 362; 71%) and only 3 (<1%) patients had

while disease specific mortality was 94 months (IQR = 92-

distant metastatic disease at presentation. Histologically,

96 months) (Table 2).

the majority of tumours were moderately differentiated (221, 44%) with 71 (14%) showing perineural invasion, 36 (7%) lymphovascular invasion and 35 (7%) with extranodal extension (Table 2).

ANZAOMS PREVIEW VERSION |

Subgroup

Total

NSND

NSND F

NSND EF

Demographics Patients

508

388

76.4%

120

23.6%

18.9%

9.8%

Male

280

55.1%

256

50.4%

4.7%

0.0%

Female

228

44.9%

132

26.0%

18.9%

9.8%

<70

310

61.0%

253

49.8%

11.2%

9.1%

0.0%

70+

198

39.0%

135

26.6%

12.4%

9.8%

Tongue

218

42.9%

157

40.5%

50.8%

49.0%

40.0%

Mand Alveolus

14.2%

13.9%

15.0%

16.7%

24.0%

Max Alveolus

6.7%

6.2%

8.3%

9.4%

12.0%

Floor of mouth

17.9%

18.8%

15.0%

12.5%

10.0%

Retromolar

8.3%

10.1%

2.5%

2.1%

4.0%

Cheek

5.5%

6.2%

3.3%

6.3%

4.0%

Hard palate

3.3%

3.6%

2.5%

2.1%

2.0%

Oral vestibule

1.2%

0.8%

2.5%

2.1%

4.0%

159

31.3%

122

31.4%

30.8%

31.3%

30.0%

119

23.4%

20.6%

32.5%

30.2%

26.0%

14.8%

15.5%

12.5%

13.5%

18.0%

17.7%

18.3%

15.8%

17.7%

22.0%

Stage I & II

245

48.2%

177

45.6%

56.7%

56.3%

54.0%

Stage III & IV

203

40.0%

161

41.5%

35.0%

36.5%

42.0%

Tumour subsite

pT (AJCC 8th)

TNM staging (AJCC 8th)

Table 1 Summary of study population Demographics, Tumour subsite, pT (pathological Tumour) staging / TNM staging (AJCC – American Joint Committee on Cancer, 8th edition)

46 | ANZAOMS PREVIEW VERSION

Subgroup

Total

NSND

NSND F

NSND EF

Treatment Surgery

462

90.9%

350

90.2%

112

93.3%

93.8%

96.0%

Adjuvant Rad

213

41.9%

165

42.5%

40.0%

39.6%

36.0%

Adjuvant Chemo

12.8%

12.6%

13.3%

14.6%

10.0%

Palliative

7.3%

7.7%

5.8%

5.2%

6.0%

Clear (>5 mm)

314

61.8%

243

62.6%

59.2%

62.5%

60.0%

Close (1-5 mm)

19.3%

18.8%

20.8%

18.8%

20.0%

Positive (<1 mm)

8.5%

7.5%

11.7%

11.5%

16.0%

Well diff

126

24.8%

24.7%

25.0%

18.0%

Mod diff

221

43.5%

170

43.8%

42.5%

43.8%

50.0%

Poorly Diff

17.7%

17.3%

19.2%

17.7%

18.0%

Perineural Invasion +ve

14.0%

13.9%

14.2%

12.5%

16.0%

Lymphovascular Invasion +ve

7.1%

7.0%

7.5%

8.3%

6.0%

Negative

362

71.3%

268

69.1%

78.3%

79.2%

84.0%

Positive

12.4%

13.7%

8.3%

7.3%

4.0%

Extranodal Extension

6.9%

7.0%

6.7%

7.3%

8.0%

Free

335

65.9%

260

67.0%

62.5%

60.0%

Recurrent

15.6%

16.2%

13.3%

13.5%

10.0%

Persistent

18.5%

16.8%

24.2%

24.0%

30.0%

Alive free of disease

302

59.4%

233

60.1%

57.5%

57.3%

44.0%

Alive with disease

0.4%

0.3%

0.8%

1.0%

0.0%

Deceased due to disease

138

27.2%

106

27.3%

41.7%

56.0%

Deceased free of disease

11.4%

10.8%

13.3%

12.5%

18.0%

Deceased with disease

1.6%

1.5%

1.7%

1.0%

0.0%

Margins

Histology

Nodal status

Post treatment disease status

Survival outcome

Table 2 Summary of study population – treatment received, margins, histology, nodal status, survival outcomes.

ANZAOMS PREVIEW VERSION |

Disease Characteristics Tumour subsite distribution for NSND elderly females was statistically different to all other patient groups (p=<0.001, Chi Sq). Oral tongue remained as the most frequent subsite, however NSND EF had higher proportion of maxilla and mandibular alveolar tumours and lower proportion of floor of mouth and retromolar tumours (Table 1). The staging of the disease, types of treatment received and the degree of differentiation of tumour otherwise showed no significant difference in each group (Tables 1-2). The treatment outcome for NSND EF showed an overall

Figure 1 Survival of Male vs Female (Median survival – 147 vs 171, p=0.414)

worse outcome compared to the other groups with a higher proportion of positive pathological margins, (p=0.035, Chi Sq) persistent disease post treatment (p=0.008, Chi Sq) and disease specific mortality (p=<0.001, Chi Sq) (Table 2).

Disease Specific Survival Outcome Disease specific median survival in males was 147 months (CI 136-158 months) compared to 171 months in females (CI 158-184 months) but this was not statistically significant (p=0.414, Log-Rank) (Figure 1). Smoking status, alcohol consumption and combined smoking and drinking did not alter disease specific survival (Figure 2-4). However, survival was reduced in elderly patients (139 months, CI

Figure 2 Survival of Smoker vs Non-Smoker (Median survival – 158 vs 159, p=0.887)

117-161 months) compared to patients under 70 years of age (171 months, CI 158-184 months) (p=<0.001, LogRank) (Figure 5). Amongst elderly patients, survival was statistically significantly reduced in females (126 months; CI 91-160 months) compared to the males (141 months; CI 106-175 months) (p=<0.001, Log-Rank) (Figure 6). The survival of elderly NSND was also reduced (125 months; CI 72 – 177 months) compared to elderly SD patient (146 months; CI 116-176 months) (p=<0.001, Log-Rank) (Figure 7). Disease specific median survival in NSND elderly females was 126 months (CI 91-160 months) compared to 159 months in all others (CI 151-166 months) and this difference was

Figure 3 Survival of Drinker vs Non-Drinker (Median survival – 152 vs 160, p=0.413)

statistically significant (p=0.004, Log-Rank) (Figure 8). There were no differences in T (p=0.51) or N-staging (p=0.49), extranodal extension (p=0.184), LVI (p=0.39), PNI (p=0.34), disease recurrence (p=0.30) or close/positive margins (p=0.49) compared between NSND elderly female to all other patients (Chi Sq).

Figure 4 Survival of SD (Smoker and Drinker) vs NSND (Non-smoker and Non-Drinker) (Median survival – 158 vs 171, p=0.505)

48 | ANZAOMS PREVIEW VERSION

Statistically significant differences were found in disease specific survival based on histological differentiation (Figure 9), PNI (Figure 10), LVI (Figure 11), nodal status (Figure 12) and margins (Figure 13). In stage I disease NSND had improved survival compared to SD (p<0.001, Log-Rank) (Figure 14). However, for stage II, III and IV disease NSND had worse disease specific survival compared to SD, especially in advanced stages (III and IV) (p=0.02, p=0.08 and p=0.02, respectively) (Figure 15-17). Figure 5 Survival of <70 year old vs > 70 year old (Median survival – 171 vs 139, p=<0.001)

Figure 6 Survival of elderly males vs elderly females

Figure 7 Survival of elderly NSND vs elderly SD (

(Median survival – 141 vs 126, p=<0.001)

Median survival – 125 vs 146, p=<0.001)

Figure 8 Survival of NSND EF (>70 year old) vs All others

Figure 9 Survival of different histological differentiation

(Median survival - 126 vs 159, p=0.004)

(Median survival – Well (161) vs Mod (147) vs Poor (124), p=<0.001)

Figure 10 Survival of PNI status

Figure 11 Survival of LVI status

(Median survival – Yes (147) vs No (162) , p=<0.001)

(Median survival – Yes (100) vs No (161) , p=<0.001) ANZAOMS PREVIEW VERSION |

Figure 12 Survival of nodal status (Median survival – Neg (171) vs

Figure 13 Survival of margin status (Median survival – Clear >10

Pos (124) vs ECS (20) , p=<0.001)

mm (162) vs Clear 5-10 mm (171) vs Close (159) vs Positive (146), p=<0.001)

Figure 14 Survival of Stage I disease NSND vs SD

Figure 15 Survival of Stage II disease NSND vs SD

(Median survival – 180 vs 159, p=<0.001)

(Median survival – 171 vs 179, p=0.02)

Figure 16 Survival of Stage III disease NSND vs SD

Figure 17 Survival of Stage IV disease NSND vs SD

(Median survival – 67 vs 147, p=0.08)

(Median survival – 100 vs 144, p=0.02)

50 | ANZAOMS PREVIEW VERSION

Multivariate Data Analysis

Hazard ratio

95% CI

p-value

Female gender

0.5

0.3 – 0.8

0.002*

NSND Male

2.0

0.6 – 2.1

0.73

NSND Female

1.7

0.7 – 1.7

0.56

NSND Elderly Female

2.9

1.9 – 4.5

<0.001*

Nodal status +ve

1.6

1.1 – 2.4

0.2

Extranodal Extension +ve

1.8

1.1 – 3.0

0.03*

Perineural Invasion +ve

1.1

0.7 – 1.7

0.66

Lymphovascular Invasion +ve

0.7

0.4 – 1.2

0.25

Variable

Female patients were found to have improved survival overall (HR = 0.5, CI = 0.3-0.8, p=0.002) however, NSND elderly females showed statistically significantly worse

Demographics

survival outcomes (HR = 2.9, CI = 1.9-4.5, p=<0.001). The smoking and drinking status did not show any statistically significant differences in the outcome. Histologically, extranodal extension showed statistically significant worse disease specific survival outcomes (HR = 1.8, CI = 1.1-3.0, p=0.03) and other histological markers (positive nodal status, PNI and LVI) were independent predictors of disease

Histology

specific survival (Table 3).

Table 3 Summary of multivariate survival analysis.

DISCUSSION This study is an extension of the previous NSND elderly

In this study, to continue the consistency with the previous

female oral SCC research from our institution. 9 Since the

study, the same criteria were used (<5 cigarettes/week

previous study the total participants have been increased

and <3 standard drinks/week with no previous history of

to 508 (from 287) and the number of NSND elderly female

heavy exposure). This was based the literature supporting

has increased to 50 (from 28) with the minimum follow up

that patients who smoke between 0 and 20 cigarettes per

duration up to 187 months (from 114 months). Despite the

day have a relative risk of 1.30 and patients who consume

additional 221 patients, there is no significant difference

alcohol 15 to 30 grams (1.5-3 standard drinks) per day

in demographics and the characteristics between the

have a relative risk of 1.27.19 Therefore, the likely risk from

additional cohort to the previous cohort. The previous

minimal exposure included in the criteria as NSND is likely

study’s outcome of poorer DSS in NSND EF appears to be

to have a minimal impact on the carcinogenesis.

consistent in this extended study. A multi-institutional study from Oh et al. (2021) also A recent literature review by Adeoye et al. (2021), who

showed that female and elderly patients in particular had

reviewed 26 articles related to NSND oral SCC patients,

worse DSS compared to other groups despite male gender

also showed that NSND oral SCC patients are largely

being a significant factor for worse DSS outcome.20 In this

elderly female patients with early-stage tumours involving

study, there was no significant difference in DSS outcome

tongue and gingivobuccal sub-sites. However, there were

for smoking and drinking status (Figures 2-4). However,

significant variations in the proportion of the NSND patients

age played a role in worse DSS outcome (Figure 5) and in

in each study which were as low as 2.4% in Wiseman et

the elderly group, female gender and NSND status showed

al. (2003) and as high as 55.45% in Bao et al. (2020).2,6

significantly worse DSS outcomes (Figures 6-7).

This large variation could be dependent on the ethnic and cultural background in exposure to smoking and drinking. Some degree of variation could also be dependent on the study’s definition of ‘non-smoker’ and ‘non-drinker’.

Overall, both Kaplan Meier survival analysis and the multivariate survival analysis concluded that NSND EF have poorer survival outcomes (Figure 8, Table 3). It is uncertain why the female gender and NSND status have negative impact in DSS outcome in the elderly group. Reviewing the differences between the NSND elderly group to others, the only significant difference was the tumour subsite, ANZAOMS PREVIEW VERSION |

whilst the staging, treatment received, and the grade of

prognosis in advanced-stage disease. (Figures 16-17).

differentiation were generally the same across patient groups (Tables 1-2).

Overall, there are a number of limitations to the study that can be improved. It is a single-centre, retrospective study

Although the most common tumour subsite for both

with risk of information bias and selection bias. It also does

groups of patients was the oral tongue, it is possible that

not reflect any changes in smoking or drinking status in

this finding could be partially explained by the higher rate

individuals during the follow-up period which may have an

of maxillary alveolar SCC in NSND EF (12% vs 6% in SD

impact on long-term outcome.

and overall population) and a lower rate of floor of mouth and retromolar SCC (FOM 10% to 18%, Retromolar 4% to 8.3%) (Table 1).18 This may explain the higher statistically significant proportion of positive margins (16% vs 8.5%, Table 2) and persistent disease status post treatment (30% vs 18.5%, Table 2) as adequate resection of advanced maxillary tumours is well known to be difficult due to complex anatomy and proximity to vital structures with higher rates of regional failure.21 However, the small sample size of NSND EF with maxillary SCC (6 patients, 12% of NSND EF) makes it difficult to draw any definitive

CONCLUSION

conclusions. In contrast to our findings, other studies have shown no difference in survival outcome for NSND in oral SCC. A recent study from Adeoye et al. (2021) showed that NSND

Our study provides an update on OSCC in the NSND elderly female population, further identifying statistically significant worse disease-free survival. Furthermore, we have identified

(largely females in 8 th decades) showed no difference in

differences in tumour subsites in NSND elderly females that

relapse free survival (RFS) and DSS while a study from

may result in higher rates of positive margins, persistent

Pytynia et al. (2004) which examined 50 never-smokers

disease and poorer survival outcome despite receiving the

with aged matched controls showed that never-smokers

same treatment. Further research is required to understand

had greater OS, DSS and RFS, although this study did not examine drinking status. 5,22

the fundamental differences in biology and prognosis of the NSND elderly female oral SCC population, and further

Another possibility for difference in survival outcome could

molecular-based research is required to understand and be

be due to difference in carcinogenesis for the NSND group

able to target the therapy to this particular group, that appears

compared to the SD group. Whilst the carcinogenesis

to be doing poorly.

associated with smoking and drinking in oral SCC is clear, there is no clear answer for NSND patients and potentially there exists an underlying genetic cause. Also, there is a

Conflict of interest statement

growing interest regarding a role of sex hormones as a

The authors declare that they have no competing and conflict

potential pathway of oral SCC carcinogenesis which may

of interests.

explain the differences in the behaviour in females.23 Perot et al. (2020) investigated for a viral aetiology for NSND oral SCC patients which concluded that known oncogenic infectious agents are absent in this sub-group.13 Koo et al. (2013) looked at the molecular alterations in NSND oral SCC patients which again excluded HPV as an underlying cause of NSND oral carcinogenesis and found a significant molecular difference between NSND and SD group (such as CDKN2A, EGFR and BRCA2 expressions).7 Lastly there are also a number of studies showing the over expression of PD-L1 with increased intensity of tumour infiltration in NSND patients.24-26 Difference in tumour biology could explain the reasons to why early-stage SCC NSND patients have better outcomes compared to SD patients (Figure 14), whilst NSND patients have a worse

52 | ANZAOMS PREVIEW VERSION

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DANIEL SIM DESIGN

O N C O L O G Y SCIENTIFIC ARTICLE

PROGNOSTIC ASSOCIATION OF POST-OPERATIVE RADIOTHERAPY AND DEPTH OF INVASION OF TONGUE CARCINOMAS Gamage SN (MBBS, BDS, BScDent (Hons))†‡ Jensen ED (BDS, BScDent (Hons), DClinDent(Paed))†‡ Sethi S (BDS, MDS (Oral Pathology), PhD)†

ABSTRACT

Cheng A (MBBS, BDS, FRACDS (OMS))†‡

Objectives:

Goss AN (DDSc, FRACDS (OMS))†‡

Depth of invasion and tumour thickness of early tongue squamous

Sambrook P (MBBS, MDS, FRACDS (OMS),

cell carcinoma (SCC) is a predictor of loco-regional disease and an

FIBCSOMS)†‡

influencer of survival. The aim of this research was to characterise the outcomes of individuals who were prescribed adjuvant post-operative

† O ral & Maxillofacial Surgery Unit, The University of Adelaide, South Australia, Australia ‡ O ral and Maxillofacial Surgery Unit, Royal Adelaide Hospital, South Australia, Australia

radiotherapy (PORT) following surgical excision tongue SCC, based on depth of invasion >4mm. Methods: Retrospective audit of individuals identified from a Head and Neck Cancer database at an Australian major hospital who had undergone surgical resection for tongue SCC with neck dissection and had clear/

Corresponding Author: EMILIJA D JENSEN

negative surgical margins. Medical history, surgical details, and investigatory reports were collected, and a long-term survival analysis produced by Kaplan Meier plots.

Oral & Maxillofacial Surgery Unit, The University of Adelaide, South Australia, Australia Email: emilija.jensen@adelaide.edu.au

Results: A total of 123 individuals were identified with tongue SCC and 31 individuals were T1 – T2, N0 and included for review (65.6% received surgery alone and 31.3% received surgery + PORT). The overall survival was 88.8%; surgery alone had an overall survival rate of 70.7%, compared with 90.5% for surgery + PORT, with no significant difference between the groups, x2(1) = 0.080, p=.777 <0.05. Conclusion: The value of adjunctive PORT for individuals with depth of invasion >4mm for tongue SCC (T1 – T2, N0) remains unclear but based on the results of this study, depth of invasion was not a significant prognostic

Keywords:

indicator for locoregional control or overall survival.

carcinoma | squamous cell | tongue neoplasms | radiotherapy ANZAOMS PREVIEW VERSION |

INTRODUCTION

Squamous cell carcinoma (SCC) accounts for approximately 95% of oral cancer, with the tongue being the most common site. Early cervical metastasis occurs with tongue SCC, as there is no fascia to resist invasion along the parallel muscle fibres and because of the rich lymphatic supply, especially in the lateral tongue locus1. Surgical excision of the primary tumour is the mainstay in management of tongue SCC; adjunctive therapy including neck dissection at the time of tumour resection for the management of individuals with early (T1 - T2) tongue SCC in the setting of no neck involvement (N0) leads to improved patient outcomes and survival.2-4 Radiation therapy may also be reserved for adjuvant therapy or as a second line therapy in individuals who may not tolerate surgical management.1-5 In contrast to surgical excision, primary radiotherapy as a primary therapy for tongue SCC is uncommon due to the associated significant morbidity and poor tumour control rates. 5-7 Indications for adjunctive radiation therapy in the management of tongue SCC include close margins of the surgical excision, perineural invasion, extensive bone invasion and neck involvement. The addition of chemotherapy to adjunctive radiation therapy is indicated in the event of positive margins and extra-nodal extension. 8 However, each case requires individual evaluation as there is no universal consensus on these indications. 5,6 It has been suggested that adjuvant radiotherapy be considered in histologic risk assessment in addition to the aforementioned indications. 9 The tumour thickness in tongue SCC is the only histological factor that has consistently predicted loco-regional disease and has the greatest influence on an individual’s survival.1,9-13 The thickness of tumour may be measured from the surface of the tumour to the deepest level of invasion, but this does not account for exophytic tumours which may present as thick but with minimal depth of invasion. Depth of invasion is the distance from the basement membrane/start of connective tissue layer to the deepest level of tumour invasion. Various surgical protocols have been established based on tumour thickness of tongue SCC. Individuals with stage 1 and 2 tongue SCC with tumour thickness >4mm were shown to have an increased risk of subsequent cervical metastasis.11 Thus, elective supraomohyoid neck dissection is recommended. Individuals with tumour thickness less than 3mm had a 5-year survival prediction of 85.7%, significantly greater than if tumour thickness were 4-7mm (five-year survival prediction of 58.3%)12. Tongue SCC tumours that were 2.1-4mm thick had a nodal recurrence rate of 11.2%, increasing to 38.5% when tumour thickness was 4.16mm.13 Although surgical excision with elective supraomohyoid neck dissection is widely accepted as the standard treatment of early tongue SCC with tumour thickness >4mm, there remains limited discussion on the role of post-operative radiotherapy (PORT) based on depth of invasion. Therefore, the aim of this research was to characterise the outcomes of individuals who were prescribed adjuvant radiotherapy following surgical excision for tongue SCC (T1 – T2, N0) when the depth of invasion was greater than 4mm. We hypothesised that depth of invasion alone as an indication to provide postoperative adjuvant radiotherapy will not significantly alter loco-regional disease control or survival in individuals that have already undergone surgical excision and subsequent neck dissection of their tongue SCC (T1 – T2, N0).

56 | ANZAOMS PREVIEW VERSION

METHODS Data source

Predictor variables

This study was a retrospective audit of individuals identified

The main predictor variable in this study was treatment

from the Oral and Maxillofacial Surgery Department, Head

provided for tongue SCC (T1 – T2, N0), which included

and Neck Cancer database, who had been discussed at the

either surgery alone or surgery with PORT. Additionally,

Head and Neck Multidisciplinary Team Meeting at the Royal

we looked at thickness of tumour and whether individuals

Adelaide Hospital (Adelaide, South Australia) from January

with a depth of invasion greater than 4mm benefited from

2011 to December 2017.

adjuvant radiotherapy.

Study population and outcome variables

Demographic variables

Ethics approval was granted from the Royal Adelaide Hospital Human Research Ethics Committee (HREC/17/ RAH/490). Individuals were eligible for inclusion if they had

The following demographic variables were included in this study: age at time of diagnosis, sex.

undergone surgical resection for oral tongue SCC (T1 – T2, N0; anterior 2/3 of tongue) with neck dissection, had clear/ negative surgical margins (>5mm). Those with positive or close surgical margins (≤5mm) were excluded from this study. Review of individuals’ details occurred in April 2022

Tumour-related variables The tumour-related variables included in this study were

and were obtained from medical records, radiological and

primary site, depth of invasion, margins, invasion front, and

histopathological reports related to the tongue SCC. This

type of treatment received.

included age, sex, married status, insurance, smoking habits, alcohol intake, site of tumour, surgical excision

Statistical analyses were performed using STATA version

details (including date of surgery and concomitant neck

15.0 (StataCorp) and SPSS version 27 (IBM SPSS Inc.,

dissection), histopathological features (including “TNM

Chicago, IL, USA). Descriptive statistics were produced by

staging”, depth of invasion (measured from the basement

two-sample t-tests for continuous variables, and Fisher’s

membrane at the start of the connective tissue layer to the deepest level of tumour invasion to the nearest 1mm using an ocular micrometre), surgical margins, presence of bone, perineural or lymphovascular invasion and presence of nodal disease).14,15 To ensure that depth of invasion rather than tumour thickness was measured, confirmation with the single reporting pathologist occurred. Subsequent

Exact test for categorical variables, to analyse variables across the two treatment groups with p-values less than 0.05 indicating a significant association. A Kaplan-Meier analysis log-rank test was used to compare the relationship between surgery-only and surgery + PORT groups for overall survival.

operations, use of adjuvant treatment including PORT and the outcome measures including evidence of loco-regional recurrence and overall survival (months) was also collected. The primary outcome measure in this study was overall survival based on vital status (alive/dead) and number of months of survival or follow-up.

ANZAOMS PREVIEW VERSION |

RESULTS Identification

Identification of individuals from Head and Neck Multidisciplinary Team Meetings

A total of 123 individuals were identified to have SCC of the tongue across the seven-year audit from the Head and Neck Multidisciplinary Team Meeting database. Of these,

Individuals identified from: MDT meeting database (n = 123)

99 individuals received surgery and 24 received radiation therapy as an adjunct to surgery. Exclusion of individuals Screening

with TNM staging greater than T2 or N0 provided a total of 31 individuals (Figure 1). A total of 31 individuals were included in the study (male,

Individuals sought for case note review Surgery alone (n = 99) Surgery + PORT (n = 24)

Individuals excluded TNM stage > T2 or N0 (n = 92)

54.8%, n=17). A total of 65.6% (n=21) received surgery alone for management of tongue SCC, whilst 31.3% (n=10) Included

received surgery + PORT. No significant difference in the age was observed with individuals who received PORT (mean ± SD: 64.2 ± 12.8, vs 64.0 ± 14.5 years; p=.551);

Individuals included in study

there was no significant difference between the treatment

(n = 31) Surgery alone (n = 21) Surgery + PORT (n = 10)

groups based on sex (p=.362; p=.280, Table 1). However, there was a significant difference in the depth of invasion

MDT, multidisciplinary team; PORT, post-operative radiotherapy

when individuals who received surgery alone were

Figure 1

compared with those who received surgery + PORT (67.8,

Flow diagram for individuals identified over seven years from a head

p=.008).

and neck cancer multidisciplinary team meeting database

Characteristics

Total individuals (n=31)

Surgery (n=21)

Surgery and PORT (n=10)

p-value

Age (years)

64.1 ± 13.8

64 ± 14.6

64.2 ± 12.9

.551

Male gender (n, %)

17 (54.8)

10 (47.6)

7 (70)

.280

Married status (n, %)

11 (35.5)

9 (42.9)

2 (20)

.335

Insured (n, %)

7 (22.6)

4 (19)

3 (30)

.518

Smoking status - current (n, %)

14 (45.2)

8 (38.1)

6 (60)

.415

Alcohol status – current (n, %)

15 (48.4)

9 (42.9)

6 (60)

.576

Site of tumour – lateral border (n, %)

26 (83.9)

18 (85.7)

8 (80)

.583

Depth of invasion >4mm (n, %)

21 (67.7)

10 (100)

11 (67.8)

.008*

Tumor margins, (negative; <5mm)

15 (48.4)

9 (42.9)

6 (60)

.306

Perineural invasion (negative)

10 (32.3)

5 (76.2)

5 (50)

.148

Lymphovascular invasion (negative)

29 (93.5)

20 (95.2)

9 (90)

.548

Invasive front, (non-cohesive)

16 (51.6)

12 (57.1)

4 (40)

.306

Survival status (deceased)

10 (32.3)

14 (66.7)

7 (70)

.595

Recurrence (positive)

5 (16.1)

3 (14.3)

2 (20)

.528

Death related to: - Cancer - Unrelated causes

2 (6.5) 7 (22.6)

1 (4.8) 5 (23.8)

1 (10) 2 (20)

.548 .599

Note: Mean ± SD unless specified. PORT, post-operative radiotherapy. *p-values less than 0.05 were considered statistically significant associations.

Table 1 Univariate analysis of demographic factors and tumour features among individuals identified as having surgical resection of tongue squamous cell carcinoma (T1 – T2, N0), with and without post-operative radiotherapy

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Type_of_treatment Surgery + PORT Surgery

Locoregional recurrence occurred for five (16.1%) individuals at an average of 54.4 months post-surgery (SD

100

40.6). Two of the individuals who had locoregional recurrence also had death individuals were deceased during the audit period; causes of death were bladder cancer (n=1), pneumonia (n=3), acute coronary syndrome (n=3) and death due to the SCC (n=2). The rates of locoregional recurrence and nature of death are described.

Cumulative Survival (%)

due to the SCC. A total of nine (29.0%)

The mean follow-up time of individuals in the study was 52.1 ± 35.7 months,

with an overall survival of 88.8%.

.00

Individuals who received surgery alone

20.00

40.00

60.00

80.00

100.00

Follow up (months)

had an overall survival rate of 70.7%,

Figure 2

as compared with 90.5% for individuals

Survival function patterns for individuals

who received surgery + PORT, with

treated with surgery alone compared to

no significant difference between the

surgery and post-operative radiotherapy

groups, 2(1) = 0.080, p=.777 <0.05

(PORT)

(Figure 2). Therefore, we fail to reject the null hypothesis and conclude that there is no difference in the overall survival distributions between the two groups. In multivariate analysis (Table 2), after adjusting for age and gender as

Variable

Unadjusted HR (95% CI)

P-value

Adjusted HR (95% CI)

P-value

Gender

0.48 (0.13 – 1.75)

.268

0.82 (0.06 – 10.65)

.883

Type of treatment

0.81 (0.19 – 3.42)

.778

1.01 (0.004 – 228.66)

.997

T-stage

0.55 (0.115 – 1.93)

.351

0.10 (0.006-1.86)

.124

Tumor margins

0.45 (0.12 – 1.63)

.229

1.31 (0.13 – 13.18)

.818

Perineural invasion

1.26 (0.31 – 5.05)

.745

0.32 (0.03 – 3.51)

.354

Lymphovascular invasion

0.45 (0.05 – 3.67)

.461

0.51 (0.002 – 169.66)

.824

Invasive front

1.15 (0.32 – 4.10)

.821

0.084 (0.003 – 2.18)

.136

covariates, there was no significant difference in overall survival between the individuals who received surgery alone and those who received surgery + PORT (hazard ratio [HR], 1.54; 95% CI, 0.196-12.02; p=.684). Positive and close tumour margins (≤5mm) did not demonstrate a significant difference in overall survival, after adjusting for multiple covariates (HR, 1.82; 95% CI, 0.43- 7.68; p=.412). For individuals with perineural invasion, there was also no significant difference in overall survival based on treatment (HR, 3.71; 95% CI,

Table 2

0.46-29,8; p=.217). The variable that

Predictors of overall survival among individuals

was a significant predictor of survival

with tongue squamous cell carcinoma

in the multivariate model included age

(Stage 2)

(HR, 1.16; 95% CI, 1.04-1.29; p=.007).

ANZAOMS PREVIEW VERSION |

in this cohort may not provide meaningful results as most

DISCUSSION

individuals who did not survive did not die from the tongue SCC. Similarly, the relationship to age and overall survival is expected as the older an individual is, the more likely they

Early tongue SCC with a tumour thickness >4mm or depth of invasion >2mm has consistently predicted loco-regional recurrence and individuals’ overall survival, and hence justified elective neck dissection. However, this study 16

found no significant difference for locoregional disease control nor overall survival using PORT following surgical management of tongue SCC (T1 – T2, N0). There was a significant difference in the depth of invasion for individuals who received surgery alone compared with those who received surgery and PORT, but this would relate to the current treatment protocols from within the surgical unit. This study also found that age was a significant predictor for overall survival of tongue SCC, in keeping with previous

are to die from something other than tongue SCC. The use of disease-free survival or disease-specific survival may improve our understanding but unfortunately this cohort remains too small to provide statistically significant results. The use of PORT following surgery for T1 – T2, N0 tongue SCC may not occur routinely in other centres based on depth of invasion >4mm, in the absence of other adverse histologic features. The decision to proceed with PORT in this study was not by any individual team or consultant but occurred at the multidisciplinary team discussion. Each patient diagnosed with tongue SCC is discussed in an open forum with other surgical, medical oncology and radiation

studies.17,18

oncology teams. Typically, tumours with depth of invasion

Surgery alone, with primary tumour resection and neck

to present the options of PORT to the patient who will

greater than 4mm trigger radiation oncology doctors

dissection, for tongue SCC (T1 – T2, N0) has been found to be highly effective in achieving locoregional disease control and overall survival.19,20 Depth of invasion is known as an important factor in predicting loco-regional recurrence and is a vital prognosticator of survival, but the evidence is limited regarding the use of PORT based on depth of invasion.

11-13

Radiotherapy provided increased overall

survival and improved quality of life for some head and neck cancers.20 However, morbidity including severe xerostomia, oral mucositis, loss of taste, dental caries, trismus and osteoradionecrosis may be experienced by individuals undergoing treatment. The risks involved in PORT should 21

therefore be carefully evaluated against the potential improvement in locoregional disease control. The results of this study do not support the use of PORT based on depth of invasion >4mm for T1 – T2 N0 tongue SCC due to a lack of statistical significance, adding to the growing evidence that additional treatment in those with clear surgical margins may not provide benefit. Larger widespread 22

ultimately make the informed decision whether to proceed. With this in mind, this study provides great strength to the literature as PORT may not be provided as an option for T1 – T2, N0 tongue SCC in other centres, making this one of the largest cohorts available to review the outcome measures following PORT. The authors acknowledge the limitations of this study. The Head and Neck Cancer multidisciplinary team includes other specialties including otolaryngology and plastic surgery. Identification of individuals who were treated by other teams within the same institution may have been overlooked by using data only from the Oral and Maxillofacial Surgery team, contributing to a potentially lower sample size and potential selection bias due to similar philosophies in management. The authors recognise the limitations of having a smaller sample size, but due to critical significance of the variables studied and the results reported; we firmly believe that these findings will be useful

longitudinal studies should be conducted to this effect.

either way. The local recurrence rate of 16.1% reported

Positive and close tumour margins (≤5mm) and individuals

institutions.7,26 However, there were inconsistent follow-up

with perineural invasion did not have a significant difference in overall survival, after adjusting for multiple covariates for those who underwent surgery compared with surgery and PORT. Locoregional control and overall survival of those with positive tumour margins is an independent adverse prognosticator, but the present study did not demonstrate a significant difference in overall survival for individuals with positive tumour margins.23 There are multiple pathological risk factors that may indicate PORT to improve overall survival and locoregional recurrence. Poor 24

histological grade has also been recognised as a predictor of locoregional control and overall survival of those with tongue SCC.

25,26

Unfortunately, this information was not

available for the cohort included in the present study and is a limitation of the dataset. The use of overall survival rate

60 | ANZAOMS PREVIEW VERSION

in this study is consistent with similar findings from other time periods for included individuals which may reduce the robustness of the local and regional recurrence estimates. Similarly, inclusion of histopathological findings like grade of tumour, angiogenesis and vascular invasion would also help to strengthen the findings of this study as a predictor of behaviour of oral SCC.27 Although tumour thickness >4mm is predictable prognosticator for locoregional control and overall survival for tongue SCC (T1 – T2, N0), the value of adjunctive PORT for individuals with depth of invasion >4mm is questionable and requires consideration of morbidities associated with PORT. An informed decision-making process with the affected individuals and multidisciplinary teams are required, rather than in histology alone.

Acknowledgements

Conflict of interest statement

The authors would like to acknowledge the work of the

This research did not receive any specific grant from funding

multidisciplinary team of oral and maxillofacial surgeons,

agencies in the public, commercial, or not-for-profit sectors.

ear nose and throat surgeons, radiation oncologists, medical

No conflict of interest is declared.

oncologists, registrars and allied health of the Head and Neck Cancer service at the Royal Adelaide Hospital who were involved in the management of the individuals included in this study.

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Sessions DG, Spector GJ, Lenox J, et al. Analysis of treatment results for oral tongue cancer. Laryngoscope 2002;112:616-625.

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Gonzalez-Moles MA, Esteban F, Rodriguez-Archilla A, et al. Importance of tumour thickness measurement in prognosis of tongue cancer. Oral Oncol 2002;38:394-397.

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Balasubramanian D, Ebrahimi A, Gupta R, et al. Tumour thickness as a predictor of nodal metastases in oral cancer: comparison between tongue and floor of mouth subsites. Oral Oncol 2014;50:1165-1168.

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Amin MB, Greene FL, Edge SB, et al. The eighth edition AJCC cancer staging manual: continuing to build a bridge from a population‐ based to a more “personalized” approach to cancer staging. Cancer J Clin 2017;67:93-99.

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O’Brien CJ, Lauer CS, Fredricks S, et al. Tumor thickness influences prognosis of T1 and T2 oral cavity cancer—but what thickness? Head Neck 2003;25:937-945.

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Brockhoff HC, Kim RY, Braun TM, et al. Correlating the depth of invasion at specific anatomic locations with the risk for regional metastatic disease to lymph nodes in the neck for oral squamous cell carcinoma. Head Neck 2017;39:974-979.

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Chang T-S, Chang C-M, Ho H-C, et al. Impact of young age on the prognosis for oral cancer: a population-based study in Taiwan. PLoS One 2013;8(9):e75855.

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Ansarin M, De Berardinis R, Corso F, et al. Survival outcomes in oral tongue cancer: A mono-institutional experience focusing on age. Front Oncol 2021;11:616653.

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Ch’Ng S, Corbett‐Burns S, Stanton N, et al. Close margin alone does not warrant postoperative adjuvant radiotherapy in oral squamous cell carcinoma. Cancer 2013;119:2427-2437.

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Abed H, Reilly D, Burke M, Daly B. Patients with head and neck cancers’ oral health knowledge, oral health‐related quality of life, oral health status, and adherence to advice on discharge to primary dental care: A prospective observational study. Spec 2019;39(6):593-602.

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Nabil S, Samman N. Incidence and prevention of osteoradionecrosis after dental extraction in irradiated patients: a systematic review. Int J Oral Maxillofac 2011;40:229-43.

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Fridman E, Na’ara S, Agarwal J, et al. The role of adjuvant treatment in early‐stage oral cavity squamous cell carcinoma: An international collaborative study. Cancer 2018;124:2948-2955.

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ANZAOMS PREVIEW VERSION |

O N C O L O G Y CASE REPORT & REVIEW

RECURRENT DESMOPLASTIC FIBROMA OF THE MAXILLA Case report and review of the literature

Tocaciu S (MBBS, BDS, MPhil, FRACDS (OMS))†

ABSTRACT

Barrowman R (BDS, MBBS, GradDip (OMS), FRACDS (OMS))†

Desmoplastic fibroma (DF) of the maxilla is a rare, intraosseous,

Lim B (BDSc, MBBS, PGDip, FRACDS (OMS))†

benign tumour which has high rates of recurrence. Signs,

Rutherford N (BDSc, MBBS, FRACDS (OMS))†

symptoms and imaging studies are generally non-specific. While

Daniel F (BBiomed, MD)†

histopathological diagnosis remains the gold standard, the lack of † D epartment of Oral & Maxillofacial Surgery, Austin Health, Heidelberg, Victoria, Australia

reliable immunohistochemical markers and overlap with malignant processes may lead to misdiagnosis. Management remains mainly surgical, with wide local resection with clear margins to reduce recurrence rates. The use of chemotherapy and radiotherapy in

Corresponding Author: FADY DANIEL Department of Oral & Maxillofacial Surgery

DF is not well established and data from their use in desmoid type fibromatosis may not be appropriate with emerging data separating these as pathologically distinct entities.

Austin Health Heidelberg, Victoria, Australia Email: f.daniel@live.com.au

INTRODUCTION Desmoplastic fibroma (DF) is a rare, benign, and locally aggressive intraosseous lesion. Long bone involvement was first described by Jaffe in 1958 and the first report of jaw DF was in 1965 by Griffith and Irby.1,2 There have been several reports of involvement of the jaws with a predilection for the mandible over the maxilla. 3 Owing to its rarity, issues with diagnosis and management at all sites are reported, as well as high rates of recurrence particularly in the mandible3. We present a case of recurrent desmoplastic fibroma of the maxilla and review of the literature of desmoplastic fibroma at this site, which aims to highlight the difficulty in surgically treating a benign but locally aggressive tumour and investigate the current diagnostic and medical and surgical treatment algorithms.

CASE REPORT A 61-year-old Caucasian male was referred to the Oral & Maxillofacial Department following an incidental finding of a right maxillary lesion on cone beam tomography (CT) during investigation for seizures. He had a past medical history for mild dilated cardiopathy with previous myocardial infarction, progressive spinocerebellar ataxia, hypertension and alcohol abuse. On presentation, he denied any related symptoms but did complain of longstanding nasal obstruction. Examination revealed a palpable swelling with associated bone loss over the right anterior maxilla. There was no infra-orbital nerve paraesthesia. The initial CT scan performed for investigation for seizures showed a soft tissue lesion in the anterior right maxilla continuous with the hard palate causing bone erosion and protruding into the anterior nasal cavity. Following this, a targeted maxillary cone beam CT showed a 2.6 x 1.5 x 1.1cm radiolucent lesion with irregular margins in the midline maxilla (Figure 1). There was destruction of the anterior and central superior cortex, with a small breach in the mucosa of the right nasal floor, but without evidence of soft tissue mass beyond the margins of the maxilla. Given concern of primary malignancy, he underwent initial incisional biopsy. Histologically, the lesion showed sclerotic hypocellular fibrous tissue with loss and infiltration of bone without significant cellular atypia or mitoses. Skeletal

Figure 1 CT facial bones showing destruction of the anterior maxillary bone, as well as the central superior cortex and the hard palate. A) Coronal view. b) Axial view

64 | ANZAOMS PREVIEW VERSION

He underwent a re-excision of the hard palate lesion, with additional margins taken from the right inferior turbinate, left inferior turbinate and posterior septum via a facial degloving approach. This offered better access than a revision Weber Ferguson approach to the site of recurrence in the cartilaginous septum, and also avoided scarring through the initial healed surgical site. The soft palate was spared. Intra-operatively, the lesion was difficult to ascertain from post-surgical scar. This had been anticipated, and for this reason, the patient was planned for a delayed reconstruction until margins were proven to be clear. In the interim, a palatal obturator prosthesis was fabricated, Figure 2 Histology of desmoplastic fibroma

and retained by circumzygomatic wires during this process (Figure 3). Histopathology showed an infiltrative submucosal infiltration of paucicellular fibrous tissue with

muscle and respiratory mucosa were seen adjacent

whorling architecture and some myxoid stromal changes.

to the lesion confirming destruction of cortical bone.

The deep aspect of the lesion infiltrated through skeletal

Immunohistochemistry was negative for S100, SMA,

muscle and showed destruction of bone. This suggested

MDM2 and beta-catenin. This result was suggestive of

recurrence of the desmoplastic fibroma. Margins were

desmoplastic fibroma (Figure 2).

positive on the left and right along the posterior half of the

Given this result, the patient underwent an anterior subtotal

recurrence and post-surgical changes.

maxillectomy via a bilateral Weber Ferguson approach and immediate free fibula osseomyocutaneous flap reconstruction (Figure 3). This approach facilitated cutting guides and virtual surgical planning (VSP), as well as assisting with the free fibular reconstruction. Histology the surgical specimen revealed a 23 x 14mm mass, supporting the diagnosis of desmoplastic fibroma with positive posterior margins. There was no extension beyond cortical bone in this specimen. Given positive margins, radiotherapy was offered to reduce recurrence, but the patient failed to attend.

specimen. Anteriorly, it was difficult to distinguish between

He underwent three further re-excisions until clear margins were achieved. An obturator was placed with circumzygomatic wires to allow speech and function during this time. At this point, reconstruction options involved another fibula free flap reconstruction or a zygomatic prosthesis. Given the improved surveillance for further recurrence possible with a retained prosthesis, he had quad zygomatic implants inserted. He continues to be followed up closely and was free of recurrence eight months following the last excision.

The patient was subsequently lost to follow-up for five months. On re-presentation, there was a firm bony prominence on the left side of the palate seen on examination. Repeat CT showed a stable post-operative appearance without evidence of recurrence but due to ongoing clinical concerns of recurrence the patient underwent a PET scan and biopsy of the new lesion. The PET scan revealed a focal intense region of FDG avidity in the anterior and right sided maxilla which was suggestive of either recurrence or post-surgical inflammatory changes. Histopathology from the biopsy showed stroma of low cellularity with a focally nodular sclerotic appearance without dysplastic appearance, with differentials including recurrence or scar formation. To further characterise the lesion, he also underwent MRI which demonstrated a rind

Figure 3 Intra-operative clinical photograph of bilateral Weber

of soft tissue of low T1/T2 signal intensity wrapping around

Ferguson approach after completion of maxillectomy, showing

the right fibular free flap reconstruction contiguous with a

surgical defect (published with patient consent)

high T2 signal mucosal thickening.

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DISCUSSION Desmoplastic fibroma, as classified by the World Health

lymphoma and leiomyosarcoma of bone.7,10 This low

Organization (WHO), is a locally aggressive but benign

intensity on T2 images is believed to be due to the relative

neoplasm of bone, accounting for less than 0.1% of all

acellularity of these lesions in DF, similar to that seen in

primary bone tumours. It is thought to be the intraosseous

aggressive fibromatosis.10 Positive Emission Tomography

counterpart of desmoid-type fibromatosis (aggressive

(PET) is not often used as a diagnostic imaging modality,

fibromatosis), and most commonly affects the mandible,

however a case report of desmoplastic fibroma of the

as well as other long bones of the body including the

scapula revealed moderate focal fluorodeoxyglucose (FDG)

femur and tibia.4 Reports of DF in the maxilla, such as that

uptake.11

presented in this case, are rare. In a review of cases of desmoplastic fibroma in the jaw in 2006, Said-Al-Naief et

DF remains a diagnosis of exclusion, given overlaps in

al found 86% of cases were reported in the mandible, with

imaging and histopathology and without any specific

the remainder in the maxilla. 3

immunohistochemical markers. On histopathology, it has an appearance of spindle fibroblasts in a collagenous matrix,

The most common presentation of desmoplastic fibroma

arranged in whorls or long fascicles, with minimal atypia

at all sites, including at the maxilla, is painless swelling4.

and mitoses. Cellularity may be variable. This appearance

Other signs and symptoms reported include pain and

is similar to desmoid type fibromatosis (or aggressive

bleeding. In the mandible, there have been reports of

fibromatosis).4 Importantly, there may be overlap in this

trismus, tooth mobility and displacement, facial asymmetry

appearance with low-grade osteosarcoma, which may

and symptoms mimicking odontogenic infection. 3,5 Previous

lack the typical herringbone appearance of high grade

reports have highlighted a slight female predilection,

osteosarcoma, and may not have overt atypia or mitoses.12

but there is a largely even gender distribution in cases of

It has been postulated that karyotyping and cytogenetics

desmoplastic fibroma of the maxilla. Most cases have been

may be useful in differentiating between the two, with the

reported in younger patients, with 84% of cases in the jaw

CTNNB1 (β-catenin) S45F mutation found in DF and the

being under 30 years of age. 5 The aetiology of DF remains

CDK4 amplification in osteosarcoma, however, there have

unknown. There have been some likely genetic factors

been very few reports to support this.13,14 Interestingly, it

identified, with several cases being described in patients

has also been shown that the CTNNB1 S45F mutation is

with tuberous sclerosis.6

present in desmoid-type fibromatosis.14

Imaging findings have been non-specific and varied. Most

The value of immunohistochemistry in diagnosis is unclear.

common findings in the literature describe a lytic, ill-

β-catenin as previously noted has been identified as a

defined lesion, with a previous review in 2005 describing

potential marker for DF. The connection between desmoid

lytic lesions in up to 65% of cases on CT, similar to the

type fibromatosis and β-catenin has been established.

rate of 50% identified in DF in the maxilla.7 However,

Desmoid type fibromatosis may present sporadically or

sclerotic or mixed lesions have also been reported. Cortical

as part of Gardner Syndrome, a genetic syndrome which

continuity is an important radiological sign in differentiating

includes familial adenomatous polyposis coli (FAP) and

between benign and malignant processes, as DF usually

multiple osteomas. FAP is characterised by a mutation in

demonstrates cortical thinning and expansion, while

the APC gene, which regulates the Wnt pathway which

malignancy usually produces erosion and perforation of

involves the regulation of β-catenin.15 However, while there

the cortex. However, a sun-ray appearance on CT may

have been some reports of positive staining for β-catenin in

mimic osteosarcoma, and while this may form an important

DF, it seems that the Wnt pathway is not implicated in DF,

differential, there is potential for misdiagnosis based

which distinguishes it from desmoid-type fibromatosis.6,15,16

on this feature. Other differential diagnoses based on 8

imaging findings include fibromatosis, rhabdomyosarcoma,

Surgical excision is the appropriate treatment for

neuroblastoma and lymphoma. 9

desmoplastic fibroma. Recurrence rates are reported to be high, with rates of 17% with resection and 55-72%

MRI has also been used to further characterise lesions

without resection.4 There have been varying levels of

suspicious for DF. T2 shortening (or low signal intensity)

recurrence based on surgical approach. Comparing excision

of an osteolytic intraosseous fibrous lesion on MRI may

or enucleation to wide local excision, Iwai et al found that

narrow down the differential diagnoses, as this has

no recurrence in patients with managed with wide local

otherwise only seen in giant-cell tumours, fibrous dysplasia,

excision and rates of up to 20-40% in simple excision, and

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up to 70% when treated with curettage alone. 8 Given the

There is limited data on the use of chemotherapy in

variable surgical approaches reported in the literature, there

DF, including the use of vincristine, doxorubicin and

is no consensus on the margins to aim for with resection,

dacarbazine with mixed response to treatment. 5 A recent

and given no current evidence, wide margins (>10mm)

case study of the use of vincristine, actinomycin D, and

would apply to ensure low recurrence rates. Evidently this

cyclophosphamide in a case of mandibular DF showed

needs to be weighed up against morbidity of resection on a

halted progression without resolution of the tumour.18

case-by-case basis.

The use of chemotherapy, with its significant morbidity must be carefully considered for benign disease such as

Data for use of radiotherapy and chemotherapy in

DF. The slow growth of DF may also make it relatively

desmoplastic fibroma are minimal, and most have been

chemotherapy resistant.

extrapolated from use in desmoid-type fibromatosis. With new literature now separating the two entities on

Desmoplastic fibroma of the maxilla remains a rare but

a pathophysiological basis, this may be inappropriate.

locally aggressive entity, at risk of misdiagnosis. We present

A comparative review of recurrence rates of surgery

here a case with diagnostic and management strategies to

alone and surgery with radiotherapy in desmoid-type

address the dilemmas of this challenging condition.

fibromatosis found better control in the latter group regardless of margins following surgery, however increased

Conflict of interest statement

rates of complications including pathological fractures and induction of osteosarcoma were identified with the use of radiotherapy.17

All authors have no real or perceived conflicts to declare in the publication of this paper.

Recurrent desmoplastic fibroma of the maxilla: Case report and review of the literature Referen ce s 1.

Jaffe HL. Tumors and tumorous conditions of the bones and joints. Academic Medicine 1959;34(1):72.

Griffith JG, Irby WB. Desmoplastic fibroma: Report of a rare tumor of the oral structures. Oral Surg Oral Med Oral Pathol 1965;20(2):269-75.

Said-Al-Naief N, Fernandes R, Louis P, Bell W, Siegal GP. Desmoplastic fibroma of the jaw: A case report and review of literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2006;101(1):82-94.

Fletcher CDM, Bridge JA, Hogendoorn PCW, et al. WHO Classification of Tumours of Soft Tissue and Bone. International Agency for Research on Cancer (IARC) (UN). Lyon, France. 2013.

Woods TR, Cohen DM, Islam MN, Rawal Y, Bhattacharyya I. Desmoplastic fibroma of the mandible: A series of three cases and review of literature. Head Neck Pathol 2015;9(2):196-204.

Fahmy MD, Gupta A, Padilla RJ, Segura A, Brookes CD. Desmoplastic fibroma associated with tuberous sclerosis: Case report and literature review. Oral Surg Oral Med Oral Pathol Oral Radiol 2019;128(2):e92-e9.

Frick MA, Sundaram M, Unni KK, et al. Imaging findings in desmoplastic fibroma of bone: Distinctive T2 characteristics. AJR Am J Roentgenol 2005;184(6):1762-7.

Iwai S, Matsumoto K, Sakuda M. Desmoplastic fibroma of the mandible mimicking osteogenic sarcoma: Report of a case. J Oral Maxillofac Surg 1996;54(11):1370-3.

Summa A, Cerasti D, Crisi G, et al. Desmoplastic fibroma of the mandible: usefulness of CT and MR imaging in diagnosis and treatment: A case report. Neuroradiol J 2010;23(1):109-13.

10.

Vanhoenacker F, Hauben E, De Beuckeleer L, et al. Desmoplastic fibroma of bone: MRI features. Skeletal Radiol 2000;29(3):171-5.

11.

Okubo T, Saito T, Takagi T, et al. Desmoplastic fibroma of the scapula with fluorodeoxyglucose uptake on positron emission tomography: A case report and literature review. Int J Clin Exp Pathol 2013;6(10):2230-6.

12.

Böhm P, Kröber S, Greschniok A, Laniado M, Kaiserling E. Desmoplastic fibroma of the bone: A report of two patients, review of the literature, and therapeutic implications. Cancer 1996;78(5):1011-23.

13.

Yoshida A, Ushiku T, Motoi T, et al. Immunohistochemical analysis of MDM2 and CDK4 distinguishes low-grade osteosarcoma from benign mimics. Mod Pathol 2010;23(9):1279-88.

14.

Song W, van den Berg E, Kwee TC, et al. Low-grade central fibroblastic osteosarcoma may be differentiated from its mimicker desmoplastic fibroma by genetic analysis. Clin Sarcoma Res 2018;8(1):16.

15.

Hauben EI, Jundt G, Cleton-Jansen A-M, et al. Desmoplastic fibroma of bone: an immunohistochemical study including β-catenin expression and mutational analysis for β-catenin. Human Pathol 2005;36(9):1025-30.

16.

Azola AM, Wartmann CT, Fischer MK, Ambro BT, Pereira KD. Desmoplastic fibroma arising from the anterior maxillary sinus in a child. Arch Otolaryngol Head Neck Surg 2012;138(9):859-62.

17.

Nuyttens JJ, Rust PF, Thomas Jr CR, Turrisi III AT. Surgery versus radiation therapy for patients with aggressive fibromatosis or desmoid tumors: A comparative review of 22 articles. Cancer 2000;88(7):1517-23.

18.

Khatib B, Pogrel M. Desmoplastic fibroma of the mandible in young children—A case series. Int J Oral Maxillofac Surg 2017;46(2):173-80.

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H I S T O R Y REVIEW ARTICLE

NEW ZEALAND PIONEERS AND LEADERS Kiwi contributions to maxillofacial surgery from the early 20th century to the present day

Snape L (MBChB, FRCS (Edin), FRCS (Eng), BDS, FFDRCS (Irel), FRACDS)†

† F ormerly Oral & Maxillofacial Surgery Unit, Christchurch Hospital, Christchurch, New Zealand

Corresponding Author: LESLIE SNAPE Email: maxfacenz@gmail.com

ABSTRACT A historical review of New Zealanders who have made a significant contribution to maxillofacial surgery over the last century.

INTRODUCTION New Zealand is a small but significant country at the southern edge of Oceania and the Asia-Pacific region. Its population is small, comprising 11% of Oceania and 0.01% of Asia. New Zealanders, however, have made a significant contribution to international knowledge and achievements, including a major contribution to the development of maxillofacial surgery. The impetus to development in the 20 th Century was sadly through world conflict, this being particularly so in maxillofacial surgery. The industrialised slaughter in the First World War resulted in a predominance of head and face gunshot wounds in the trenches, and in the Second World War, major burns in aeroplane crashes.1 The Germans had well-advanced medical teams which they had developed in the Franco-Prussian War of 1870. The British were slower, but by 1916 had developed specific Maxillofacial Units. These were established and led by New Zealanders who were known as the ‘Kiwi Big Four’, comprising Gillies, McIndoe, Pickerill, and Mowlem.2,3,4

Keywords: New Zealand | maxillofacial surgery | history | leadership

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PIONEERS Henry Percival Pickerill (1879-1956) was born in the United Kingdom (UK) and completed dental, medical and some surgical training there. In 1907, at the age of 28 years, he was appointed the first full-time Dean of the Dental School, at the University of Otago, Dunedin in New Zealand. At the start of World War I he was appointed to lead the NZ Medical Corps and establish a Maxillofacial Unit at Queens Hospital in Sidcup (south east London), in parallel with Harold Gillies for the British (see below for more on Gillies). In 1919 Henry Percival Pickerill (1879-1956)

he sailed back to New Zealand with his staff and remaining patients who were needing further reconstruction. He continued to work at Dunedin Hospital, had a brief time in Sydney, Australia, and in 1939 established the first New Zealand Cleft Unit. He was accompanied by his second wife Cecily, who was herself a surgeon. Together they pioneered the then novel, but now widely accepted, concept of mothers ‘living in’ and nursing their babies while they underwent staged operations. This dramatically reduced the spread of infection. 5,6

Sir Harold Gillies KBE (1882-1960) was born in New Zealand but travelled to the UK for his medical and surgical education. In 1910 he commenced practice as an ear, nose and throat surgeon. At the commencement of World War I, he appreciated the need for maxillofacial units and established the British Unit at Queens Hospital in Sidcup. There was a huge influx of badly wounded patients following the disastrous Battle of the Somme in 1916. Gillies developed the pivotal concept of introducing distant vascularised tissue to Sir Harold Gillies KBE (1882-1960)

reconstruct large soft-tissue defects, a technique known as the ‘tubed pedicle’. A record of these cases was illustrated by Henry Tonks, and dental and facial models were made by dental members of the team. Much later, many of these records were found in a cupboard in the Dunedin Dental School by Professor Sandy Macalister, who then returned them to the UK. They are now housed in the Royal College of Surgeons, London with some also kept in the Hocken Library in Dunedin, New Zealand. In the 1930s, Gillies practised in London and became known as the ‘Father of Plastic Surgery’. He first described the temporal approach to elevation of depressed zygomatic fractures and completed the first English description of the LeFort I osteotomy. He also designed many surgical instruments which are still used today.7,8

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PIONEERS

Archibald McIndoe (1900-1960) was born in New Zealand and studied medicine at Otago, New Zealand. In 1924 he travelled to the Mayo Clinic in the United States, where he spent four years as a General Surgical Fellow. Following this, in 1930 he joined his cousin, Harold Gillies, and learnt plastic surgical techniques. At the outset of World War II, he established the Airforce Burns Unit at the Queen Victoria Hospital in East Grinstead, East Sussex, UK. This became world renowned for the management of severely injured pilots and flight crew. These brave men started the support group known as the ‘Guinea Pig Club’. Archibald McIndoe (1900-1960)

McIndoe pioneered many novel treatments, including saline baths for the initial treatment of burns. This was based on the observation that airmen who ditched in the sea recovered better than those who ditched on land. He was the first overseas-born surgeon to be elected Vice President of the Royal College of Surgeons of England, and in 2014 a bronze statue of McIndoe was erected by grateful patients in East Grinstead.

Rainsford Mowlem (1902-1966) was born in New Zealand and completed his medical degree at Otago University. He then travelled to the UK in 1929, where he completed his surgical Fellowship, and later worked with Harold Gillies. In World War II he worked in the British Maxillofacial Surgery Unit. Although less famous than the other three, after the war he developed the Maxillofacial Unit at Mount Vernon in London, UK. He was the first to describe Rainsford Mowlem (1902-1966)

the use of cancellous bone chips in the reconstruction of mandibular defects and revolutionised the management of osteomyelitis in the jaw by decortication. 9,10

Sir William Manchester KBE (1913-2001) was born in New Zealand and completed his initial medical training in New Zealand. He then trained with Gillies, McIndoe and Mowlem in the UK. In 1950 he established the Plastic Surgery Unit at Middlemore Hospital, Auckland, NZ. He covered the full scope of plastic surgery; however, he and his team were particularly influential in the management of cleft palate internationally. He developed a technique for surgical repair of bilateral cleft lip and palate which is still used by many surgeons today.11 The technique is now modified by the Delaire Sir William Manchester KBE (1913-2001)

functional technique of muscular rings. In the period of 1960-1990, there was a large dissemination of knowledge in oral and maxillofacial surgery, and a massive increase in scope. Consequently, there were ‘turf wars’ in many parts of the world largely driven by other surgical specialities. These became the key challenge to leaders in that period.

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Tom Crewe (1926-1999) was born in New Zealand and graduated in dentistry from the University of Otago. He then travelled to the UK to sit the Fellowship in Dental Surgery of the Royal College of Surgeons of England (FDS, RCSEng) with the intention of returning home to New Zealand. However, he decided to stay in the UK and settled in Plymouth. Although quietly spoken, he described himself as “avant garde and heretic”, but actually he was a man before his time. Many surgeons were using plaster head caps for (ineffective) external fixation of mid-face fractures. Tom recognised that halo frames, like in cervical spine fractures, were Tom Crewe (1926-1999)

a better form of stable mid-face fixation. Modified halo frames are used today for mid-face distraction.12 Tom’s enduring legacy is in the management of orbital fracture which was at the time considered beyond the scope of oral surgery or not requiring fixation. Tom developed the current standards of orbital floor injury management.13,14 He was the first overseas-born President of the British Association of Oral and Maxillofacial Surgeons.

FOUNDATION MEMBERS OF ANZSOS, THE FORERUNNER OF ANZAOMS Sir John Patrick Walsh KBE (1911-2003) was born in Australia and completed dental and medical degrees at the University of Melbourne. In World War II he served as a surgeon in the Royal Australian Airforce at the Repatriation Hospital in Adelaide. In 1956 he was appointed Dean of the Dental School at Otago University. He had not applied for the position, so was surprised when it was offered. On arrival in Dunedin, he described himself as the “Brash Australian” and his mantra, which he espoused to his students was “service, co-operation, the open Sir John Patrick Walsh KBE (1911-2003)

mind and the hard road”. His first task was to get government funding for a new Dental School building, which he achieved in 1962. He was a man who inspired many – Edwards and Goss included – and was a font of ideas. One was the development of the air turbine drill, for which he was awarded a Doctorate of Dental Surgery from the University of Melbourne. This drill was the forerunner of drills now used in orthopaedics and oral and maxillofacial surgery. Walsh was a leading figure in dental and medical politics, served on the Dunedin City Council and led the battle for fluoridation of the water supply. His memory is celebrated in the ‘Sir John Walsh Oration’ of the New Zealand Branch of ANZAOMS. He was Knighted for his services to dentistry.15

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FOUNDATION MEMBERS OF ANZSOS, THE FORERUNNER OF ANZAOMS

Alton Donald “Sandy” Macalister (1924-1992) was born in New Zealand and graduated dentistry from Otago University. He was a very likeable teacher and clinician at the School, teaching dentoalveolar surgery to generations of students. He was promoted to Professor of Oral Surgery in 1972. He was elected the fourth President of the International Association of Oral and Maxillofacial Surgeons in 1974. In 1989 he located a cupboard full of Gillies and Pickerill-era illustrations and models, which he presented to the Sidcup Hospital, Alton Donald “Sandy” Macalister (1924-1992)

where it is known as the ‘Macalister Archive’. He was a keen Rotarian, President of the Dunedin Club and supporter of the Otago University Rugby Club.

Donald B Adams was born in New Zealand, graduated Dentistry from Otago University and was the first Master degree in Oral Surgery graduate under the supervision of Sir John Walsh. He established a private practice in Wellington, New Zealand, where he had many patients who were Parliamentarians or members of the public service. He was a supreme political operator, and in his quiet and effective way helped the needs or oral and maxillofacial surgery in New Zealand with organisations such as Donald B Adams

Pharmac, the Accident Compensation Corporation, pathology services and much more.

Ian Donaldson (1925-2007) was born in Australia, was a graduate in dentistry at the University of Sydney and then completed his FDS RCSEng. He came to Dunedin in New Zealand to teach oral medicine and diagnosis with Walsh and Macalister. He was always immaculately dressed and polite in all circumstances. He was the first New Zealand President of ANZSOS, from 1976 to 1978.

Ian Donaldson (1925-2007)

Clifford Black (1929-2017) was born in New Zealand and graduated dentistry from Otago University. He completed the FDS RCSEng. He then worked for five years in Perth, Western Australia before settling back in private and public specialist practice in Auckland, New Zealand. He was elected the second New Zealand President of ANZAOMS (1985-1987). His particular challenge was to lead the negotiations between ANZAOMS and the Royal Australasian College of Dental Surgeons to establish the Division of Oral Clifford Black

and Maxillofacial Surgery of the RACDS, which became the Board of Studies in

(1929-2017)

2003.

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William Marsden Bell (1933-2023) was born in New Zealand and graduated in dentistry from the University of Otago. He then travelled to the UK to complete the FDS RCSEng. Whilst there, he attended the first meeting of the International Association of Oral and Maxillofacial Surgeons in London in 1962. He then attended every meeting of the International Association until his last in 2015. He was Chair of the International Association Foundation Committee from 2001-2002.

William Marsden Bell (1933-2023)

Marsden’s main claim to fame, besides his surgical skills, was his administrative excellence. He was the Honorary Secretary of ANZAOMS for the Presidencies of both Ian Donaldson (1976-1978) and Cliff Black (1985-1987). In 1988 he became the Foundation Chair of the Division of Oral and Maxillofacial Surgery of the RACDS. In his 10 years as Chair, along with committee members Frank Monsour, Bob Cook, Alastair Goss and John Norman, he established the current structure of the Board of Studies and the FRACDS(OMS).

David Ernest Poswillo CBE (1927-2003) was born in New Zealand and studied dentistry at the University of Otago. He then completed the FDS RCSEng. In 1953 he returned to Christchurch, New Zealand to be the oral and maxillofacial surgeon at Burwood Hospital’s Plastic and Maxillofacial Unit. Besides his surgical work, he meticulously carried out animal research into the aetiology of cleft lip and palate in his garden shed. He defined a number of pharmacological agents responsible for cleft and other orofacial deformities.16 David Ernest Poswillo CBE (1927-2003)

In 1969 David was offered a Hunterian Professorship at the Royal College of Surgeons of England, and subsequently the Chair of Teratology. He published further research on the pathogenesis of numerous craniofacial syndromes, including Treacher Collins and craniofacial microsomia.17 David relocated briefly to Australia to be the Foundation Professor of Oral and Maxillofacial Surgery at the University of Adelaide. He then returned to London as Professor at Guys and St Thomas Hospital. He was the Secretary General of the International Association of Oral and Maxillofacial Surgeons for six years, and became the second overseas born President of the British Association of Oral and Maxillofacial Surgeons. Clinically he popularised the use of cryosurgery and described in the Englishspeaking literature the use of the zygomatic hook for elevation of the zygomatic bone. He was Chair of several scientific health committees in the UK, including ‘Smoking and Health’, as well as authoring the Poswillo Report on anaesthesia, sedation and resuscitation, which proved to be a watershed in the improvement of safety in dental treatment.

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LEADERS John Edwards ONZM (1940-2022) was born in New Zealand and graduated in dentistry from the University of Otago in 1963. He went to the UK for postgraduate training and completed his FDS RCSEng. He returned to Dunedin in 1969 as oral and maxillofacial surgeon to the Dunedin Public Hospital and Senior Lecturer at the School. In 1987 he moved to Auckland as Head of Oral and Maxillofacial Surgery at the Auckland Hospitals. He was also in private practice with Marsden Bell.

John Edwards ONZM (1940-2022)

John was a very astute leader and committee man, who served in numerous roles for New Zealand dentistry and oral and maxillofacial surgery. He was Chair of the ACC/ANZAOMS Committee on Temporomandibular Joint Replacement. John was the third New Zealand President of ANZAOMS (1997-1999) and was made an Officer of the Order of Merit in New Zealand in 2008. Shortly after he retired from clinical practice, he died of an aggressive cancer. In his memory his family established the John Edwards Memorial Lecture, the first of which was presented by Dr Lou Mercuri.

Alastair Goss was born in the United Kingdom, migrated to New Zealand as a 13-year-old and graduated in dentistry from the University of Otago in 1966. He then headed briefly to the UK, before going to the USA for surgical and research training. In 1970 he obtained a junior position at the University of Adelaide and has stayed there, working firstly with David Poswillo, then Henk Tideman, before he was appointed Professor and Director of Oral and Maxillofacial Surgery in 1988. He was awarded a Doctorate in Dental Surgery from the University of Otago in 1981 for a dissertation on cleft palate. His first challenge as Director was to successfully resist the attempts by Plastic Surgery to take over Oral and Maxillofacial Surgery. Alastair Goss

Alastair is an excellent organiser and committee man. He was the Honorary Secretary of ANZAOMS (1988-1991), Foundation Board Member of the Division of OMS (1988-1996) and elected member of the RACDS Council (1994-2004), where he became the Censor in Chief. His current roles for ANZAOMS are the Honorary Archivist and Temporomandibular Joint Replacement Adviser to the ANZAOMS TMJR Register. It has been said that he has “printers’ ink in his veins”, as he has published approximately 300 scientific publications, including books. He was co-author of the History of ANZAOMS and is the Foundation Editor of the Australasian Journal of Oral and Maxillofacial Surgery.18

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LEADERS

This entry was contributed by the Editor (AJOMS).

Les Snape was born in the UK and completed his dental and medical degrees there. Interestingly, he completed two years of basic surgical training in Christchurch, New Zealand before returning to the UK to complete his higher surgical training. He was in the first cohort to complete the FRCS(Edin)(OMFS) in 1985. He then took up a public and private hospital consultancy in Christchurch. He was Director of Training in NZ from 1999 to 2011, and Chair of the FRACDS (OMS) Les Snape

examiners (2002-2011). He was the fourth New Zealand President of ANZAOMS (2011-2013). During this period he was instrumental in lobbying for recognition of the speciality with the Medical Council of New Zealand, and establishing salaried registrar positions for approved trainees in New Zealand. Lately he has run the trainee days at meetings of the ANZAOMS NZ Branch, been involved as international faculty for numerous instructional courses for AO in the Asia Pacific region and carried out voluntary support at a Craniofacial Clinic in Nepal.

Jeremy McMahon was born in New Zealand and completed his dental and medical degrees at the University of Otago. He then completed his higher surgical training in the UK. He was Head and Neck Fellow at the Chris O’Brien Lifehouse Centre in Sydney, Australia, before returning to the UK. His particular interest is oncology, and currently he works in Glasgow. He was Clinical Lead for the BAOMS initiative ‘Quality Outcomes in Oral and Maxillofacial Surgery’, which leads to quality care for our patients. Jeremy McMahon

Jocelyn Shand was born in New Zealand and completed her dental degree at Otago University. She then travelled to Melbourne, where she completed her medical degree and higher training leading to the FRACDS(OMS). Currently she is Head of Oral and Maxillofacial Surgery at the Royal Children’s Hospital of Melbourne, with a special interest in paediatric patients. She was the President of ANZAOMS (2009 -2011), Chair of the Board of Studies, Chair of the ANZAOMS Research & Education Foundation and has just been Jocelyn Shand

elected Vice President of the IAOMS.

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Summary

Acknowledgements

This review confirms the major contribution that New

I acknowledge the work of the Editor of AJOMS, who kindly

Zealanders have made to oral and maxillofacial surgery,

wrote the section on Les Snape. Henry Pickerill portrait with

both regionally and internationally. Many challenges have been met, but there still remains the need for new leaders to step up and resolve issues as they arise.

permission of the Alexander Turnbull Library, Wellington. Sir Harold Gillies portrait with the permission of the National Portrait Gallery, London.

“We are like dwarfs, perched on the shoulders of giants. We see more, and things that are more distant, than they did, not because we have keener vision or greater height, but because we are lifted up and borne aloft on their gigantic stature.” John of Salisbury, theologian and author, in a treatise on logic called Metalogico written in Latin in 1159.

New Zealand Pioneers and Leaders. Kiwi contributions to maxillofacial surgery from the early 20th century to the present day Referen ce s 1.

Stoelinga PJW, Williams J Ll. 50 years of IAOMS: The development of the specialty. 2012. International Association of Oral and Maxillofacial Surgeons.

2. 3.

Brown E, Klaassen MF. War, facial surgery and itinerant Kiwis: The New Zealand plastic surgery story. Australas J Plastic Surg 2018;1(1);51-63. Tong DM, Bamji A, Brooking T, Love RM. Plastic Kiwis-New Zealanders and the development of a specialty. J Military Veterans’ Health 2008;17(1);11-18.

Morrison M. Plastic surgery origins and the Antipodean influence. Australas J Plastic Surg 2018;1(1);6-16.

Brown H. Pickerill: Pioneer in plastic surgery, dental education, and dental research. Otago University Press. Dunedin, NZ. 2007.

Fowler PV, Snape L, Thomson JMD. The history of cleft services in New Zealand. NZ Dent J 2021;117;179-184.

Bamji A. Sir Harold Gillies: Surgical pioneer. Trauma 2006;8(3);143-56.

Gillies H. Temporal approach for elevation of the zygomatic arch. Br J Surg 1927;24;652-656.

Mowlem R. Iliac cancellous chip grafts for the restoration of mandibular bone defects. Proc R Soc Med 1945;38(4);171-174.

10.

Mowlem R. Management of osteomyelitis of the jaw. Proc R Soc Med 1945;38(8);452-452.

11.

Manchester WM. The repair of bilateral cleft lip and palate. Br J Surg 1965;52(11);878-82.

12.

Crewe TC. A halo frame for facial injuries. Br J Oral Surg 1966;4;147-149.

13.

Crewe TC. Significance of the orbital floor in zygomatic injuries. In J Oral Surg 1978;7(4);235-239.

14.

Crewe TC. Facts and fallacies of orbital floor injury. Int J Oral Surg 1981;10(1);225-228.

15.

Brown H. Sir John Walsh and his legacy to the Dental Profession. New Zealand Dental Association. Auckland. 2017.

16.

Poswillo D. Observation on fetal posture and causal mechanisms of congenital deformities of the palate, mandible and limbs. J Dent Res 1966;45;584-596.

17.

Poswillo D. The aetiology and surgery of cleft palate with micrognathia. Ann R Coll Surg Engl 1968;43(2);61–88.

18.

Goss A, Linn R. Extractions to reconstruction: The development of oral & maxillofacial surgery in Australia and New Zealand. 2015. Historical Consultants Pty Ltd.

76 | ANZAOMS PREVIEW VERSION

S C O P E A N D W O R K F O R C E ANZAOMS REPORT

SURVEY OF AUSTRALIAN AND NEW ZEALAND ORAL AND MAXILLOFACIAL SURGEONS 2020 Forty years on monitoring scope and workforce

Ricciardo PV (BSc, BDSc, MBBS, FRACDS (OMS), MRCPS)†

ABSTRACT

Vujcich NJ (BDSc (Hons), MBBS (Hons), FRACDS (OMS))†

Introduction:

Bobinskas AM (MBBS, BOH, GradDip (Dent), MPhil, FRACDS (OMS), FRCS (OMFS))‡ Goss AN (DDSc, FRACDS (OMS))§

† D epartment of Oral & Maxillofacial Surgery, Royal Perth Hospital, Perth, Western Australia, Australia

Australian and New Zealand oral and maxillofacial surgeons have conducted detailed scope and workforce studies over the last 40 years. This paper presents the 2020 results and compares them to past results and the international scene. Method:

‡ C anberra Hospital, Australian Capital Territory, Australia

An electronic survey consisting of over sixty questions was forwarded

§ Oral & Maxillofacial Surgery Unit, Adelaide Dental School, University of Adelaide, Adelaide, South Australia, Australia

Zealand.

to all registered oral and maxillofacial surgeons in Australia and New

Results: One hundred and ninety-five surveys were forwarded and complete

Corresponding author: PETER RICCIARDO Royal Perth Hospital Perth, Western Australia, Australia Email: petericciardo@hotmail.com

replies received for 116: a response rate of 59%. Conclusion: After 40 years oral and maxillofacial surgery in Australia and New Zealand is achieving its goal of being a well-qualified surgical speciality with a wide scope delivering high quality services to the community. Its workforce privately is well balanced but with a need for more in public practice.

INTRODUCTION

The specialty of oral and maxillofacial surgery (OMS) in Australia and New Zealand has undergone major changes in the last 40 years. Training in the 1960s and 1970s was individual, without standardisation, and this was subject of criticism from the medical surgical specialties. A full review of training by the education subcommittee of the Australia and New Zealand Association of Oral and Maxillofacial Surgeons (ANZAOMS) found that the training was predominantly university-based and built upon a dental degree. There was wide variation in not only qualifications but also duration of training. A review published in 1982 titled Oral surgery training in Australia and New Zealand: A plan for the eighties made three recommendations: a joint advisory committee should be set up to standardise training and specialist requirements across Australia and New Zealand; a survey of all current OMS specialist, trainees, and training programs should be performed; and the minimum requirement for training should be established.1 The process of implementing these recommendations has extended ever since. In 1988, the Board of Studies in OMS of the Royal Australasian College of Dental Surgeons, (RACDS) was established and the requirements for the fellowship [FRACDS)(OMS)] were developed. This involved accreditation of training centres and trainees. Initially this was dentally based, but by 1994 training required registrable, medical and dental degrees, (dual degree) and hospital based surgical training for a minimum of four years, with the exit examination for Fellowship, the FRACDS(OMS).2 This model has been refined progressively since then. OMS received recognition as a principal surgical speciality in 1998 from the Commonwealth Government of Australia. The pathway has not been straightforward and has been driven by the leaders of the speciality. The full story is the subject of a book titled From extraction to reconstruction. The development of oral and maxillofacial surgery in Australia and New Zealand.3 Monitoring of progress has been a key component. The baseline studies were in the subcommittee report and a postal survey of all full members of ANZAOMS in 1986.1,4 The effect of training on scope from the same dataset was analysed separately. 5 In 1986, only eight registered specialists had the FRACDS, DOS – the Diploma in Oral Surgery which was the forerunner of the FRACDS(OMS) – and of these, six were dentally qualified and two were dually qualified. These studies showed that, in the 1980s, most oral and maxillofacial surgeons (89%) were dentally qualified with four years of postgraduate training (81%). A smaller subset had both medical and dental degrees (dual, 11%) and only 19% had less than three years of training. In general, the greater the training, the greater the scope of practice. Dentoalveolar surgery, particularly for those in full-time private practice, was the bulk of practice scope.6 The second group of studies relate to the period 1986-1995. During this period, the national dual degree program with a minimum of four years training was instituted. These studies compared those who had recently completed their training and those who were currently in training. By 1996, 33% of recent specialists were dual degree FRACDS(OMS) and 84% of the trainees were on a dual degree FRACDS(OMS) track.7,8 An integrated logbook of surgical experience of trainees was also evaluated. 9

78 | ANZAOMS PREVIEW VERSION

The extent of continuing professional development (CPD) of the whole workforce was also investigated.10 A detailed workforce evaluation including the projected needs for

RESULTS

the OMS workforce was carried out by the Australasian Institute of Health and Welfare, Dental Statistics and

One hundred and sixteen useable replies were received

Research Unit, which is based at The University of

from the 195 questionnaires sent out. The response rate of

Adelaide.6 All of these studies were sponsored by

59% was less than previous studies which received 83%,

ANZAOMS and funded by its Research and Education

70%, and 77% responses.4,5,7,8

Foundation.11-13 The 2011 workforce study found a broadening scope of

Procedure

2011

2020

Orthognathic

67%

98%

Mandibular osteotomy

95%

Maxillary osteotomy

93%

Segmental maxillary osteotomy

81%

Although Australia and New Zealand are neighbouring

Subapical osteotomy

28%

countries with many similarities there are also some marked

Zygomatic osteotomy

47%

differences in the provision of health services. These have

Temporary Anchorage Device

80%

Ablative

53%

39%

and maxillofacial surgeons in Australia and New Zealand.

Neck Dissection

18%

Comparison is made to past surveys and the international

Microvascular

literature.

Salivary gland

54%

87%

Submandibular

46%

72%

Sublingual

45%

77%

Parotid

10%

22%

Skin Pathology

60%

50%

Arthrocentesis

73%

72%

reference group, refined and forwarded by group email to

Open Joint Surgery

70%

47%

all full members of ANZAOMS. The survey was open for

Total Joint Replacement

28%

a period of two months, and two reminders were sent to

Aesthetic Surgery

28%

potential participants.

Blepharoplasty

Facial Feminisation

21%

practice related to the dual degree requirement. The study also confirmed that increased training numbers were required to keep up with the increase in population and the number of general health practitioners.14

been separately explored and documented in a recent study.15 The aim of the present study was to document the current training, scope, and workforce situation for oral

16-18

METHODS A detailed 60-question questionnaire was developed based on previous questionnaires. It was trialled by a small

Malignant Pathology

TMJ

Procedure

2011

2020

Facial Implants (alloplastic)

Dentoalveolar

99%

Rhinoplasty

11%

41%

Rhytidectomy

99%

Injectables

15%

28%

Tooth transplant Dental Implants

88%

Craniofacial Implants

56%

38%

Cleft Lip and Palate Surgery

Open sinus lift

94%

Primary Palate and Lip

Trauma

77%

89%

Secondary Grafting

27%

60%

Mandible

52%

89%

Secondary Growth/Orthognathic

27%

67%

Maxilla

66%

88%

Craniofacial

Nasal

61%

77%

Craniofacial Implants

30%

38%

Zygoma

51%

85%

Cranioplasty

Orbital

42%

80%

Distraction Osteogenesis

27%

75%

Facial implants (alloplastic)

28%

Frontal

33%

Table 1 Scope of surgery performed in 2020 is compared to 201115

NQ: Question not asked in the 2011 questionnaire.

ANZAOMS PREVIEW VERSION |

Female surgeons were slightly under-represented (10%)

37% in 2011. Orthognathic surgery included bimaxillary

in the survey compared to 13% ANZAOMS membership.

advancement surgery for obstructive sleep apnoea. There

This proportion is similar to the United Kingdom in 2020,

was also an increase in head-and-neck cancer surgery

recording 13.2% female OMS consultant workforce, while

including both ablative and reconstructive. A number of

a United States sample survey had a response of 20.5%

surgeons were trained in microvascular surgery.

female surgeons.16-18 There was an increase in temporomandibular joint Sixty-seven per cent of surgeons were born in either

replacement (from 4% in 2011) with surgeons across

Australia (53%) or New Zealand (14%), and 94% of

all states offering this procedure. Twenty-eight per

surgeons qualified in Australia or New Zealand. Eighty-four

cent of surgeons now perform TMJ replacement, with

per cent of surgeons have the FRACDS (OMS) qualification.

7% of surgeons operating on more than 10 cases per

This has increased from 77% in 2011, and correlates

year. This has led to ANZAOMS, in association with

with the retirement of surgeons with prior recognition of

the Australian Orthopaedic Association, establishing a

qualification – particularly Master qualifications. Eighty-

temporomandibular joint replacement registry comparable

eight (76%) surgeons had both medical and dental degrees

to the national hip and knee registries.

plus the FRACDS(OMS). Ten years prior, only 42% were There has been a marked increase in interest in aesthetic

dual-qualified. 8

surgery with 33 (28%) surgeons being actively involved This pattern is similar in the USA. OMS residency is

in placement of facial implants, dermal fillers, rhinoplasty,

accredited by the Commission on Dental Accreditation

rhytidectomy and liposuction. Only 13% of surgeons were

(CODA), dual-degree surgeons often practice under the

involved in aesthetic surgery in 2011.14

auspices of both Doctor of Dental Surgery/Doctor of Dental Medicine and Doctor of Medicine (MD) licensures. In 1986

The greatest number of surgeons were interested in

there were five dual-degree residency programs; 30 in

increasing their subspecialty skills in aesthetic surgery

1990; 46 today, along with 54 single-degree programs.

Sixty-four (54%) surgeons had post-training fellowships of at least one-year duration. These fellowship qualifications were in trauma (16%), temporomandibular joints (TMJ) (15%),

(21%). Other areas of interest (in descending order) were temporomandibular joint replacement, oncology, salivary pathology, cleft, skin, orthognathic, reconstruction and craniofacial surgery.

oncology (15%), orthognathic (14%), craniofacial/paediatric

Seventy-four per cent of respondents are comfortable

(11%), head and neck reconstruction (9%), salivary gland

performing tracheostomy, and 39% have performed this

disease (7%), cleft (4%), skin (4%), facial aesthetic surgery

procedure in a theatre emergency. Forty-eight per cent of

(4%), and dentoalveolar surgery (3%). The scope of surgery performed as compared to 2011 is presented in Table 1.14 Dentoalveolar surgery remains the core scope of the speciality. Fifty-eight (50%) surgeons reported performing third molar surgery on more than 500 patients per year. Forty-eight (41%) surgeons placed more than 100 dental implants per year. Eighty-one (70%) surgeons received more than 80% of their referrals from dentists. Generally, most oral and maxillofacial surgeons participating in the survey had a wider scope of practice, with 54% of respondents proceeding to postgraduate fellowship. Eighty-one per cent of postgraduate fellowships were for one year or longer in duration, and 49% of postgraduate fellows attended two or more fellowships. Of the respondents, 54% attended a postgraduate fellowship in

respondents have performed CPR in theatre. The projected number of oral and maxillofacial surgeons in different recruitment scenarios was first presented in the workforce study based on a consensus approach projection (Figure 1).6 The maximum number of newly qualified surgeons was put at 10 per year (from 2010) whereas the current actual number is 12 to 14 per year. However, this projection stopped at 2011 when the population of Australia was projected to be approximately 20 million. The population of Australia has increased more than anticipated, with the population reaching 22.48 million in 2011 and had reached 26.3 million by 2020.19 Thus, the number of newly qualified surgeons is within the recommended limits. A further consideration is length of time to retirement (Figure 2). This study found that 25% of currently practicing surgeons intended to retire within the next five years and

the UK, 37% in Australia, and 29% in the USA.

40% within 10 years. By then the OMS surgical workforce

Forty per cent of surgeons operate more than 20

retirement of all those dental-only qualified.

orthognathic surgery cases per year, compared with

80 | ANZAOMS PREVIEW VERSION

will be homogeneous as dual-degree fellows, with the

The typical hours of work per week are presented in

Figure 3. Sixty-two per cent of surgeons felt they were as busy as

Surgeons (number) 250

they wished to be, while 24% felt they were too busy. Ten per cent of surgeons regularly perform elective

200

operations on a Saturday. Most surgeons worked 41-50 hours per week, while one third worked more

150

than 51 hours per week. Thirty-four per cent of respondents felt they were overworked. Sixty-two per cent

x e

x x

x d

c b a

100

felt that the number of surgeons in private practice was about right.

(Figure 4). The reverse was so for the perception of the number of surgeons in the public workforce with most respondents (53%) believing that there were too few (Figure 5). The number of surgeons in public employ is of particular concern in New

0 1991

1996

2001

2006

2011

2016

2021

Figure 1 Projection for the number of OMS in Australia under different recruitment models. Superior line (f) expressed an incremental increase to 10 new surgeons per year from 2010. Note: This is based on an underestimate of Australian population growth (20M) as compared to actual (22.48M) in 2010. The actual population in 2020 is 21.3M

Zealand, to the extent that a formal enquiry has been established.20 The issue in both Australia and New Zealand relates mainly to the declining number of full-time public, either academic or hospital, surgeons. There are multiple parttime public positions with 30 (26%) surgeons having academic titles and 80 surgeons (70%) being involved in OMS training. Appropriate ratios of OMS to other health professionals have been

“This year” “1-5 years” “6 - 10 years” “11 - 15 years” “16 - 20 years” “21 - 25 years” “26 - 30 years” “> 30 years”

established (Table 2). The majority of surgeons are based in the capital

10%

15%

20%

25%

Figure 2 Response to question “When do you intend to retire?”

cities and regional areas, with 10 (9%) practising entirely regionally. Overall, the majority of oral and

maxillofacial surgeons responding

– 98 (86%) – were happy with their

career and would recommend oral and maxillofacial surgery as a career

to recent graduates in dentistry or

medicine.

20 15 10 5 0 “0 - 10” “11 - 20” “21 - 30” “31 - 40” “41 - 50” “51 - 60” “61 - 70” “81 - 90”

Figure 3 Response to question “In a usual week, how many hours do you work?” ANZAOMS PREVIEW VERSION |

60%

50%

40%

30%

20%

10%

0% “Too few”

“The right number”

“Too many” Figure 4 Response to question

“Regarding the number of OMS in the public workforce, there are:”

70% 60% 50% 40% 30% 20% 10% 0% “Too few”

“The right number”

“Too many” Figure 5 Response to the question

“Regarding the number of oral and maxillofacial surgeons in the private workforce there are:”

2020

2010

Australia

New Zealand

Australia

New Zealand

Population

26.3M

5.2M

22.7M

4.0M

Number GDP

16,550

3,330

10,437

1,715

Ratio of GDP to population

1:1,590

1:1,560

1:2,156

1:2,290

Number of GMP

37,785

18,700

43,000

13,883

Ratio of GMP to population

1:696

1:278

1:522

1:310

Number of OMS

238

185

Ratio of OMS to GMP

1:160

1:480

1:235

1:385

Ratio of OMS to GDP

1:70

1:85

1:57

1:48

Table 2 Workforce of health care practitioners in Australia and New Zealand 202015 GDP: General Dental Practitioners GMP: General Medical Practitioners

82 | ANZAOMS PREVIEW VERSION

DISCUSSION This study shows that, in the last 40 years, oral and

palates as well as oral and orthognathic surgery. In

maxillofacial surgery in Australia and New Zealand

Germany, a double qualification in medicine and dentistry is

has matured from a heterogeneous, individual dental-

required.22

based training which was non-standardised, to a homogeneous dual-degree hospital-based training

With its origins in dentistry, OMFS is established as one

program at a high standard. This is reflected in the wide

of the 10 United Kingdom surgical specialties. Training

scope of the speciality. Each step along the way has been

requires dual-degree qualification, successful completion of

carefully monitored and peer reviews published.

Core Surgical Training (CST) and passing the Membership

1-15

This

documentation is unique within the full range of surgical

of the Royal College of Surgeons (MRCS) exam. This is

specialities in Australia and New Zealand. It is also unique

followed by a five-year approved training program and exit

internationally in oral and maxillofacial surgery.21

fellowship (FRCS) examination.16,17

More than 80% of the worldwide OMS groups require only

Core OMS in practice is dentoalveolar surgery, orthognathic

a dental degree (North America, Nordic countries, South

surgery, OSA surgery, surgical airway, facial trauma,

East Asia, India, Africa, Latin America, Middle East), less

benign lesions, salivary surgery and TMJ.16,17 Workforce is

than 5% are medically qualified (France, Spain), and the

a balance between recruitment, retirement and community

rest require dual qualification (United Kingdom, Australia,

need, with potential concerns regarding workforce

New Zealand, North Eastern Europe).21

sufficiency when considering population growth, surgeon retirement, and evolving workforce shortages overseas.

Ninety per cent of surgeons in this study consider OMS to be a specialty of dentistry and medicine. In the United

This study shows that oral and maxillofacial surgery in

States, OMS is a specialty of dentistry that focuses on

Australia and New Zealand has evolved considerably

the treatment of injuries, disease and defects of the head,

from 40 years ago. It is now a mature, well-qualified

neck, face, and jaws. In Germany, Oral and Maxillofacial

surgical specialty with a broad scope delivering care to its

Surgery (OMFS) is primarily a medical specialty focused

community.

on the treatment of facial injuries, trauma, oncology, reconstruction, and malformations such as cleft lip and

Survey of Australian and New Zealand Oral and Maxillofacial Surgeons 2020. Forty years on monitoring scope and workforce. Referen ce s 1.

Monsour FN, Goss AN. Oral surgery training in Australia and New Zealand. A plan for the eighties. Education Subcommittee Report. Australian & New Zealand Association of Oral & Maxillofacial Surgeons. 1982.

OMS Training Handbook. Royal Australasian College of Dental Surgeons. https://racds.org/wp-content/uploads/2023/02/RACDS_EXT_ ACA_218_3.0_Accredited-Training-in-OMS-Handbook_March-2023.pdf (Accessed 17 January 2024)

Goss AN, Linn R. Extractions to reconstruction. The development of oral and maxillofacial surgery in Australia and New Zealand. Historical Consultants Pty Ltd. Adelaide, Australia. 2015.

Goss AN, Gerke DC. The scope of oral and maxillofacial surgery in Australia and New Zealand. A postal survey. Aust Dent J 1991;36(1):57-62.

Goss AN, Gerke DC. Effect of training on the scope of oral and maxillofacial surgery. Int J Oral Maxillofac Surg 1990;19(3):184-189.

Spencer AJ, Szuster FS, Brennan DS, Goss AN. A consensus approach to projections of the supply of oral and maxillofacial surgeons in Australia. Int J Oral Maxillofac Surg 1993;22(5):314-317.

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Szuster FS, Nastri AL, Goss AN, Spencer AJ. Survey of Australian and New Zealand Oral and Maxillofacial Surgery trainees and recent specialists-education and experience. Int J Oral Maxillofac Surg 2000;29(4):305-308.

Szuster FS, Nastri AL, Goss AN, Spencer AJ. Survey of Australian and New Zealand Oral and Maxillofacial Surgery trainees and recent specialists-workforce issues. Int J Oral Maxillofac Surg 2000;29(3):227-30.

Sambrook P, Smith A, Hewitt G, Goss A. Development of a national clinical logbook system for oral and maxillofacial surgery trainees--the RACDS, OMS system. Int J Oral Maxillofac Surg 1996;25(3):242-4.

10.

Sambrook P, Goss A. Continuing education for oral and maxillofacial surgeons--new expectations. Ann R Australas Coll Dent Surg 1996;13:1937.

11.

Brennan DS, Spencer AJ, Singh KA, Teusner DN, Goss AN. Service provision by patient and visit characteristics in Australian oral and maxillofacial surgery: 1990 to 2000. Int J Oral Maxillofac Surg 2004;33(7):700-8.

12.

Brennan DS, Spencer AJ, Singh KA, Teusner DN, Goss AN. Practice activity trends among oral and maxillofacial surgeons in Australia. BMC Health Serv Res 2004;4(1):37.

13.

Szuster FSP, Goss AN, Spencer AJ, et al. Oral and maxillofacial surgeons – 1996 study of trainees and recent specialists. AIHW Catalogue N, Dent 44. AIHW Dental Statistics and Research Unit. The University of Adelaide, Australia. 1998.

14.

Ricciardo P, Bobinskas A, Vujcich N, Nastri A, Goss A. Survey of Australasian oral and maxillofacial surgeons 2011--scope and workforce issues. Int J Oral Maxillofac Surg 2015;44(12):1569-73.

15.

Bridgman JB, Fulton G, Lou SM-Y, Thomson WM, Goss AN. The New Zealand oral and maxillofacial surgeon workforce in 2017-18: characteristics, practice and prospects. N Z Med J 2020;133(1513):11-22.

16.

Magennis P, Begley A, Dhariwal DK, Smith A, Hutchison I. Oral and Maxillofacial Surgery (OMFS) Consultant Workforce in the UK: reducing consultant numbers resulting from recruitment issues, pension pressures, changing job-plans, and demographics when combined with the COVID backlog in elective surgery, requires urgent action. Br J Oral Maxillofac Surg 2022;60(1):14-19.

17. 18.

Ilankovan V. Training in the United Kingdom: are we fit for purpose? Br J Oral Maxillofac Surg 2020;58(10):1225-1228. Roudnitsky E, Hooker KJ, Darisi RD, Peacock ZS, Krishnan DG. Influence of residency training program on pursuit of academic career and academic productivity among oral and maxillofacial surgeons. J Oral Maxillofac Surg 2022;80(2):380-385.

19.

ABS Data. 2012. https://www.abs.gov.au/ausstats/abs@.nsf/lookup/3101.0Media%20Release1Dec%202011

20.

Smith M, Ferguson CA, Thomson WM. Public sector oral health service provision for high needs and vulnerable New Zealanders. Wellington and Dunedin Health Promotion & Policy Research Unit. University of Otago. New Zealand. 2019.

21.

Goss AN, Helfrick JF, Szuster FS, Spencer AJ. The training and surgical scope of oral and maxillofacial surgeons: the International Survey 1994. Int J Oral Maxillofac Surg 1996;25(1):74-80.

22.

Zeller AN, Thiem DGE, Bartella AK, et al. Training in oral and maxillofacial surgery in Germany - Residents’ satisfaction and future challenges. J Craniomaxillofac Surg 2021;49(5):415-421.

84 | ANZAOMS PREVIEW VERSION

I N A U G U RA L J O H N E DWA R D S M E M O R I A L L E C T U R E

ALLOPLASTIC TEMPOROMANDIBULAR JOINT REPLACEMENT Past, present and future considerations

Mercuri LG (DDS, MS)†‡

INTRODUCTION

† V isiting Professor, Department of Orthopaedic Surgery, Rush University Medical Centre, Chicago, Illinois, USA

Sir John Charnley, an orthopaedic surgeon from Bury, England, is

‡ A djunct Professor, Department of Biomedical Engineering, University of Illinois Chicago, Chicago, Illinois, USA

considered the Father of alloplastic joint replacement. His pioneering work, which started in the ’60s, resulted in the modern practice of orthopaedic surgery being unthinkable without such devices.1 With the advancements made in alloplastic temporomandibular joint replacement (TMJR) devices since the late ’80s, the same can be said for the use of these devices in maxillofacial surgery for the

Corresponding Author: LOUIS MERCURI Department of Orthopaedic Surgery, Rush University Medical Centre, Chicago, Illinois, USA Email: louis_g_mercuri@rush.edu

management of end-stage TMJ disorders.2, 3 The history of the use of alloplastic devices in the TMJ has had a dark past, out of which grew greater than 30 years of success with TMJR devices, however the future still presents challenges. It is the purpose of this paper to discuss the past, present and future of TMJR devices.

Keywords: temporomandibular joint (TMJ) | alloplastic TMJ replacement (TMJR) ANZAOMS PREVIEW VERSION |

THE PAST Alfred Stille said, “Medicine, like all knowledge, has a

that could only be managed by employing alloplastic TMJR

past as well as a present and a future...in that past is the

devices. However, due to the severity of the anatomical

indispensable soil out of which improvement must grow.”

architectural destruction to the host bone created by the foreign body giant cell response to the PTFE, the available

In 1840, John Murray Carnochan, a New York ENT surgeon,

stock TMJR devices (e.g. Christensen, Kent-Vitek) were

is credited with the idea of interposing material between

almost impossible to implant and stabilise properly.

the surfaces of a joint. He attempted to mobilise a patient’s ankylosed jaw by placing a small block of wood between

In 1988, Mercuri, with the assistance of the developmental

the raw bony surfaces after resection at the condylar neck.4

engineers at Techmedica (Camarillo, California, USA), created a custom computer-aided and computer-

Thereafter, single case reports surfaced about the use of

manufactured (CAD/CAM) TMJR utilising the same

materials such as ivory, gold and tantalum foil, zirconium

materials successfully utilised in orthopaedic joint

and stainless steel. The authors reported the use of these

replacement for decades. The custom nature of this

as interpositional materials in the management of ankylosis,

device allowed for implantation and stabilised in these

trauma or reconstruction after ablative tumour surgery.5

anatomically challenging cases. This device received FDA approval in 1999 as TMJ Concepts (Ventura, California,

In the early 1960s, Christensen developed the vitallium

USA); Stryker/TMJ Concepts (Ventura, California, USA)

fossa/eminence as an interpositional device for a TMJ

since 2021 (see Figure 1A).13

ankylosis case.6 When functional loading against the metal resulted in condylar bone loss, he responded with a total

In 2005, the FDA approved a stock TMJR developed by

TMJ replacement device with a vitallium ramus component

Quinn with Lorenz Microfixation (Jacksonville, Florida, USA),

and a polymethylmethacrylate (PMMA) condyle.7 When

now Zimmer Biomet (Jacksonville. Florida, USA) (see Figure

the PMMA failed under functional loading, Christensen

1B). Zimmer Biomet also manufactures a custom

converted the condyle to all metal in the later 1990s. 8 Failure of the metal-on-metal iteration led to removal of the Christensen TMJR device from the marketplace in the

TMJR which has yet to be approved for use in the US by the FDA but is available in other countries.14

2010s. George Santayana said, “Those who cannot remember the past are condemned to repeat it.” An example of this was the disaster created by the use of Proplast-Teflon, a porous form of polytetrafluoroethylene (PTFE) in the TMJ in the late 1970s and early 1980s. Charnley, in a 1963 letter to the editor of The Lancet, reported that he had abandoned the use of PTFE in 1962 because “Teflon particles found in the living tissues had produced serious tissue reactions”. 9

In 1964, Scales and Stinson reported the same findings.10 Charnley once again clearly restated this finding in a chapter entitled ‘Arthroplasty of the Hip’ in Progress in Clinical Rheumatology11; as well as in a 1966 letter to the editor of the Journal of Bone and Joint Surgery12. The developers of TMJ Proplast-Teflon devices and the US FDA obviously failed to remember or find these earlier reports concerning this material before approving its use in the late 1970s in the TMJ, under an FDA’s substantial equivalency exemption that was then in place. Ultimately, over 20,000 TMJ devices containing ProplastTeflon were implanted, resulting in significant TMJ anatomical damage and masticatory functional disorders

86 | ANZAOMS PREVIEW VERSION

B Figure 1. Temporomandibular joint replacement systems. A) Stryker/TMJ Concepts. B) Zimmer Biomet.

THE PRESENT

THE FUTURE

Due to the many clinical reports of success reported with

Peter Drucker said, “The best way to predict the future is

the use of the two FDA-approved TMJR devices, in 2019

to create it.” For medical devices, this involves research

there were 15 countries producing 27 TMJR devices. Of

to improve the materials, designs and manufacturing

concern was that only 44% presented any preclinical

processes involved in TMJR devices. The term ‘embodiment’

laboratory data, 33% had yet to report clinical outcomes, and only 15% had been given regulatory body approval.15 By 2021, there were now 20 countries manufacturing 41 TMJR devices (nine stock and 32 custom designed) with 12 companies using additive manufacturing (3D printed) for

includes device materials, design and manufacturing. There is a delicate balance amongst these three elements because the failure of any one of them leads to failure of the device.16 Looking to the future of TMJR, each of these elements must be judged.

the metal components of their devices.

MATERIALS ASTM International, formerly known as the American

Titanium alloy

Society for Testing and Materials, is an international organisation that develops and publishes voluntary

Despite the fact that Ti6AlV4 has been the major alloy

consensus technical standards for a wide range of

used in implantable metal devices, concern over reports of

materials, products, systems, and services. The ASTM

the alloy’s aluminium and/or vanadium content leading to

International assigns a number to each material technical

hypersensitivity has led to investigation of novel titanium

standard.

alloys with similar or improved mechanical and clinical properties by incorporation of non-toxic alloying elements

Because of their biocompatibility and strength, cobalt

such as niobium, molybdenum, tantalum, zirconium and tin

chromium (CoCr – ASTM F1537) and titanium (Ti6AlV4

in the formulation of the alloy.20-24

– ASTM F136) are the two metal alloys used in the manufacture of TMJR devices.

So-called beta titanium alloys also have the theoretical advantage of a lower elastic modulus and therefore lower

Cobalt chromium

stress-shielding at implant-bone interfaces.25-28

Because of its high strength, temperature endurance and

Ultra-high Molecular Weight Polyethylene

wear resistance against polyethylene, CoCr is commonly

(UHMWPE)

alloyed with molybdenum and traces of other elements when used to manufacture TMJR and orthopaedic implants.

UHMWPE has demonstrated the potential for enhanced biomechanical interactions in orthopaedics over the past

However, the residual nickel component (<1%) of the

six decades. With respect to UHMWPE’s biocompatibility,

CoCrMo alloy is often considered the culprit when material

especially those associated with osteolysis, and revision

hypersensitivity becomes a concern. Therefore, researchers

rate the peri-prosthetic response can be improved by

in both alloplastic orthopaedic and TMJR are seeking to

modifying surface and bulk properties. Notably, XL-

develop new nickel-free alloys with similar biologic and

UHMWPE and Vitamin-E reinforced XL-UHMWPE both

bioengineering characteristics.17,18

show promising clinical outcomes.29-33

The nickel-free, high-nitrogen stainless steel

Additive manufacturing (AM) (3D printing) of UHMWPE

(Fe18Cr14Mn3.5MoN0.9) being studied at Rush, is one

implants appears to be a promising approach. AM

example of such a material.19 This, or a similar Ni-free alloy,

technologies reduce several processing steps in

may be the future replacement for CoCr in both orthopaedic

manufacturing complex-shaped implants. However,

and TMJR devices.

strategies such as fused deposition modelling and selective laser sintering are not yet successful for developing UHMWPE implants, and more studies are required to ANZAOMS PREVIEW VERSION |

optimise 3D printing of UHMWPE and to validate the

alternative to uncoated metal implants, for patients with

biocompatibility of the resulting 3D printed implants. 34

metal hypersensitivity.

Alternate Bearing Surfaces

Commercially available coatings for implants include

Ceramics (alumina, zirconium) components are commonly used in total hip systems because of their favourable wear properties and chemically inert nature. However, there are difficulties designing this material into TMJR bearing surface. 35,36

titanium nitride (TiN), titanium niobium nitride (TiNbN), oxidised zirconium (OxZr), and zirconium nitride (ZrN). Current research is focused not only on these, but also on diamond-like carbon (DLC), silicon nitride (SiN), chromium nitride (CrN) and tantalum-based coatings (TaN and TaO), plasma immersion ion deposition (PIID), and plasma-

Polyether-ether-ketone (PEEK), due to its structure, confers

enhanced magnetron sputtering (PEMS). 38-40

outstanding chemical resistance, inertness, and thermal stability for in vivo conditions. However, despite the good

The coating materials referred to above that are still at the

in vitro testing in low-contact stress situations, there are

research stage have been shown to be noncytotoxic and to

questions about its suitability under high-contact stress. 37

reduce wear in a laboratory setting. However, the adhesion of implant coatings remains a main area of concern, as poor

Coatings

adhesion can cause delamination and excessive wear

Chromium alloy levels measured in the plasma of patients

(Figure 2).

were lower and nickel allergy symptoms were relieved. Therefore, coated implants could be considered an

Figure 2 SEM image of the condylar surface of explanted TMJR demonstrating areas of cracking and loss of the TiN coating exposing the Ti alloy surface 2 years after implantation.41 Reproduced from: Kerwell S, Alfaro M, Pourzal R, Lundberg HJ, Liao Y, Sukotjo C, Mercuri LG, Mathew MT. Examination of Failed Retrieved Temporomandibular Joint (TMJ) Implants. Acta Biomaterialia 32:324-335, 2016.

88 | ANZAOMS PREVIEW VERSION

DESIGN

Stock TMJ Devices

Custom TMJ Devices

Must make fit

Made to fit

3 all UHMWPE fossa sizes

CAD/CAM Ti metal backed UHMWPE (Stryker/TMJ Concepts)

3 Co-Cr ramus lengths, 2 widths

CAD/CAM Ti alloy (Stryker/TMJ Concepts)

components. This is based on previous literature as well

Off the shelf

Must be ordered

as the available refereed and edited literature to date that

Longer surgical time

Shorter surgical time

Bone must be removed at interface

No bone removal at interface

stable, improved long-term outcomes over stock devices.15

Difficult to gain primary stability

Easy to get primary stability

Brown et al. reported that custom TMJR devices should be

Potential for micromotion

Little or no potential for micromotion

Placement variability

No placement variability

Limited use for large defects

Accommodates large defects

No fossa posterior stop

Fossa has a posterior stop

While there is definitely a place for stock devices in certain clinical situations, because of the advantages for the use of custom TMJR devices listed in Table 1, 78% of the present manufacturers of TMJR devices marketed as of 2021 use a custom design for their fossa and ramus/condyle

demonstrates that custom TMJR devices, by their nature, design and biomaterial composition, appear to provide

the standard of care in cases where there is a significant deviation from normal anatomical form or a large change in mandibular position (Table 1).42

Table 1 Stock vs Custom Temporomandibular Joint Replacement43

MANUFACTURING 70% of TMJR metallic components were either cast or

microstructure has a direct effect on a material’s physical

wrought, 30% are additively manufactured (3D printed).15

and mechanical properties.44

Cast alloy components are the result of an investment-

This study compared the electrochemical behaviour of

casting process where the metal solidifies to its final shape.

wrought and additively manufactured Ti6Al4V alloys

Wrought components are milled from a stock plate or bar of

and found that the wrought alloys were superior with

the metal that has been pressed through a set of dies while

respect to the corrosion rate, polarisation resistance, and

the material is in a semi-molten or plastic state. Devices

capacitance.44

manufactured using wrought materials (e.g. Ti alloy) have a more homogenous and organised grain structure than cast

Therefore, it will be important for future designers and

devices (e.g. CoCr).

manufacturers to determine the best microstructure to provide biocompatibility, mechanical strength, wear and

Additive manufacturing (AM) is the industrial production

corrosion resistance, especially with the introduction of

name for 3D printing, a computer-controlled process that

AM components that require a critical evaluation of the Ti

creates three-dimensional objects by depositing materials

alloy microstructure to prevent potential premature implant

in layers.

failures.

Neto et al. demonstrated that metal AM is still at its early

Concepts for artificial intelligence (AI) designed patient-

stage of development, and the fundamental processing-

specific (custom) devices with geometrically congruent

microstructure-property relationships are not fully

bearing surfaces to assure minimal functional loading wear

understood. 44 Without optimised processing parameters, defects can often occur in parts produced with AM. In this study, such

are under consideration. Additive manufacturing (3D/4D printing) of instrumented active device components to monitor performance and signal early failure are also under investigation.45

defects were shown to have led to failures of AM TMJR parts (Figure 3). Therefore, these authors concluded that ANZAOMS PREVIEW VERSION |

Figure 3 Electron Backscatter Diffraction (EBSD) images of the structure, phase (α & β), and grain orientation map differences between present day wrought titanium alloy and 3D printed TMJR ramus components. The left wrought panels demonstrate homogeneous and well oriented grain structure and the proper ratio of the hard (α) and soft (β) phases of titanium alloy. The 3 right panels demonstrate 3 failed 3D printed TMJR ramus components with disorganised, needlelike grain structure prone to failure under functional loading, and a phase map showing hard (α) and soft (β) phase rations further prone to fracture under functional loading.44

FUTURE POST-IMPLAN TATION DEVELOPMENTS Periprosthetic Joint Infection (PJI)

(Ahsg), a key player in the stabilisation and clearance of

immunosuppression

the amorphous mineral precursor phases of excess mineral from the circulation, has shown the potential to inhibit the

The most common orthopaedic and TMJR device postimplantation complication is infection.

development of HO. 51,52

The improper use

and overuse of antibiotics has added to the development

High-frequency stimulation of bone to increase

of the increasing prevalence of antimicrobial-resistant

osseointegration

bacteria. This, and the fact that the development of new antibiotic classes has been stagnating, has limited

Bone is a tissue for which mechanical stimuli are crucial for

the options for dealing with bacterial infections. The

maintaining its structure and function. Bone cells react to

development of a humanised monoclonal antibody that

their biomechanical environment by activating molecular

targets a universal component of bacterial biofilms, leading

signalling pathways, which regulate their proliferation,

to rapid biofilm collapse by engaging three modes of action

differentiation, and matrix production. Alloplastic

– the sensitisation of bacteria to antibiotics, host immune

device implants influence the mechanical conditions in

enablement, and the suppression of site-specific tissue

the adjacent bone tissue. Optimising the mechanical

inflammation – offers a potential future solution.47,48

properties of these devices can promote bone regeneration. Therefore, external biomechanical stimulation applied to

Heterotopic ossification prevention and

improve implant osseointegration using vertical whole-

prophylaxis

body low-magnitude high-frequency vibration has been shown to accelerate bone regeneration. 53 In the future, this

The second-most common complication after TMJR surgery

technology applied to orthopaedic and TMJR implants could

is heterotopic ossification (HO) at 3-5%.49 HO, also known

result in earlier integration of the fixation elements, leading

as heterotopic bone formation, involves the development

to greater long-term stability and viability for these devices.

of ectopic lamellar bone in soft tissues as the result of inflammation. 50 Despite literature discussing HO risk factors and diagnosis, the etiology of HO, its prevention and management still remain abstract. However, research into the use of a naturally occurring glycoprotein, Fetuin-A

90 | ANZAOMS PREVIEW VERSION

Acoustic emission feedback to monitor for early

• Develop a TMJR Registry like the orthopaedic AJRR

component failure

• Better understanding of diagnosis and management of PJI

Acoustic emission (AE) is the phenomenon of sonic and

• Develop strategies to manage and control heterotopic ossification

ultrasonic wave generation by materials as they undergo the processes of deformation and/or fracture. AE monitoring is widely used throughout civil and mechanical engineering as a sensitive and non-destructive technique for structural

• Better understanding of material sensitivity • Develop TMJR revision and replacement criteria and strategies

integrity.54,55 In alloplastic joint replacement devices,

• Role of eTMJR in management for mandibular reconstruction

fixation screw loosening leads to micromotion, osteolysis,

• Indications for use of TMJR in the growing patient

implant fatigue and possible implant fracture with catastrophic implant failure. Screw loosening also prevents osseointegration necessary for component stability and implant success. In order to prevent complete implant failure, and complicated revision surgery, detection and diagnosis of screw loosening is important. Acoustic emission is being studied as a non-invasive diagnostic approach to early detection of implant fixation loosening. 56

Other TMJ-related considerations for future investigation • Better understanding of TMJ disease processes • Better understanding of TMJ biomechanics

The goal of every bioengineer, designer, manufacturer and clinician involved with the development of future TMJR devices must take to heart what C. William Pollard said: “The arrogance of success is to think that what you did yesterday will be sufficient for tomorrow.” This is the only way we can assure that these devices will continue to be safe and effective in the management of end-stage TMJ disease for our patients.

Conflict of Interest Statement Professor Mercuri is a Clinical Consultant for Stryker/TMJ Concepts, Ventura, California, USA.

• Appropriate in vitro biomechanical studies • Prospective clinical outcome studies with strict inclusion and exclusion criteria

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T E M P O R O M A N D I B U L A R J O I N T CASE REPORT

A RARE COMPLICATION OF SPHENOID SINUSITIS: Septic arthritis of the temporomandibular joint

Jensen ED (BDS, BScDent (Hons), DClinDent (Paed))†‡ Foreman A (BPhysio, BMBS (Hons), PhD, FRACS)§ Cheng A (MBBS, BDS, FRACDS (OMS))†‡

ABSTRACT Septic arthritis of the temporomandibular joint is uncommon and is either from haematogenous, contiguous, or direct spread of microbial pathogens. Diagnosis involves clinical examination, imaging,

† O ral & Maxillofacial Surgery Unit, University of Adelaide, South Australia, Australia

histopathology, and microbiology. In the case presented, it was

‡ O ral and Maxillofacial Surgery Unit, Royal Adelaide Hospital, South Australia, Australia

the medial aspect of the temporomandibular joint. Treatment involved

epartment of Otolaryngology, D Head and Neck Surgery, University of Adelaide, South Australia, Australia

demonstrated that the source was a sphenoid sinus infection which spread along the emissary vein, through the lateral pterygoid muscle to endoscopic sinus surgery, drainage of the infratemporal fossa and temporomandibular joint arthrocentesis.

INTRODUCTION Septic arthritis of the temporomandibular joint (TMJ) is a rare condition characterised by infection of the joint space. Infection is thought to

Corresponding Author:

arise by haematogenous or contiguous spread, or direct inoculation of

ANDREW CHENG Oral and Maxillofacial Surgery Unit Royal Adelaide Hospital Adelaide, South Australia, Australia Email: ahacheng@hotmail.com

the joint in a trauma or surgical setting.1 Direct spread of pathogens is less common than haematogenous spread from a distant infected site, but can be found in patients with concurrent infection, usually from otologic or odontogenic sources. Acute otitis media has been a site of concurrent infection in reported cases of septic arthritis of the TMJ; including a case which had an abscess in the infratemporal space as an acute complication.2 Less commonly, acute otitis externa has also been reported as a concurrent infective site. 3 Odontogenic infections, specifically maxillary tooth abscess or post-extraction sites, have also been implicated.4-6 While septic arthritis of the TMJ from otologic and odontogenic infection have been described, there are no reports of contiguous spread through the sphenoid sinus. This case report aims to present a case of septic arthritis of the TMJ likely to have resulted from

Keywords:

contiguous spread via the sphenoid sinus, presenting an uncommon

septic arthritis | temporomandibular joint |

etiological pathway for this rare condition. It provides valuable insights

sphenoid sinusitis

into its clinical presentation, diagnostic challenges, and management. ANZAOMS PREVIEW VERSION |

CASE REPORT A 20-year-old male presented with right-sided earache

Complete opacity of the right sphenoid sinus had occurred.

which had exacerbated following a three-hour domestic

The right lateral pterygoid muscle was bulky compared

flight. The pain did not respond to oral antibiotics, so the

to the left. There was an effusion within the superior joint

patient was referred to an oral and maxillofacial surgeon

space of the right temporomandibular joint (Figures 2a-b).

as he now had severe right jaw pain, with difficulty opening and closing his mouth. Medically he had a history

A combined operation plan consisting of right uncinectomy

of thalassemia minor and a three-year history of cigarette

and middle meatal antrostomy, posterior ethmoidectomy,

smoking. He had no prior history of temporomandibular

sphenoidotomy and ethmoid bulla was performed by

joint dysfunction.

the otolaryngologist. The polyps were submitted to haemopathology and confirmed to be benign inflammatory

Detailed imaging confirmed acute right posterior ethmoid

polyps. The microbiology showed gram-negative bacilli

and sphenoid sinusitis with evidence of spread to the infratemporal fossa. It was noted that there was significant penetration of the lateral recesses of the sphenoid in the bone between the sinus and the infratemporal fossa (Figures 1a-b).

Figure 2a-b Magnetic resonance imaging of the individual’s face and skull base in a) axial and b) sagittal planes with orientations of A, anterior; P, Figure 1a-b Computed tomography images of the individual’s face in axial planes with orientations of A, anterior; P, posterior; R, right; L, left. a) complete opacification of the sphenoid sinuses bilaterally is noted (white arrows); b) contrast is used for this slice with bulkiness of the right lateral pterygoid muscle (black arrows) seen

94 | ANZAOMS PREVIEW VERSION

posterior; R, right; L, left; H, head; F, foot. a) Turbo spin echo (TSE) view is used for this slice with bulkiness of the right lateral pterygoid muscle (white arrows) with large collection seen and the unaffected left lateral pterygoid for comparison (dashed white arrow); b) T1-weighted contrast is used for this slice with visualisation of the infective process on the superior aspect of the temporomandibular joint space (black arrow)

The oral and maxillofacial surgeon drained 4mL of purulent liquid from the infratemporal fossa and placed a drain. The right temporomandibular joint was aspirated, and 3mL of pus removed (Figure 3). Similar gram-negative bacilli were confirmed. He was kept on intravenous amoxicillin with clavulanic acid for an additional 24 hours. He was discharged on a one-week course of oral amoxicillin with clavulanic acid. At one week postoperative review, all symptoms had resolved including jaw and sinus pain, returning to normal mouth opening and occlusion. Figure 3 Intraoperative photograph during aspiration of the right temporomandibular joint with septic arthritis; the aspirate from the joint space was creamy yellow pus

DISCUSSION Preauricular pain and limitation of jaw opening are common

posterior section of the ethmoidal sinuses was consistent

presentations in oral and maxillofacial surgery practice,

with contiguous spread. Pus collection within the lateral

usually relating to temporomandibular joint osteoarthritis

pterygoid muscle and a lesser extent to the medial

or internal derangement. However, one does need to be

pterygoid muscle then occurred, in addition to the sub-

aware of unusual conditions which may present with similar

masseteric space. The infection then tracked through the

symptoms. These include oropharyngeal neoplasms and

incomplete medial TMJ capsule. It is speculated that the

infection.

three-hour domestic flight at reduced air oxygen may have flared the infection.

Septic arthritis of the TMJ from contiguous spread has been reported previously from otologic and odontogenic

Almost all previously reported cases of septic arthritis

sources, but the presentation following sphenoid sinusitis is

of the TMJ have described diagnostic needle aspiration

unusual. Previous reports have presented with significant

of the TMJ with subsequent cultures. Management has

bony destruction and negative cultures which suggested

included arthrocentesis and arthroscopy most commonly,

advanced disease by the time of diagnosis.7,8 In this report,

but incision and drainage has been reported in a number

early diagnosis and management led to a short recovery

of case reports.1,3-4 In this case, we report an immediate

time and a minimal risk of negative long-term sequelae.

improvement in symptoms post-operatively, suggesting

Negative long-term outcomes reported in the literature

early diagnosis and management can lead to significantly

include persistent pain, limited range of movement,

reduced morbidity for the individual.

persisting malocclusion, erosive changes to the condylar head, fibrosis, osteoarthritis, and ankylosis. 9

Acknowledgement

Most likely the mode of transmission for this individual

The patient gave written consent for this case report.

involved the initial infection in the sphenoid sinuses, with transmission through the valveless system of the sphenoidal emissary vein (vein of Vesalius), which commonly travels through the sphenoidal emissary

Conflict of interest statement

foramen.10 Spread of infections through the emissary veins

The authors declare no conflicts of interest related to this

have been reported previously, typically to the maxillary

case report.

sinus, ethmoidal cells, nasopharynx, soft tissues of the face and intracranially.11 The opening of the sphenoid sinuses articulate with the ethmoidal labyrinth; therefore, the radiographic finding of opacification in the right ANZAOMS PREVIEW VERSION |

A rare complication of sphenoid sinusitis: Septic arthritis of the temporomandibular joint Refere n ce s 1.

Omiunu A, Talmor G, Nguyen B, et al. Septic arthritis of the temporomandibular joint: a systematic review. J Maxillofac Oral Surg 2021;79(6);12141229.

Bast F, Collier S, Chadha P, Collier J. Septic arthritis of the temporomandibular joint as a complication of acute otitis media in a child: a rare case and the importance of real-time PCR for diagnosis. Int J Pediatric Otorhinolaryngol 2015;79(11);1942-1945.

Thomson H. Septic arthritis of the temporomandibular joint complicating otitis externa. J Laryngol Otol 1989;103(3);319-321.

Bounds G, Hopkins R, Sugar A. Septic arthritis of the temporo-mandibular joint—a problematic diagnosis. Br J Oral Maxillofac Surg 1987;25(1);6167.

Moses JJ, Lange CR, Arredondo A. Septic arthritis of the temporomandibular joint after the removal of third molars. J Maxillofac Oral Surg 1998;56(4);510-512.

Gams K, Freeman P. Temporomandibular joint septic arthritis and mandibular osteomyelitis arising from an odontogenic infection: A case report and review of the literature. J Maxillofac Oral Surg 2016;74(4);754-763.

Leighty SM, Spach DH, Myall RW, Burns JL. Septic arthritis of the temporomandibular joint: review of the literature and report of two cases in children. In J Maxillofac Oral Surg 1993;22(5);292-297.

Chaves Netto HD, Nascimento FFAdO, Chaves MdGAM, et al. TMJ ankylosis after neonatal septic arthritis: literature review and two case reports. Maxillofac Oral Surg 2011;15;113-119.

Cai X-Y, Yang C, Zhang Z-Y, et al. Septic arthritis of the temporomandibular joint: a retrospective review of 40 cases. J Maxillofac Oral Surg 2010;68(4);731-738.

10.

Leonel LCPC, Peris‐Celda M, de Sousa SDG, Haetinger RG, Liberti EA. The sphenoidal emissary foramen and the emissary vein: anatomy and clinical relevance. Clini Anat 2020;33(5);767-781.

11.

Turton N, McGoldrick DM, Walker K, Martin T, Praveen P. Septic arthritis of the temporomandibular joint with intracranial extension: A case report. J Maxillofac Oral Surg 2022;21(1);120-123.

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T R A U M A SCIENTIFIC ARTICLE

SIXTY YEARS ON

Reflections of a major maxillofacial trauma centre

Bordbar P (BDSc, MBBS, PGradDip (SurgAnat), MDSc, FRACDS (OMS), FRCS (Ed))† Manchella S (MBBS, BDS, FRACDS (OMS))† Delpachitra SN (MBBS (Hons), BDS, MPH, MBA, FRACDS (OMS))† Singleton C (BS, MBBS, BDS, PGradDip (SurgAnat))†

ABSTRACT Introduction: Maxillofacial trauma, either isolated or in combination with other injuries, represents a significant disease burden globally.

O’Donnell M (BSc (Hons))† Nastri A (MBBS, MDSc, FRACDS (OMS))†

Objectives: The purpose of this research study was to report on the modern epidemiology, management, and outcomes of operative facial trauma

† D epartment of Oral & Maxillofacial Surgery, Royal Melbourne Hospital, Parkville, Victoria, Australia

over a five-year period at the Royal Melbourne Hospital (RMH) Oral and Maxillofacial Surgery (OMS) unit, and contrast this against the comparative data published by our surgeons at various time points over the last 60 years. Specifically, our aim was to evaluate the major

Corresponding Author: SETH DELPACHITRA Department of Oral & Maxillofacial Surgery Royal Melbourne Hospital Parkville, Victoria, Australia Email: seth.delpachitra@unimelb.edu.au

advances in local policy and technology over this time period, and to what extent such changes influenced the presentation and operative management of oral and maxillofacial trauma. Methods: This single-centre, retrospective analysis was based upon the population of patients presenting to Royal Melbourne Hospital over a period of 2011-2016 with facial fractures, and who underwent assessment and management by the Oral and Maxillofacial Surgery service. The records of all patients included in the study were reviewed for demographic data, details of presentation, mechanism of injury, and associated systemic injuries. Results and conclusion: The operative management of facial trauma at this institution has evolved significantly, mirroring the advances in technology worldwide, as well as the shift away from closed reduction to open reduction and internal fixation. The introduction and rapid uptake of 3D virtual surgical planning will no doubt continue to revolutionise the management of maxillofacial trauma in the decades to come.

Keywords: maxillofacial trauma | demographics | oral surgery techniques | complications ANZAOMS PREVIEW VERSION |

INTRODUCTION

Maxillofacial trauma, either isolated or in combination with other injuries, represents a significant disease burden globally. Untreated injuries to the facial skeleton can result in disfigurement and functional impairment, with substantial psychosocial morbidity.1,2,3 This necessitates the presence of a specialist maxillofacial surgery service in tertiary trauma centres, for assessment and management of trauma patients. 3 Several epidemiologic studies investigating maxillofacial trauma have been published in the literature, demonstrating significant locoregional variation in aetiology, demographics, and trends over time.1,5,6,7 Furthermore, recent advances in management of facial trauma, such as a shift to miniplate osteosynthesis, and an overall increase in use of open reduction and internal fixation, have fundamentally changed the mechanics of treatment planning and provision of surgical treatment, as well as the nature and frequency of complications. 8,9 Sound, evidence-based public policy decisions require up-to-date and regionallyrelevant research data. Analysis of this data is important for both resource allocation at the hospital and departmental level, but also more broadly in risk mitigation at a primary prevention level. The Royal Melbourne Hospital (RMH) is one of the largest trauma hospitals in Australia, and the only adult Level One trauma centre which provides an oral and maxillofacial surgery service in the state of Victoria. Historic research publications on the aetiology and management of maxillofacial trauma by RMH oral and maxillofacial surgeons were instrumental in the development of both public policy changes regarding compulsory seat belt legislation, as well as the development of novel techniques for midfacial fracture management.10,11 In this context, the purpose of this research study was to report on the modern epidemiology, management, and outcomes of operative facial trauma over a five-year period at the RMH OMS unit, and contrast this against the comparative data published by our surgeons at various time points over the last 60 years. Specifically, our aim was to evaluate the major advances in local policy and technology over this time period, and to what extent such changes influenced the presentation and operative management of oral and maxillofacial trauma.

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METHODS This single-centre, retrospective chart review was based upon a population cohort who:

Presented to Royal

Sustained facial trauma,

Underwent assessment

Received operative

Melbourne Hospital

either isolated or in

by the Oral and

management for their

between 1 January 2011

combination with other

Maxillofacial Surgery

facial injury.

and 31 December 2016;

injuries;

service; and,

During the study period, 3,930 patients presented with facial

face, midface, lower face) as well as anatomic conventions

trauma. Of these, 2,000 cases were referred to the OMS

described by the AO Foundation. Details regarding operative

service, and 878 went on to receive operative intervention.

management, including operative time, operative technique,

The remaining cases were conservatively managed and thus

and admission/discharge data were collected from operative

excluded from this study. Two patients presented twice during

notes and patient management software. Complications were

the study period and required operative management both

classified based upon the well-established and validated

times – these patients were counted twice for epidemiologic

Clavien-Dindo system of surgical complications, a five-grade

analysis. Fourteen patients required more than one operation

classification system based upon the type of therapy needed

as a result of their presenting injuries but were only counted

to correct the complication.12

once in the analysis. Data was collected and tabulated by author SM using Microsoft Excel.

The records of all patients included in the study were reviewed for demographic data, details of presentation, mechanism of injury, and associated systemic injuries. All bony facial injuries

Ethical approval for this study was obtained via the RMH

identified were confirmed on radiologic imaging. Facial

Human Research Ethics Committee (HREC Reference

injuries were classified based on facial zones/thirds (upper

#QA2016115).

RESULTS Demographics From a gender perspective, overwhelmingly, the population group of this study was dominated by

350

300 Assault/IPV 250 Sporting and leisure

men (740 male versus 138 female). Analysis of the data by age revealed that younger adults were

200

Road trattic accident

also overrepresented; the median age of patients undergoing operative management of facial trauma was 29 (range: 15-87 years), and 58% of all cases occurred in patients between the ages of 21-40 (Figure 1).

150

Falls

100

Work-related

Self-harm

Other <20

21-30

31-40

41-50

51-60

61-70

71- 80

>80

Figure 1 Mechanism of injury by age

ANZAOMS PREVIEW VERSION |

Mechanism The most common mechanism of injury for the entire cohort was

Males (n = 740)

Assault/IPV

Females (n = 138)

interpersonal violence (39%),

Falls

followed by sporting/leisure (25%) and road traffic accidents (18%).

Other

Other mechanisms by order of Road traffic accident

frequency included falls (12%), work-related injuries (3%) and self-

Self-harm

harm (1%). Within the male cohort, interpersonal violence remained

Workplace accidents

the most common mechanism; in the female cohort, sporting/leisure

Figure 2 Mechanism of injury by gender

related mechanisms were most likely (Figure 2). Injury mechanisms were attributed to each facial third and summarised in Figure 3. 100% 90% 80%

70% 60% 50%

60%

40% 30% 20%

49%

10% 0% 41-50

51-60

61-70

71- 80

th er oa d ac tra ci ffi de c nt Se lfha rm Sp or tin g le an is d ur e W o ac rk ci pla de c nt e s

31-40

Fa l

21-30

t/I

<20

Figure 3 Injuries by facial third

Type of injury and patterns In the analysis of our dataset, an ‘isolated maxillofacial

A stricter definition for panfacial trauma was applied in

injury’ was defined as trauma which was confined to the

this study than has been used in some historic studies. In

facial skeleton only, whilst a ‘multi-trauma’ required at

this study, the term ‘panfacial’ was reserved for injuries

least one other body system to be involved. These other

involving all facial zones (upper, middle and lower) and

body systems were categorised as (in order of frequency):

nine cases satisfied this criterion. Five were caused by

upper torso, intracranial, upper limb, cervical spine, lower

attempted self-harm, two from road traffic accidents, one

limb, lower torso. Majority of operative cases were isolated

from assault and one from a fall. Seven of the nine cases

trauma (76%). Isolated maxillofacial injuries were mostly

had concomitant injuries reflecting the greater forces

associated with interpersonal violence (45%), whereas

involved (the attempts at self-harm were ballistics injuries

multi-traumas were typically associated with road traffic

and falls from great height). Five of these cases required

accidents (47%).

surgical tracheostomies and four of the cases required multiple (>3) surgeries to address all the facial injuries.

100

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Referra l Pa tte r n s

100% 90%

Fifty-four per cent of cases presented directly to RMH via the emergency

Assault/IPV

80%

department; 36% presented to other

70%

hospitals and were subsequently transferred as an inpatient; 7% were

60%

referred to outpatient clinic via a

50%

general medical practitioner; 2%

Falls Other

were referred to outpatient clinic

40%

Road traffic accident

via a general dental practitioner;

30%

Self-harm

and 1% of patients were referred to

20%

outpatient clinic via a private surgeon

Sporting & leisure

10%

(Figure 4). Most (94%) of cases presented within 48 hours after

time of injury; as was expected, late

Work-related GDP

presentations were far more likely to

GMP

Other Hospital

RMH

Specialist

be isolated maxillofacial injuries. Figure 4 Referral patterns

Time to Surgery Median time from admission to surgery was three days

Patients for whom surgery was performed on the same

(range: 0-29 days). There were three distinct patient groups

admission as their initial presentation were grouped into

identified based on the timing of operative management

an ‘inpatient surgery’ group. Patients for whom surgery

relative to the initial admission: those admitted following

was performed on a separate admission to their initial

initial presentation who underwent surgery during

presentation were grouped into the ‘outpatient’ surgery

that admission; those admitted following their initial

group. The average total length of stay for the inpatient

presentation but who underwent surgery as outpatients

group was 6 days (range: 0 - 66) and the outpatient group

(that is, during a separate admission); and those who did

was one day (range: 0-7) (Figure 6).

not require admission for their initial presentation and had surgery as outpatients. The median time from referral to surgery for these groups was 2, 10 and 7 days respectively (Figure 5). 30

Days

X 5

X Admit + Inpt Sx (n=489)

Admit + Outpt Sx (n=115)

No Admit, Outpt Sx (n=260)

Figure 5 Time to surgery

ANZAOMS PREVIEW VERSION |

101

Middle Third

70 60

Days

50 40 30 20 10

Orbit – ≥2 wall defect

- Reconstruction – titanium

- Reconstruction – porous polyethylene

- Reconstruction - composite

- Reconstruction – bone graft

- Conservative

Maxilla – Le Fort I

- ORIF +/- IMF

- Conservative

Maxilla – Other

- ORIF +/- IMF

- Closed reduction/IMF

Outpatient Surgery

- Conservative

NOE/Nasomaxillary

- ORIF

- Closed reduction

A total of 1,472 facial fractures were identified in the study.

- Medial canthopexy (wire, bar, plate)

Fracture subunits and their management are listed in

- Conservative

Table 1. Eighty-three patients (9%) had significant

Le Fort II

associated dentoalveolar injuries requiring extraction or

- ORIF +/- IMF

splinting and 196 (22%) had significant associated soft

- Conservative

tissue injuries requiring debridement and repair. In the

Le Fort III

midface, the zygoma was the most commonly operated

- ORIF +/- IMF

midfacial subunit, followed by the orbit (33% and 19% of

- Conservative

Isolated nasal bone

- MUA

- ORIF

- Conservative

LowerThird

695

Frontal bone – anterior table only

Mandible - angle

198

- ORIF

196

- Conservative

- Conservative/closed reduction

Frontal bone – anterior and posterior table

Mandible - parasymphysis

158

- ORIF

156

X 0 Inpatient Surgery Figure 6 Total length of stay

Type of Operation

midfacial injuries, respectively). In the mandible, the angle was the most commonly operated subsite, followed by the parasymphysis (28% and 22% of mandible injuries respectively). Upper Third

- ORIF

- Cranialisation

- Conservative

Middle Third

- Conservative/closed reduction

Mandible - condyle - neck/subcondyle/extra-capsular

145

- ORIF

739

- Conservative/closed reduction

113

ZMC

227

Mandible - body

- ORIF

200

- ORIF

- Reduction without fixation

- Conservative/closed reduction

- Conservative

Mandible - condyle - head/ intra-capsular

- Conservative/closed reduction

Mandible - symphysis

- ORIF

mandible - ramus

- ORIF

- conservative/closed reduction

Mandible - Coronoid

- conservative

ZMC – isolated arch

- Reduction without fixation

Orbit – 1 wall defect

128

- Reconstruction - titanium

- Reconstruction - porous polyethylene

- Reconstruction - composite

- Conservative

Table 1 Incidence of fractures by anatomic location and management

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Operative Complications

Long-Term Outcomes

Three intraoperative complications were noted, comprising

Four hundred and fifty-one patients (51%) had no issues at

two IMF screw fractures and one surgical drill fracture.

two-year follow-up. Three hundred and thirty-nine patients

These were addressed intra-operatively and did not result in increased morbidity to the patient or prolonged hospital stay.

(39%) failed to attend their post-operative appointments and were lost-to-follow up. The remaining 10% of patients experienced postoperative issues, including anaesthesia of the infraorbital or inferior alveolar nerves, scarring, malocclusion, epiphora, diplopia and infected metalware.

Six cases required return to theatre for re-operation

Although these issues resulted from a combination of

(Clavien-Dindo Grade IIIb). Three of these cases were for

the original injury as well as the surgeries performed, in

revision of orbital plates, one for re-fixation of mandible

51 patients (5.8%), they were significant enough to be

fractures due to malocclusion, one for drainage of a haematoma and one for retrieval of loose screw. Four cases had events resulting in prolonged hospital stay (Clavien-

considered complications and required unplanned reoperation. Of these re-operative cases, the most common causes were infection including infected non-union and infected metalware (49%); ocular symptoms such as

Dindo Grade II). One patient who was managed by both the

diplopia, proptosis, enophthalmos or entropion (20%);

Neurosurgical and Maxillofacial Surgery Units for a frontal

dental malocclusion (18%); and aesthetic concerns/scar

bone and orbital roof fracture was monitored for suspected

(10%).

CSF leak which resolved spontaneously. Another patient had post-operative hypoxia requiring re-intubation and transfer to ICU for delayed extubation, with subsequent

Four deaths were noted during the study period. Three were multi-traumas with significant injuries to multiple body systems, where the patients died during their

full recovery. A third patient developed Clostridium

admission due to neurological deterioration. The other

difficile infection requiring IV antibiotics, and the fourth

death was in a patient with isolated facial trauma, who

patient developed a wound infection requiring medical

developed massive pulmonary embolus and subsequent

management with multiple bedside washouts.

cardiorespiratory arrest three weeks following discharge from hospital. The recency of death following discharge from hospital prompted a formal coronial inquest, finding that the thromboembolic event was likely due to factors unrelated to the patient’s hospital stay, and that appropriate thromboembolism prophylaxis strategies were employed.

DISCUSSION A journey back in time to the late 1950s reveals a surgery

severely comminuted and displaced panfacial fractures

and trauma landscape far different to the present.

were a typical presentation for maxillofacial surgery units.

The rapid rise in automobile transport in Australia had

Hueston & Cook, both consultant surgeons at RMH at the

unfortunately arrived with a commensurate rise in

time, published their series of severe middle-third facial

associated road fatalities and major injuries. Despite

fractures, inevitably finding over 80% of such fractures

this, few motorists chose to wear seatbelts, and few car

were associated with high-velocity road accidents.10,13

manufacturers offered this as a fit-out option at the time of manufacture. The local public appetite for road safety

Compounding this issue was that the surgical management

policy was centred on accident reduction, rather than

of extensive facial fractures was incredibly challenging;

injury prevention, although recognition of the need for car

three-dimensional imaging did not exist, and implantable

restraints was developing overseas.

miniplate fixation had not yet been developed for use in the maxillofacial skeleton. Wire osteosynthesis, combined with

Road traffic accidents rarely led to favourable outcomes.

the use of plaster head caps or Halo-type frames, were the

The magnitude of kinetic energy associated with vehicle

mainstay of fracture reduction and fixation. Levant, another

collisions would typically lead to driver ejection from the

senior surgeon at RMH, recognised the inherent mechanical

car, resulting in either immediate mortality, or severe

and physical limitations of such surgical techniques, and

traumatic injuries. As the head was often the first part

his contemplation on improving management of midfacial

of the body to sustain such high forces, extensive and

fractures culminated in the development of the Levant ANZAOMS PREVIEW VERSION |

103

frame, providing rigid and centrally placed external fixation

has fundamentally changed. Several authors attribute this

using supraorbital pins.14 This itself was a paradigm shift

change to the development and installation of airbags,

in management of midfacial injuries, allowing for stable

which while decreasing the likelihood of multi-trauma or

control of impacted and posteriorly displaced maxillary

panfacial injuries, increase the likelihood of less severe

fractures.15

orbital and eye injuries.25,26,27 Within our cohort, of the nine patients who presented with panfacial fractures, only two

The published work of Hueston, Cook, and Levant would

were associated with road traffic accidents.

go on to become part of the growing public campaign advocating for mandatory seat belt installation and use.

Policy changes to address facial trauma seem to have had

This would draw the attention of the Joint Select Committee

limited effect on the demographic of patients who sustain

on Road Safety in Victoria, commissioned by the Victorian

operative facial injuries. Young adult men continue to be

state government at the time. This was the first

overrepresented in the study group; over 50% of trauma

legislature in the world to bring forward compulsory fitting

cases encountered were in patients between the ages of

and wearing of seat belts, mandated in 1967, achieved in

20 and 40, and 84% of all cases were in patients of male

no small part due to the significant lobbying by the medical

gender. This has largely remained unchanged within the

community. Given the magnitude of the problem, it was no

unit over the last 60 years – Hueston and Cook commented

surprise that the maxillofacial leadership at RMH saw this

that nearly two thirds of trauma cases in their series were

as the defining health issue of the specialty and went to

in the 20-40 age bracket, and Steidler et al. reported a

such great lengths to resolve the issue.

male dominance of 83% in their audit of facial trauma.11,13

Despite an aging population, the proportion of elderly With the development of manufacturing technologies came

patients with operative facial trauma has also remained

new methods to accurately diagnose and treat complex

nearly constant at our unit over this time period.

facial fractures, namely devices for internal fixation. By the mid-1990s, Wiesenfeld and colleagues in their prescient

The most common mechanisms of injury in males were, in

study reported on the improved operative experience

order of frequency, interpersonal violence, sport/leisure,

and outcomes of plate and screw fixation for mandible

and road traffic accidents; in women, the most common

fractures as initially described by Champy et al., and

mechanisms were sport/leisure, falls, and road traffic

effectively obviated the need for wire fixation methods.

accidents. Sport and leisure are much more likely to be

Concurrently, the rise of

responsible for operative facial trauma presentations

high-resolution CT enabled faster, and much more precise

to our unit now compared to sixty years ago, and this

diagnosis of fractures of the maxillofacial region.

is particularly true for women. It may be surmised that

17,18

sociocultural advances promoting female participation in As it became recognised that multi-system traumatic

sports may be at least in part responsible for this change,

injuries and their sequelae constituted a specialist mindset,

though injuries in this category in men were more likely

trauma education was formalised through the Advanced

to be due to contact sports, whereas in women this was

Trauma Life Support (ATLS) course, now mandatory for

usually due to other recreational activities or horse-related

all training surgeons. The protocols outlined by this course

trauma. One limitation of this study is that interpersonal

have increased the survival rates (and thus presentation

violence, particularly within the female gender category,

to hospital) of patients with severe traumatic injuries, and

was not explored in further detail to look for trends in

provided a framework for triaging of injuries and timing of

domestic violence as a cause of facial injury. Assessment

surgical management.19,20,21

and reporting of domestic violence remains difficult as victims may hesitate to identify this is a cause of injury due

RMH was the epicentre of major political and technical

to ongoing fear, shame, or repercussions.

advancements in the last 60 years, achieving marked reductions in the frequency and severity of facial trauma,

Improvements in the management of oral and maxillofacial

as well as paving the way for new paradigms in the

trauma over time are reflected in an overall shorter

management of such injuries through research and

inpatient stay, a change in the time between admission to

development . Such a frame of reference was used in the

surgery, and a differential pattern of complications after

interpretation of data obtained from this research paper.

surgery. High-resolution CT permits the rapid and accurate

diagnosis of facial fractures without the need for urgent Evident in the findings of our large study is that road traffic

evaluation under anaesthesia, such that clinicians can more

injuries have been superseded by interpersonal violence

safely determine which types of injuries require immediate

as the leading cause of trauma presenting to this unit.

attention, and also which can be safely discharged until

This appears to have been the experience at several other

conditions are more favourable for operative intervention

tertiary trauma centres worldwide.23,24 Additionally, the

(for example, following resolution of swelling and

type of injuries sustained through road traffic accidents

ecchymosis). The development and refinement of new

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surgical techniques, including a trend toward internal

by Anthony Lynham of Queensland in their evidence-based

fixation and less reliance on external fixation and IMF,

approach to alcohol-related violence, and resulted in the

has improved patient comfort following surgery, reduced

establishment of reduced liquor trading hours (‘lockout

surgical time, and enabled earlier discharge from hospital

laws’) across many states.28 However, these policies proved

and return to function.

to be unpopular, carried economic costs to small business, failed to achieve bipartisanship, and were subsequently

Consequently, complications outlined in this series

reversed. The authors of this paper recommend revisiting a

compared with previous studies 60 years ago are related

risk-factor based approach to male interpersonal violence,

to open approaches to facial fractures, and the failure

and a discussion of policies that can ultimately reduce

of internal fixation devices. The complexity of such

drug use, alcohol overuse, lack of resources, and provision

procedures increases the range of complications which

of appropriate educational and psychological support

may be encountered; in particular, scarring, nerve injury,

services.

and metalware infection rates are now more common than when closed approaches were utilised. Few cases

Surgeons carry an ethical obligation to take part in the

experienced issues considered significant enough by the

public health conversation, and can provide useful,

patient and clinician to warrant an unplanned reoperation

evidence-based insights to guide public policy. Close

(5.8%) and this was taken as the long-term complication

collaboration between government and health practitioners

rate (at two-year follow-up) for this study.

led to landmark cultural change in road safety and represents a great step forward in reducing the trauma

The reflections of this paper inevitably conclude that

burden in Victoria. This paper provides useful insights on

the policy focus to reduce the incidence of facial trauma

the nature of maxillofacial trauma in Australia through a

should be centred on the main mechanisms of injury

historical lens, with the hope of inspiring new approaches

and their associated major population groups; that is, a

to risk reduction.

reduction in interpersonal violence in young men. Policy approaches to address this problem in Australia were led

CONCLUSION This study is one of the largest of its kind to be published

distinction between management and outcomes of isolated

in the Australasian region and provides a comprehensive

facial fractures versus high-velocity, complex, multi-unit

overview of the experience at a single major trauma centre.

facial fractures, is paramount for the oral and maxillofacial surgeon.

Efforts to reduce the incidence of maxillofacial injuries associated with road traffic accidents are noteworthy

Future developments in surgery and their tangible benefits

and appear to have made significant changes in the

can only be assessed through regular audit for impact and

severity of facial injuries sustained during such events.

outcome analysis. This is particularly important for larger

Young males continue to be overrepresented in the trauma

trauma centres, as the leaders of social and public policy

population, despite various legislative changes to address

change to reduce incidence and improve outcomes.

the major mechanisms of injury associated with this group, specifically interpersonal and alcohol-related violence.

Conflict of interest statement

The operative management of facial trauma at RMH has

The authors have no conflicts of interest to declare.

evolved significantly, mirroring the advances in technology worldwide, as well as shifting away from closed reduction to open reduction and internal fixation. The introduction

Funding

and rapid uptake of 3D virtual surgical planning will no doubt continue to revolutionise the management of

The authors thank the ANZAOMS Education and Research

maxillofacial trauma in the decades to come.

Foundation for providing funding for data interpretation and analysis.

Complex facial fracture patterns from high-velocity injuries continue to present the greatest challenge with respect to restoring form and function. Thus, recognition of the ANZAOMS PREVIEW VERSION |

105

Sixty years on Reflections of a Major Maxillofacial Trauma Centre Referen ce s 1.

Kieser J, Stephenson S, Liston P, Tong D, Langley JD. Serious facial fractures in New Zealand from 1979 to 1998. Int J Oral Maxillofac Surg 2002;31;206-209.

Down K, Boot D, Gorman D. Maxillofacial and associated injuries in severely traumatized patients: implications of a regional survey. Int J Oral Maxillofac Surg 1995;24;409-412.

Qudah MA, Bataineh AB. A retrospective study of selected oral and maxillofacial fractures in a group of Jordanian children. Oral Surg Oral Med Oral Pathol Oral Radiol Endodontol 2002;94;310-314.

Bell RB. The role of oral and maxillofacial surgery in the trauma care center. J Oral Maxillofac Surg 2007;65;2544-2553.

Al Ahmed HE, Jaber MA, Fanas SHA, Karas M. The pattern of maxillofacial fractures in Sharjah, United Arab Emirates: a review of 230 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endodontol 2004;98;166-170.

Singaram M, Sree Vijayabala G, Udhayakumar RK. Prevalence, pattern, etiology, and management of maxillofacial trauma in a developing country: a retrospective study. J Korean Assoc Oral Maxillofac Surgeons 2016;42;174.

Jose A, Nagori SA, Agarwal B, Bhutia O, Roychoudhury A. Management of maxillofacial trauma in emergency: An update of challenges and controversies. J Emerg Trauma Shock 2016;9;73.

Meara DJ, Jones LC. Controversies in maxillofacial trauma. Oral Maxillofac Surg Clinics 2017;29;391-399.

Louis PJ, Morlandt AB. Advancements in maxillofacial trauma: A historical perspective. J Oral Maxillofac Surg 2018;76;2256-2270.

10.

Hueston J, Cook R. The incidence of the major fracture patterns in the face. Med J Aust 1956;2;141-143.

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Steidler N, Cook R, Reade P. Incidence and management of major middle third facial fractures at the Royal Melbourne Hospital: a retrospective study. Int J Oral Surg 1980;9;92-98.

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Clavien PA, Sanabria JR, Strasberg SM. Proposed classification of complications of surgery with examples of utility in cholecystectomy. Surgery 1992;111;518-526.

13.

Hueston J, Cook R, Langford A. Face fractures in motor accidents. Med J Aust 1964;1;940-941.

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Levant B, Gardner-Berry D, Snow R. An improved cranio-maxillary fixation. Br J Plastic Surg 1969;22;288-290.

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Levant B, Cook R, Macfarlane W. Experience with the Levant frame for cranio-maxillary fixation. Br J Plastic Surg 1973;11;30-35.

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Jessop G. Victoria’s unique approach to road safety: a history of government regulation. Aust J Politics History 2009;55;190-200.

17.

Champy M, Lodde J, Schmitt R, et al. Mandibular osteosynthesis by miniature screwed plates via a buccal approach. J Maxillofac Surg 1978;6;1421.

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Renton T, Wiesenfeld D. Mandibular fracture osteosynthesis: a comparison of three techniques. Br J Oral Maxillofac Surg 1996;34;166-173.

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Ali J, Adam R, Butler AK, et al. Trauma outcome improves following the advanced trauma life support program in a developing country. J Trauma 1993;34;890-8; discussion 898.

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Van Olden GD, Meeuwis JD, Bolhuis HW, et al. Clinical impact of advanced trauma life support. American J Emerg Med 2004;22;522-525.

21.

Perry M. Advanced Trauma Life Support (ATLS) and facial trauma: can one size fit all?: Part 1: Dilemmas in the management of the multiply injured patient with coexisting facial injuries. Int J Oral Maxillofac Surg 2008;37;209-214.

22.

Mcdermott F, Hough D. Reduction in road fatalities and injuries after legislation for compulsory wearing of seat belts: experience in Victoria and the rest of Australia. J Br Surg 1979;66;518-521.

23.

Boffano P, Kommers SC, Karagozoglu KH, Forouzanfar T. Aetiology of maxillofacial fractures: a review of published studies during the last 30 years. Br J Oral Maxillofac Surg 2014;52;901-906.

24.

Cabalag MS, Wasiak J, Andrew NE, et al. Epidemiology and management of maxillofacial fractures in an Australian trauma centre. J Plastic Reconst Aesthetic Surg 2014;67;183-189.

25.

Murphy Jr RX, Birmingham KL, Okunski WJ, Wasser T. The influence of airbag and restraining devices on the patterns of facial trauma in motor vehicle collisions. Plastic Reconstructive Surg 2000;105;516-520.

26.

Duma SM, Kress TA, Porta DJ, et al. Airbag-induced eye injuries: a report of 25 cases. J Trauma Acute Care Surg 1996;41;114-119.

27.

Hwang K, Kim JH. Effect of restraining devices on facial fractures in motor vehicle collisions. J Craniofac Surg 2015;26;e525-e527.

28.

Liu T, Ferris J, Higginson A, Lynham A. Systematic review of Australian policing interventions to reduce alcohol-related violence—A maxillofacial perspective. Addictive Behaviors Rep 2016;4;1-12.

| ANZAOMS PREVIEW VERSION

D E N T O A L V E O L A R S U R G E R Y SCIENTIFIC ARTICLE

THE EVIDENCE BASE FOR CONTEMPORARY ORAL AND MAXILLOFACIAL SURGERY RESEARCH IN DENTOALVEOLAR SURGERY IN AUSTRALIA Woo V (BBiomed (Hons), DDS, MBBS)† Chandu A (BDSc, MBBS (Hons), MDsc, PhD, FRACDS (OMS), FRCS (Eng)‡ § Goss A (DDSc, FRACDS (OMS))¶

ABSTRACT Objectives: This study aims to review the evidence-based research contribution of

† I ntern, Royal Adelaide Hospital, Adelaide, South Australia, Australia ‡ S enior Surgeon, Oral & Maxillofacial Surgery, Royal Dental Hospital of Melbourne, Carlton, Victoria, Australia

Australian oral and maxillofacial surgeons in the area of dentoalveolar surgery. Methods: The impact factor, citation index and h-index of the publications

linical Associate Professor, C University of Melbourne, Victoria, Australia

were assessed using a search of both PubMed and Scopus between

meritus Professor, Oral & Maxillofacial Surgery, E University of Adelaide, South Australia, Australia

articles were also assessed.

the years 1993 and 2018. The number of papers and the content of

Results: Corresponding Author:

A total of 241 articles were identified with the majority being in high impact factor journals. The key findings of these articles are presented

ARUN CHANDU

of the 10 highest cited papers, seven related to minimising the risk of

Royal Dental Hospital of Melbourne Carlton, Victoria, Australia Email: chandua@unimelb.edu.au

medication related osteonecrosis (MRONJ) by knowing the frequency and the use of C-terminal crosslinking telopeptide (CTX) tests. The highest cited paper in this series was ‘Nature and frequency of bisphosphonate-associated osteonecrosis of the jaws in Australia’ with 453 citations. The h-index, citation indices and citation in Therapeutic Guidelines Oral and Dental were assessed. There were significant differences between the number of papers and the h index of publications by oral and maxillofacial surgeons (OMS) as compared to oral surgery (OS), with a significantly higher h-index (p<0.0001). Conclusion: The speciality of OMS in Australia has presented an increasing amount of clinically relevant evidence-based research on topics related to dentoalveolar surgery, allowing for care for these patients to be

Keywords:

optimised.

INTRODUCTION

Oral and maxillofacial surgery (OMS) is the principle surgical speciality in Australia involving the maxillofacial and oral region, with its basis in both dentistry and medicine. Australian oral surgery (OS) arose out of dentistry 70 years ago, but some 30 years ago it was appreciated that full medical surgical training was also required to ensure the optimum level of quality care for our patients. This development has been fully described in a recent series of books and articles.1-3 In parallel with this, detailed studies of the workforce and scope of practice have also been published.4-8 Dentoalveolar surgery is a key basis for training in OMS and has also been shown to be a key element in workforce requirements.6-7 However, recently a dental school in Australia has attempted to go back in time and develop a dentally based three-year training speciality of OS and dentoalveolar surgery. 9 Dentoalveolar surgery is the most common procedure performed by oral and maxillofacial surgeons.10 It ‘encompasses those surgical procedures that involve teeth and supporting structures associated with the oral cavity’ and includes procedures such as management of odontogenic infections; surgery of erupted, unerupted, and impacted teeth; third molars; periradicular pathology; implant surgery, traumatic injuries, pathologic conditions, and reconstructive surgery of the dentoalveolar complex.11 Although good surgical technique is paramount in the treatment of patients requiring dentoalveolar surgery, a solid evidence base is also essential. A recent supplement of the Australian Dental Journal (ADJ), highlighted papers from the speciality and became the stimulus for a deeper assessment of the research contribution to the speciality in Australia.12 OMS as a speciality in Australia has produced some seminal research in the field of dentoalveolar surgery and OS, however, the total volume of literature and the relevance of this literature has not been previously assessed. The aim of this study is firstly to determine the number and type of papers by registered Australian oral and maxillofacial surgeons and Australian oral surgeons in the broad area of dentoalveolar surgery, and secondly to measure the evidence base through impact factor, citation and h indices.

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METHODS A search using both PubMed and SCOPUS was undertaken

Seattle, USA) spreadsheet. Sections included wisdom teeth,

for articles published between the years 1993 to 2018

teeth, complications related to implants and teeth removal,

of dentoalveolar research performed by or involving oral

odontogenic infections, odontogenic pathology related to

and maxillofacial surgeons in Australia. Searches were

the dentoalveolar segments, intraoral dental implants and

conducted using unit names and consultants/researchers

pre-prosthetic surgery (excluding zygomatic implants),

in each state and territory, as well as oral and maxillofacial

alveolar augmentation and reconstruction; and

surgeons registered through the Australian and New

anaesthetics (both local and general) medications and

Zealand Association of Oral and Maxillofacial Surgeons.

medical conditions.

Keywords were then linked to identified consultants including, dentoalveolar, dental and maxillofacial, to identify

Initially this was grouped by state and were then combined.

papers that met the inclusion criteria. Directors of Training

Data was descriptive in nature and presented in tabulated

for each state/territory were also consulted in regards to

and graph format according to both state and topic. To

research in progress and higher degrees completed by

assess changes in the number of publications per year,

trainees.

a simple regression was performed using SPSS (Version 22, IBM Corp). Chi-squared analysis was also used in

Only published papers that were concerned with

relation to types of papers published and comparisons of

dentoalveolar surgery were included in this paper. All

paper numbers and h values. The h-index is an index that

consultants/researchers had to be registered specialists in

measures the productivity and impact of published work

OMS and OS. Exclusion criteria included studies relating to

and allows evaluation of an author’s articles, and is defined

non-attached oral soft tissues and the jaws, zygomatic and

as the number of papers with citation number higher or

extraoral implants, maxillofacial trauma, oral cancer, oro-

equal to h.15

facial pain, reconstruction not related to the dentoalveolar complex, facial osteotomies, and temporomandibular

For each subsection of dentoalveolar surgery, the most

disorders. These will be the subjects of future studies.

highly cited studies according to SCOPUS were then

Papers not published by registered specialist oral and

identified, as were key papers in the field. SCOPUS was

maxillofacial surgeons and oral surgeons were excluded.

also used to analyse the h index of the group. The findings

The classification system for articles used by Sandhu et al. and Maran et al. was adopted in assessing the classification of papers.13-14 Observational studies were categorised as either descriptive or analytical. Descriptive studies consisted of case reports or series and review articles. Three types of analytical studies were included: cross-sectional studies (including questionnaires and surveys), prospective studies and retrospective studies. Case-controlled studies and randomised controlled trials were also included. Non-clinical studies included letters, editorials, animal studies, technical notes, and scientific articles.

of those were then further described. Benchmark studies with high citation index of value to all practitioners of dentoalveolar surgery were also identified and assessed. The main publications available to general dentists in Australia are the Australian Dental Journal and Therapeutic Guidelines – Oral and Dental.16 A similar search for publications on dentoalveolar surgery by registered Australian oral surgeons, whose practice is restricted to dentoalveolar surgery, was also performed. The results were analysed and compared to the results obtained for registered oral and maxillofacial surgeons.

The abstracts of all articles were assessed, and those that were unable to be classified from the abstract alone were read in full. Papers were then placed into the following sections/headings and tabulated as an Excel (Microsoft,

ANZAOMS PREVIEW VERSION |109

RESULTS Overall, 241 articles on dentoalveolar surgery published by oral and

State

Number of Articles

Australian Capital Territory

over a 25-year period from 1993 to

New South Wales

2018 were identified. One hundred

Northern Territory

Queensland

training hospitals over five training

South Australia

centres throughout the country, were

Tasmania

Victoria

Western Australia

maxillofacial surgeons in Australia

and seventy-five consultants, associated with 25 accredited

included in the search results. The three states with the most published articles were South Australia (75 articles), Victoria (64) and

Table 1 Origin of papers by state

Queensland (53) (Table 1). There has been a progressive increase in the number of articles published over the past 25 years and this was statistically significant (p<0.001, F = 27.769). The years the most articles were published was in 2017 and 2018, with 20 papers respectively (Figure 1). There were 50 journals in which articles were published. Most articles were published in the ADJ, with 101 articles over the 25-year period, followed by the International Journal Figure 1 Number of articles published by year

of Oral and Maxillofacial Surgery (IJOMS) and Journal of Oral and Maxillofacial Surgery (JOMS). The impact factor for these journals respectively was 1.494, 2.164 and 1.779 (Clavirate Analytics 2019). Seven papers were published in the British Journal of Oral and Maxillofacial Surgery (BJOMS) over a 25-year period, which has an impact factor of 1.061 (Clavirate Analytics 2019). The type of studies are presented in Table 2 and the number of research papers were significantly greater than case reports or letters (p<0.0001).

Type of Paper

Number

Observational study Case series

Review

Comparative study

Analytical study Cross sectional design

Prospective

Retrospective

Case controlled trial (non-randomised)

Randomised controlled trials

Non-clinical Scientific

Animal

Notes

Editorial

Letters

Consensus

2 Table 2 Published articles by type

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Topic

Number of Articles

The h-index for this selection was 5. More papers were published by oral and maxillofacial surgeons (p<0.0001)

Anaesthetics and medications

Complications

Grafting and reconstruction

Implants

Highly citated articles were also identified using SCOPUS,

Infections

with the ten highest-citated papers collated in Table 6.

Miscellaneous

The most-cited paper was by Mavrokokki et al. with 453

Pathology

citations at the time of publication.17 Of the top ten cited

Teeth

papers, seven were related to MRONJ, and two related to

Trauma

Third molars

27 Table 3 Analysis of topics

with a significantly higher h-index (p<0.0001) than oral surgeons.

dental extractions for patients or anticoagulated patients.

State

Articles

h index

Using the classification system used by Sandhu et al., the

New South Wales and Australian Capital Territory

majority of articles were observational studies, namely

Western Australia

reviews (61 articles) and case series, which including case

Queensland

Victoria and Tasmania

South Australia and Northern Territory

reports (57 articles)13. Further classification of the articles included in this study are shown in Table 3. The most popular topic for discussion within these articles was teeth (excluding third molars) with 53 articles with papers mainly

Table 4 h-index by articles published by state

discussing the interaction between dental extractions and medical conditions, followed by pathology with 44 articles, anaesthetics and medications (37 articles) and third molars

Articles

h index

Anaesthetics and medications

Analysis of papers cited in Therapeutic Guidelines: Oral

Complications

and Dental, which is the evidence based principal advisory

Grafting and reconstruction

for Australian dentists, found 24 papers of which 14 were

Implants

general pharmacology and ten were evidenced based

Infections

studies published in the ADJ. The first ADJ Supplement,

Miscellaneous

Pathology

Teeth

Trauma

Third molars

(27 articles).

‘Medications in Dentistry’, which was also cited in the Therapeutic Guidelines, contained a further 10 evidencebased papers. A total of 16 of these 20 papers were authored by oral and maxillofacial surgeons. The data assessment from SCOPUS identified 236 articles

State

Table 5 h-index by articles published by topic

in the included study (out of 241). The h-index of this group of papers was 5 to 12, with the highest individual OMS at 44. The highest-cited paper (453 citations) was on bisphosphonate-related osteonecrosis.17 The highestcited case report was on ‘calcifying epithelial odontogenic (Pindborg) tumour with malignant transformation and metastatic spread’ (44 citations) while the highest-cited letter (105 citations) was one of the first in the world to introduce the connection between bisphosphonate use and osteonecrosis.18-19 Tables 4 and 5 describe the h-index by state and by topic respectively. In contrast, a similar assessment of studies published by registered oral surgeons (as opposed to oral and maxillofacial surgeons) revealed only 16 papers published during the same 25 years with the majority of papers in the field of implantology and anaesthetics/sedation. ANZAOMS PREVIEW VERSION |111

Article

Authors

Citations

Nature and frequency of bisphosphonate-associated osteonecrosis of the jaws in Australia

Mavrokokki, et al. (2007)17

453

Bisphosphonates and avascular necrosis of the jaw: a possible association

Carter, et al. (2005)26

128

Clinical investigation of C-terminal cross-linking telopeptide test in prevention and management of bisphosphonate-associated osteonecrosis of the jaws

Kunchur, et al. (2009)20

109

Bisphosphonates and avascular necrosis of the jaws

Carter and Goss (2003)19

105

The dental implications of bisphosphonates and bone disease

Cheng, et al. (2005)27

Single-stage surgery for rehabilitation of the edentulous mandible: preliminary results

Henry and Rosenberg (1994)28

Tranexamic acid mouthwash - A prospective randomized study of a 2-day regimen vs 5-day regimen to prevent postoperative bleeding in anticoagulated patients requiring dental extractions

Carter and Goss (2003)23

Tranexamic acid mouthwash versus autologous fibrin glue in patients taking warfarin undergoing dental extractions: a randomized prospective clinical study

Carter, et al. (2003)22

Surgical management of bisphosphonate induced osteonecrosis of the jaws

Williamson (2010)29

Bisphosphonates and osteonecrosis of the jaw

Sambrook, et al. (2006)30

Table 6 The ten highest cited articles on dentoalveolar surgery by Australian oral and maxillofacial surgeons from 1993 to 2018

DISCUSSION This study has highlighted the great breadth of topics

patients on antiresorptive agents for osteoporosis requiring

in dentoalveolar surgery, investigated by oral and

extractions (one in 296 to 1130) and malignancy (one

maxillofacial surgeons and trainees in Australia. This is a

in 11 to 15).17 Extensive studies were also conducted in

critical area of research as dentoalveolar is predominately

assessing bone turnover by means of the fasted serum

a high-volume, high-quality area and referral base for oral

beta cross laps test for CTX. This showed that when the

and maxillofacial surgeons in Australia. This is evidenced

CTX test was below 150pg/mL, the risk of MRONJ was one

by workforce studies in 2011 finding that 60% of the

in 42.20-21 Other highly cited papers were advisory reviews

workload of Australian oral and maxillofacial surgeons

on MRONJ for dental and medical practitioners. Prior to

was dentoalveolar surgery.7 This amount of dentoalveolar

2000, the traditional management of patients on warfarin

surgery performed in 2011 was similar to that found in

was to cease the drug prior to dental extractions and then

2000. 5

reintroduce it. This was time consuming and with a risk of embolic cerebrovascular and cardiovascular adverse

Overall, 241 studies were identified over the last 25-year

events. In a series of large-scale clinical trials, the benefits

period with the number of papers published per year

of using local measures without drug alteration were

increasing rapidly, especially since 2010. The predominate

demonstrated.22-23

type of study was clinical or scientific papers, with case reports mainly restricted to pathology. The majority of

There are no comparable studies by other dental

studies were published in high quality, high impact factor

specialities in Australia nor of oral and maxillofacial

journals. The large number of medical management papers

surgeons in other countries, making this study unique in

highlights the importance of the introduction of the dual

the assessment of a craft group’s contribution to the dental

degree with medicine for the management of such patients

literature and the dissemination of practical knowledge

and the crucial role oral and maxillofacial surgeons play in

to the wider dental profession. There are, however,

the management of such patients and in the education of

reviews over a two-year period of articles relating to OS

general dental colleagues.

which have been published in the British Journal of Oral and Maxillofacial Surgery (BJOMS).24-25 These papers,

The findings of the top ten cited papers presented the

however, were specifically related to articles published in

first independent data on the incidence of MRONJ for

that journal, unlike the current study that looked at papers

112| ANZAOMS PREVIEW VERSION

in any journal, published solely by oral and maxillofacial

In the present study, comparison was made with the output

surgeons and oral surgeons from Australia. In their

of another dental speciality publishing in dentoalveolar

analysis, Spencer, et al. found that over a two-year period

research in Australia and this highlighted the significant

from 2008 to 2009 there were 81 papers relating to both

contribution to dentoalveolar surgery research made by

OS and the temporomandibular joint.25 The review paper

oral and maxillofacial surgeons in Australia who published

by Tahim et al. in 2015 focused on OS articles from 2011

significantly more papers with a significantly higher total h

to 2012 and identified 57 articles discussing the topics of

value. This dominance reflects the transcendence of OMS

implantology, dentoalveolar surgery, bone augmentation,

in Australia to encompass the older and limited dental

analgesia and anaesthesia and bisphosphonate-related

speciality of OS. 9 Comparing Australian dentoalveolar

osteonecrosis of the jaw. The breadth of research topics

research output to another country, say the United Kingdom

within the field of dentoalveolar surgery was similar to the

or the United States of America, would not be a fair

present study. Sandhu compared articles published in two

comparison due to differences in workforce numbers and

journals, the BJOMS and the IJOMS, over a 10-year period

varying subspeciality interests in different countries, which

to identify evidence based research in OMS, however topics

may place greater emphasis on other aspects of OMS

or areas of interest were not identified, making it difficult to

such as head and neck surgery. The number of key papers

comprehend the wider scope of published articles in OMS.13

being published by OMS and the importance of evidence-

based practice has led to the recent establishment of the The h-index was chosen as it provided a simple way

Australasian Journal of Oral and Maxillofacial Surgery.

to measure productivity and number of citations. In his original paper describing the h-index, Hirsch considered

The majority of the key or benchmark papers found in this

that a scientist was successful with a h-index of 20 over

present study have not only contributed to the knowledge

20 years and outstanding, ‘likely to be found only at the

of oral and maxillofacial surgeons, but dentistry in general.

top universities or major research laboratories’ with a

These underlie the importance of such studies and the

h-index of 40 over 20 years. The h value of the output of

significant contribution made by OMS to the understanding

Australian oral and maxillofacial surgeons in regards to the

and knowledge in the fields of dentoalveolar surgery.

field of dentoalveolar surgery was 34, which sits between successful and outstanding. Another comparison used by Hirsch was that most Nobel Prize winners in physics had a h-index of 35 to 40.15

Conflict of interest statement This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

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Tahim AS, Goodson AM, Payne KF, Brennan PA. A review of oral surgery-related papers published in the British Journal of Oral and Maxillofacial Surgery during 2011 and 2012. Br J Oral Maxillofac Surg 2015;53;e3-8.

25.

Spencer HR, Morrison A, Braga A, Brennan PA. Review article: oral surgery and TMJ-related papers published in BJOMS in 2008 and 2009. Br J Oral Maxillofac Surg 2010;48;544-548.

26.

Carter G, Goss AN, Doecke C. Bisphosphonates and avascular necrosis of the jaw: a possible association. Med J Aust 2005;182;413-415.

27.

Cheng A, Mavrokokki A, Carter G, Stein B, Fazzalari NL, Wilson DF, et al. The dental implications of bisphosphonates and bone disease. Aust Dent J 2005;50 (Suppl 2);s4-s13.

28.

Henry P, Rosenberg I. Single-stage surgery for rehabilitation of the edentulous mandible: preliminary results. Pract Periodontics Aesthet Dent 1994;6;15-22; quiz 4.

29.

Williamson RA. Surgical management of bisphosphonate induced osteonecrosis of the jaws. Int J Oral Maxillofac Surg 2010;39;251-255.

30.

Sambrook P, Olver I, Goss A. Bisphosphonates and osteonecrosis of the jaw. Aust Family Phys 2006;35;801-803.

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D E N T O A L V E O L A R S U R G E R Y SCIENTIFIC ARTICLE

INCIDENCE RATE OF MEDICATION RELATED OSTEONECROSIS OF THE JAW IN PATIENTS ON DENOSUMAB THERAPY FOR OSTEOPOROSIS UNDERGOING DENTAL EXTRACTIONS Lababidi EA (BHSc (Dent), MDent, MBBS (Hons))† Beteramia D (BSc, DDS)† Garg K (MDS (OMS))† McCullough MJ (BDSc, MDSc (Oral Med), DPhil)† Kumar R (BDS MBBS, FRACDS (OMS))†

† O ral & Maxillofacial Surgery Unit, Royal Dental Hospital of Melbourne, Melbourne, Victoria, Australia

ABSTRACT Objectives: Patients undergoing dental extractions on chronic denosumab therapy are at risk of medication related osteonecrosis of the jaws (MRONJ). The aim of this study was to establish an incidence rate of MRONJ in patients on chronic denosumab therapy for osteoporosis post dental extraction. Methods:

Corresponding Author: EMAD LABABIDI Oral & Maxillofacial Surgery Unit Royal Dental Hospital of Melbourne Carlton, Victoria, Australia Email: emad.lababidi@gmail.com

A records review between January 2016 and December 2018 identified patients on chronic denosumab therapy undergoing extractions. A control group of patients with no history of antiresorptive therapy was age and gender matched to the denosumab group. The primary outcome measure was incidence of MRONJ. Secondary outcome measures were previous anti-resorptive therapy history and timing of denosumab dose to extraction. Results: One hundred and thirty-five patients (370 extractions) on denosumab therapy and a control group of 135 patients (282 extractions) were identified. The mean time between last denosumab dose and extraction was 8.1 months. Five patients (3.7%) in the denosumab group developed Stage 1 MRONJ. No statistically significant association was noted between incidence of MRONJ and timing of last denosumab dose, although the cumulative number of doses approached significance (p=0.07). Conclusion: MRONJ is an uncommon risk in patients undergoing extractions on denosumab therapy for osteoporosis. Further prospective studies are

Keywords:

required to establish whether delaying denosumab therapy peri-

denosumab | anti-resorptive | RANKL inhibitor |

procedurally reduces the incidence of MRONJ.

medication-related osteonecrosis of the jaw | MRONJ | extraction ANZAOMS PREVIEW VERSION |

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INTRODUCTION

Medication-related osteonecrosis of the jaw (MRONJ) is an uncommon adverse drug complication defined by the American Association of Oral and Maxillofacial Surgeons as “Exposed bone or bone that can be probed through an intraoral or extraoral fistula(e) in the maxillofacial region that has persisted for more than eight weeks” in the presence of anti-resorptive or anti-angiogenic medications and absence of history of radiotherapy to the head and neck. MRONJ is a relatively new term, superseding previous definitions such as anti-resorptive related osteonecrosis of the jaw (ARONJ) and bisphosphonate-related osteonecrosis of the jaw (BRONJ) to reflect the wider series of medications associated with osteonecrosis. Denosumab is a relatively new anti-resorptive medication approved for the treatment of osteoporosis and bony metastatic disease since 2010. Denosumab is a monoclonal antibody to the receptor activator of nuclear factor kappa-Β ligand (RANKL). Inhibition of RANKL prevents its binding to the receptor activator of nuclear factor κ B (RANK) onto the surface of osteoclasts and competitive agonism with osteoprotegerin, thus inhibiting the differentiation of osteoclasts and reducing bony turnover.1 Although denosumab produces a similar net effect of reduced bony turnover by inhibition of osteoclast maturation and production, unlike bisphosphonates, denosumab is not incorporated within the bony matrix and its effects are modulated by its serum concentration. The serum half-life of denosumab is approximately 26 days resulting in virtual elimination of its effects within six months, although denosumab may be detectable in the serum for nine months.2,3 Since the introduction of denosumab, numerous case reports and case series have implicated the drug in the development of MRONJ, although this appears to be far more common in patients receiving denosumab for oncological indications.1,4,5 The phase III (FREEDOM) trial of denosumab for osteoporotic therapy reported a spontaneous incidence of MRONJ of 0.04% over a study period of 5 years.6 The aim of this study is to investigate the incidence for MRONJ following dental extractions in patients receiving chronic denosumab therapy for osteoporosis. Secondary outcome measures included whether the timing of denosumab dose prior to extraction affects this incidence.

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METHODS Ethics

Age and gender matched healthy controls with no history of anti-resorptive therapy were identified from a retrospective

This study was approved by the University of Melbourne

records review of the same time period. Exclusion criteria

Health Sciences Human Ethics Sub-Committee. HREC:

from analysis were similar to the denosumab group, with

1748781.1

the exception of follow-up; Where formal follow-up was less than eight weeks, the remainder of the record was

Study design

reviewed to exclude further issues or complications.

A retrospective records review was undertaken in the Oral

Procedure

& Maxillofacial Surgery Unit of the Royal Dental Hospital of Melbourne to identify all patients over the age of 50 who

A departmental protocol was established in early 2016

had undergone an extraction with a history of denosumab

for patients undergoing extractions while on denosumab

therapy between January 2016 to December 2018.

therapy. Where clinically possible, extractions were delayed for at least four months following an injection of

Exclusion and inclusion criteria

denosumab to allow for a reduced serum concentration of

Patients were included for analysis if they had undergone

following a discussion with the prescribing medical

extraction of at least one tooth with a recorded history of

practitioner until mucosal coverage of the extraction site

denosumab injections in the previous 24 months.

was confirmed.

Exclusion criteria included:

the medication, and any further injections were delayed

All extractions were carried out under antibiotic prophylaxis (2g Amoxycillin orally one hour before procedure or 600 mg clindamycin orally if allergic to penicillin). No attempt

1. Patients with previous head and neck radiotherapy;

at primary closure of the sockets was attempted for nonsurgical extractions. However, sockets were sutured if there was deemed to be a high chance of mucosal dehiscence

Denosumab injections for multiple myeloma, bony metastases;

occurring post-operatively. Where extractions of multiple adjacent teeth were required, practitioners were advised to consider staging of extractions to avoid creation of larger

3. Established MRONJ at the time of procedure;

alveolar defects. Post-operative antibiotics were prescribed

documentation (timing between 4. Incomplete denosumab injection and extractions not recorded

All patients were instructed to commence at least one

on a case-by-case basis.

week of chlorhexidine mouth rinses four times a day. Clinical review of all patients occurred at two weeks

5. Follow-up less than 8 weeks post extraction; and

postoperatively to identify early postoperative issues, with a subsequent follow-up eight weeks postoperatively to confirm mucosal coverage of the extraction sites.

Patients who have had subsequent oral

or IV bisphosphonate therapy after 6. bisphosphonate a denosumab injection.

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Variables Data obtained for analysis included demographic data (age,

RESULTS

gender), previous bisphosphonate history and duration, number of teeth extracted, whether the extraction sites

One hundred and seventy-six patients receiving

were sutured or closed surgically (requiring raising of a

denosumab therapy who also underwent dental extractions

mucoperiosteal flap) and whether postoperative antibiotics

were identified in the review period. Following application

were prescribed.

of exclusion criteria, 135 unique patients remained for analysis. Eighteen patients were excluded due to lack of

A detailed medical history was obtained, and patients

timing recorded between denosumab dose and extraction

were grouped into three categories utilising a similar

and seven patients were excluded due to less than eight

approach to Hutcheson et al.7 Group I was medically fit

weeks follow-up. Remaining exclusions were patients

with no significant systemic medical comorbidities beyond

on denosumab therapy for oncological indications, a

osteoporosis/osteopenia. This group included patients

history previous head and neck radiotherapy, established

with issues such as well-controlled hypertension, or

MRONJ at the time of consultation and subsequent oral/IV

well-controlled diabetes. Group II included patients with

bisphosphonate therapy after a denosumab injection.

significant medical comorbidities not typically associated with reduced bony healing. This group included patients

One hundred and thirty-five unique patients on denosumab

with significant cardiac and respiratory comorbidities.

therapy underwent 173 procedures, with a total of 370

Group III included patients with significant medical

teeth extracted. Consistent with the demographics of

comorbidities that may affect bony healing. This group

osteoporosis, the majority of patients were female, with

included patients on immunosuppressive medications

113 female patients and 22 male patients. The control

such as corticosteroids, and insulin dependent or poorly

group was age and gender matched to the denosumab

controlled diabetes.

group, comprised of 135 unique patients undergoing 148 procedures for a total of 282 extractions. A summary of

MRONJ was defined utilising the criteria described by

demographic data and medical co-morbidities is outlined

Ruggerio et al. 8 Where MRONJ was suspected on records

in Table 1. There was no statistically significant difference

review, records were separately assessed by two senior authors (KG and RK) to provide confirmation. If there was any disagreement regarding the diagnosis, a third senior author (MM) was approached to confirm the diagnosis. The subsequent course of MRONJ, further treatment and whether resolution had occurred at the time of analysis were then subsequently recorded. Within the control group, the postoperative course was checked for evidence of any delayed healing or exposed bone at 8 weeks, utilising criteria similar to the classification of MRONJ.

between the two groups in terms of age and gender matching, however a greater proportion of patients in the denosumab group had a greater degree of comorbidities. Forty-three patients in the denosumab group had a previous history of bisphosphonate therapy, of which 22 patients had a duration greater than or equal four years. Ten patients did not have the duration of previous bisphosphonate therapy recorded, and 23 patients who were identified with a previous history of oral bisphosphonates did not have this quantified in years. The mean number of denosumab doses per unique patient was

Statistics Chi-squared was performed for all paired data, with students’ t-test utilised where a variable had less than five subjects. Logistic regression analysis was utilised to test for associations between number of teeth extracted, duration

four (range: 1-14). Twenty-nine patients did not have the number of previous denosumab doses recorded. The mean number of months between the last denosumab dose and procedure was 8.1 months. The majority, 62.4% (108/173) of these procedures had greater than six months between last denosumab dose and extraction. Twenty-six

of previous bisphosphonate therapy and number of months

procedures (from 15 unique patients) were carried out with

between last denosumab dose and dental extraction.

the last denosumab dose greater than one year from the time of extraction.

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Denosumab patients N=135

Control patients N=135

Denosumab procedures

N=173

76.4+/-6.81

75.7

Number of procedures

173

Number of extractions total

370

Age (Mean+/-SD) Sex Male

Previous bisphosphonate history

Female

113

>4 years

<4 years

Medical status Grade I

Nil

Grade II

Yes- Unknown duration

Grade III

Number of doses of denosumab prior to extraction

Smoker

Current

Never

118

121

unknown

173

148

Months between last denosumab dose and extraction Mean

8.07

Range

1 to 24

Simple

142

Surgical

received a prophylactic oral antibiotic dose one hour prior

Primary

to their procedure. No patients in the control group received

Secondary

152

Yes

124

Number of procedures

Table 1 Patient demographics Extraction type

Provision of post-operative antibiotics occurred in 124 procedures in the denosumab group, and four procedures in the control group. All patients within the denosumab group

prophylactic oral antibiotics.

Wound closure

Postoperative Abx

The average number of extractions in the denosumab group was 2.1 teeth per procedure. Seventy-seven procedures in the denosumab group were for a single tooth extraction, and 43 procedures in the denosumab group involved the extraction of three or more teeth. Two procedures in the

Number of teeth removed per procedure (mean)

2.1 (1-12)

denosumab group underwent an adjunctive dento-alveolar procedure at the time of extraction. Thirty procedures involved at least one surgical extraction, and primary closure of extraction sockets was attained in 21 procedures

MRONJ

5 Table 2 Denosumab procedure summary

(Table 2).

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Patients within the control group had a mean number of extractions of 1.9 teeth per procedure. No adjunctive procedures were performed for any of the patients. A lower proportion of patients (28/135) underwent extraction of three or more teeth when compared to the denosumab

Control procedures

N=148

Number of procedures

148

Number of extractions total

282

Extraction type

group. Primary closure of extraction sockets was attained in 23 procedures in the control group (Table 3).

Simple

110

Surgical

Other (bone biopsy, alveoplasty)

A total of five patients in the denosumab group developed MRONJ, all of which were classified as Stage 1. Detailed characteristics of these patients, including the course of

Wound closure

MRONJ have been outlined in Table 3. No statistically

Primary

Secondary

125

(p=0.07). Within the control group no patients were noted

Yes

to have exposed bone eight weeks post extraction.

144

significant correlation between development of MRONJ and any measured variables were identified, although the Postoperative Abx

number of doses of denosumab approached significance

Number of teeth removed per procedure (mean) Osteonecrosis

1.91 0

Table 3 Control procedure summary

MRONJ Case

Age/ Gender

60F

83F

Medical status

Grade II

Cumulative number of doses of denosumab

Bisphopshonate history prior to starting denosumab

Nil

Yes - 6 years

Timing between extraction and last denosumab dose (months)

Procedure

Stage 1 MRONJ 22 site

Yes at 14 weeks, treated with conservative measures

Simple extraction of 17 - closure by secondary intention

Yes

Stage 1 MRONJ 17 site

Yes at 4 months, treated with conservative measures

Simple extraction of 23, 24, 25, 26, 45 - closure via secondary intention

Stage 1 MRONJ 25 site

Yes at 8 months, treated with conservative measures

Yes

Stage 1 MRONJ 23 site

Yes at 14 months following smoothing of exposed bone at 13 months

Yes

Stage 1 MRONJ 23 site

Unknown - failed to attend followup appointments

Surgical extraction of 23, 24 - closure by primary intention

Simple extraction of 23

77F

Grade II

75F

Grade I

Nil

Resolution

Yes - 5 years

Outcome

Simple extraction of 22 - closure by secondary intention

Yes - unclear duration

Postoperative Antibiotics

Table 4 Summary of MRONJ cases

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DISCUSSION The purpose of our study was to evaluate the incidence of

found in the present study is higher than the majority of

MRONJ in patients undergoing dental extractions while on

similar previous studies exploring the incidence of MRONJ

chronic denosumab therapy for osteoporosis, as well as

post extraction for other anti-resorptive medications

assess whether a shorter duration between denosumab

utilised for osteoporosis therapy, although a more recent

administration and timing of extraction was a risk factor for

prospective cohort study by Colella et al., specifically aiming

the development of MRONJ.

to quantify the risk of development of MRONJ post dental extraction in patients on denosumab for osteoporosis

Five of the 135 patients (1 in 27, 3.7%) in the denosumab

calculated an incidence rate of MRONJ of 2.3% in 427

group subsequently developed MRONJ post extraction.

patients (10 patients developing MRONJ), compared to a

Remarkably, previous incidence rates of MRONJ

cohort of 299 control patients not on any antiresorptive

calculated for patients taking denosumab therapy for

therapy undergoing extractions, in which no incidences

osteoporosis was 0.04% (1 in 2,500) from the FREEDOM

of MRONJ were recorded.12 Incidence rates of MRONJ

trial extension. Interestingly, in that previous study, the

following dental extractions on oral bisphosphonate

rate of osteonecrosis in the placebo group was 0.02% (1

therapy have ranged from 0.78%-4.3%.7,11,13,14 Thus,

in 5,000). No further specifiers of whether these cases

to honestly gain informed consent from any patient

of osteonecrosis were precipitated by an event such as

requiring tooth extraction who is taking medications that

an extraction or oral surgery were noted in the study. Of

have been shown to increase risk of MRONJ and used to

note, the duration of denosumab therapy did not appear

treat osteoporosis, the treating clinician should explain

to increase the risk of MRONJ when given at six-monthly

that, according to the best evidence currently available,

intervals, although the follow-up of the study was limited to

osteonecrosis of the jaw may occur at a rate of 1 in 30

five years.

patients. It should also be noted that our results should

be interpreted in the context of our sample size. An A previous study by Matsumoto et al. evaluated a dental

appropriately powered study would require a significantly

extraction protocol in 19 patients receiving denosumab

larger cohort of patients (approximately 804 subjects in

for osteoporosis (5/19) and treatment of malignancy

each group), assuming a hypothesised MRONJ incidence of

(14/19).10 The extractions were performed under antibiotic

1% in the denosumab group. Addressing these limitations

prophylaxis and primary closure of extraction sites were

for future studies in a timely manner will likely be a

attained. Two patients (10.5%) subsequently developed

significant logistical undertaking in the form of a multi-

MRONJ, both who had been receiving denosumab for

centre prospective study to reach such a sample size.

oncological reasons. Within our cohort of patients, primary closure was only attempted where the morphology of

A significant confounding variable in calculating the

the alveolus did not require significant adjustment. A

incidence of denosumab related osteonecrosis at this

further study assessing 2,458 patients undergoing dental

current time occurs when a patient has a previous

extractions on bisphosphonate therapy did find that

history of bisphosphonate therapy, especially as patients

unclosed wound edges was associated with a statistically

transition from oral bisphosphonates to the better tolerated

significant increase in the incidence of MRONJ (OR = 2.51).11

denosumab for osteoporosis therapy. Bisphosphonates

However, in that same study single tooth extractions were

demonstrate high affinity for hydroxyapatite and are

also associated with an increase in incidence of MRONJ

incorporated within bony matrices, resulting in half-lives

(OR = 3.74).11 The majority of extractions performed in

measured in years, and as such are likely to exhibit clinical

the present study were single teeth in otherwise dentate

and adverse effects on bone turnover long after cessation.

patients, approximation of mucosal edges would have

Currently there is still no clear data on whether the risk

required significant height reduction of the alveolus and/or

of MRONJ secondary to oral bisphosphonate therapy

periosteal release incisions to facilitate closure, especially if

is substantially reduced with short-term or longer-term

the adjacent teeth to the extraction site were still present.

cessation of the drug, raising the possibility of a cumulative

Whether the increased surgical trauma from facilitating

or synergistic effect in the development of MRONJ in

primary closure counterbalances a minimally traumatic

patients who had recently started denosumab on a

extraction left to heal by secondary intention remains a

background of previous prolonged bisphosphonate therapy.

topic of contention.

A protocol by Damm and Jones based on trough levels of serum bisphosphonates recommends a two-month drug

The rate of MRONJ in the denosumab patients of 1 in 27

holiday prior to invasive dental procedures.15 Conversely,

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121

a recent large multicentre study found no difference in

initial procedure. The denosumab injection would then

rates of MRONJ between patients who had undergone

only be re-started at least eight weeks following the final

two or three months’ cessation of oral bisphosphonates,

procedure. Such an extended delay of denosumab therapy

versus no cessation prior to dental extraction. Further

is likely to substantially increase the patients’ risk of serious

studies are required to determine whether longer periods

osteoporotic events, namely vertebral fractures or low

of cessation may have a measurable reduction in the post-

impact limb fractures. No incidences of vertebral or low

operative rates of MRONJ. Within our study, the variables

impact fractures were documented on record review of

of previous bisphosphonate therapy and duration of

patients in whom denosumab therapy was delayed in the

previous bisphosphonate therapy in patients currently on

context of dental extractions within our study cohort.

denosumab was not statistically significant in chi-squared analysis or logistic regression analysis respectively for the

In conclusion, MRONJ in patients on chronic denosumab

development of MRONJ, with two of the five patients who

therapy for osteoporosis following dental extractions

subsequently developed MRONJ post dental extraction in

occurred at a rate of 1 in 27 in the present study within

the study being bisphosphonate-naïve.

the limitations described. Antibiotic prophylaxis did not prevent this occurrence and further, there was no

The balance of discontinuing denosumab to reduce risk

relationship between the use of post-operative antibiotics

of MRONJ needs to be carefully weighed against the risk

for these patients and the development of MRONJ. The

of osteoporotic fractures. The rationale behind delaying

implementation of extensive drug holidays to reduce the

extractions from the last denosumab dose is grounded in

risk of MRONJ must be questioned in the setting of the

the pharmacokinetics of denosumab; given the half-life of

known increased rebound fracture risk associated with long

approximately 25 days serum concentration of the drug

periods of cessation of denosumab therapy.

would be lower than 2.5% of the initially administered dose by the time of re-administration at six months.2 While this is a logical presumption, to date no large-scale study directly evaluating the impact of a denosumab drug holiday in preventing the occurrence of MRONJ has been conducted. Within our cohort, no statistically significant correlation between the timing of extraction relative to the last dose of denosumab and incidence of MRONJ was noted.

This research did not receive any specific grant from funding agencies in the public, commercial or not for profit sectors. We do not have any commercial associations that might pose a potential, perceived or real conflict of interest with the subject of this study.

Conversely, the risk of rebound fractures with medium to long term cessation of denosumab is well documented. A recent systematic review has highlighted the rapid increase in bone turnover markers and rapid decrease in bone mineral density in osteoporotic patients discontinuing denosumab, with a number of studies demonstrating reversal of the drug effect on bone mineral density within one year of discontinuation16. Further, a recent case report highlights this risk within a dental context, with a patient developing new acute lower back pain and wedge compression fractures of L1 and L2 during a dental extraction undertaken subsequent to a drug holiday initiated so as to reduce the risk of MRONJ.17 A similar incident was noted by Colella et al, where the authors note a patient had sustained a fractured humerus following a fall who had their denosumab dose in the context of planned dental extractions, although the exact period of delay was not quantified.12 Within our cohort of patients denosumab injections were often delayed past six months in the setting of requiring multiple procedures or staged extractions. While the timing of the initial procedure was as close to six months as possible, depending on the clinical situation, subsequent procedures in the same patient occurred often at least two months after the

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Conflict of interest statement

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Incidence rate of medication related osteonecrosis of the jaw in patients on denosumab therapy for osteoporosis undergoing dental extractions References 1.

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Deeks ED. Denosumab: A review in postmenopausal osteoporosis. Drugs Aging 2018;35;163-173.

Narayanan P. Denosumab: A comprehensive review. South Asian J Cancer 2013;2;272-277.

Aljohani S, Gaudin R, Weiser J, et al. Osteonecrosis of the jaw in patients treated with denosumab: A multicenter case series. J Craniomaxillofac Surg 2018;46;1515-1525.

Dodson TB. The frequency of medication-related osteonecrosis of the jaw and its associated risk factors. Oral Maxillofac Surg Clin North Am 2015;27:509-516.

Papapoulos S, Chapurlat R, Libanati C, et al. Five years of denosumab exposure in women with postmenopausal osteoporosis: results from the first two years of the FREEDOM extension. J Bone Miner Res 2012;27;694-701.

Hutcheson A, Cheng A, Kunchar R, et al. A C-terminal crosslinking telopeptide test-based protocol for patients on oral bisphosphonates requiring extraction: a prospective single-center controlled study. J Oral Maxillofac Surg 2014;72;1456-1462.

Ruggiero SL, Dodson TB, Fantasia J, et al. American Association of Oral and Maxillofacial Surgeons position paper on medication-related osteonecrosis of the jaw--2014 update. J Oral Maxillofac Surg 2014;72;1938-1956.

Socrates P, Roland C, Cesar L, et al. Five years of denosumab exposure in women with postmenopausal osteoporosis: Results from the first two years of the FREEDOM extension. J Bone Min Res 2012;27;694-701.

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Matsumoto A, Sasaki M, Schmelzeisen R, et al. Primary wound closure after tooth extraction for prevention of medication-related osteonecrosis of the jaw in patients under denosumab. Clin Oral Investig 2017;21;127-134.

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Hasegawa T, Kawakita A, Ueda N, et al. A multicenter retrospective study of the risk factors associated with medication-related osteonecrosis of the jaw after tooth extraction in patients receiving oral bisphosphonate therapy: can primary wound closure and a drug holiday really prevent MRONJ? Osteoporos Int 2017;28;2465-2473.

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Colella A, Yu E, Sambrook P, et al. What is the risk of developing osteonecrosis following dental extractions for patients on denosumab for osteoporosis? J Oral Maxillofac Surg 2023;81;232-237.

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Sedghizadeh PP, Stanley K, Caligiuri M, et al. Oral bisphosphonate use and the prevalence of osteonecrosis of the jaw: an institutional inquiry. J Am Dent Assoc 2009;140;61-66.

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Borromeo GL, Brand C, Clement JG, et al. A large case-control study reveals a positive association between bisphosphonate use and delayed dental healing and osteonecrosis of the jaw. J Bone Miner Res 2014;29;1363-1368.

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Damm DD, Jones DM. Bisphosphonate-related osteonecrosis of the jaws: a potential alternative to drug holidays. Gen Dent 2013;61;33-38.

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Tsourdi E, Langdahl B, Cohen-Solal M, et al. Discontinuation of Denosumab therapy for osteoporosis: A systematic review and position statement by ECTS. Bone 2017;105;11-17.

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Leaney A, Sztal-Mazer S. Rebound vertebral fracture in the dental chair during a tooth extraction whilst on a treatment holiday from denosumab to avoid ONJ! Bone 2018;108;43.

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A U T O T R A N S P L A N T A T I O N CASE REPORT

MAXILLARY PREMOLAR AUTOTRANSPLANTATION FOR A CHILD WITH BILATERAL CLEFT LIP AND PALATE Jensen ED (BDS, BScDent (Hons), DClinDent (Paed))†‡ Cheng A (MBBS, BDS, FRACDS (OMS))‡

ABSTRACT Autotransplantation has become a reliable and accepted treatment

† O ral & Maxillofacial Surgery Unit, University of Adelaide, South Australia, Australia ‡ O ral & Maxillofacial Surgery Unit, Royal Adelaide Hospital, South Australia, Australia

option in cases of agenesis. The use of autotransplantation for children with cleft lip and palate is not well documented in the literature. We report a case of autotransplantation of a premolar into the cleft site for a 15-year-old female with bilateral cleft lip and palate. Due to the pneumatisation of the sinus in the planned site, a concurrent sinus lift was necessary to provide adequate space for the root structure. Thorough clinical and radiographic assessment in combination with

Corresponding Author: ANDREW CHENG Oral & Maxillofacial Surgery Unit Royal Adelaide Hospital Adelaide, South Australia, Australia Email: ahacheng@hotmail.com

Keywords: autotransplantation | sinus lift | bilateral cleft lip and palate

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multidisciplinary treatment planning enabled successful surgery. The transplanted tooth maintained a response to vitality testing, had no discolouration and no radiographic signs of root resorption or infection at 12 months’ follow-up. This paper documents a case where sinus lift without bone augmentation was successful with a single-stage autotransplantation of a premolar into a cleft site in bilateral cleft lip and palate.

INTRODUCTION

Autotransplantation is the accepted treatment option for individuals with missing teeth with a relatively high long-term survival at over 80%.1 However, sufficient alveolar bone height is required and the alveolar bone height may be compromised when in close proximity to the maxillary sinus. Orthodontic treatment and tooth replacement options can be limited by the quantity of bone in the area. Dental anomalies have been recognised to occur more frequently in children with cleft lip and palate, potentially due to the cleft itself, the genetic or epigenetic cause of the cleft also affecting the developing dentition or due to the early surgical correction of the cleft defects. Ninety-six per cent of children with bilateral cleft lip and palate had at least one dental anomaly: most commonly agenesis, supernumeraries, microdontia, taurodontism and ectopic eruption.2 Within the cleft defect, discontinuation and commonly a deficiency in the alveolar bone often necessitates a secondary bone graft to fill the bony defect; this facilitates canine eruption and support, closure of oronasal fistulas and improves the condition of the soft tissues in the area. 3 Although secondary alveolar bone grafting has high success, ectopic eruption can still occur around the cleft site which limits the use of orthodontic treatment and implant therapy.4 The use of autotransplantation in children with cleft lip and palate is not well documented in the literature. The aim of this case report was to report the feasibility and effectiveness of autotransplantation as a treatment option for an individual with bilateral cleft lip and palate, while considering the challenges posed by the cleft site’s alveolar bone deficiency.

CASE DESCRIPTION A 15-year-old female presented to the Adelaide Women’s and Children’s Hospital for routine check and treatment planning following consultation with a specialist orthodontist. She had

Age

Primary bilateral lip repair - Uneventful surgery

9 months

Primary bilateral palate repair - Uneventful surgery

9 years

cleft history followed the Cleft

Stage 1 orthodontic treatment - Maxillary Quadhelix expander - Maxillary partial fixed appliances

and Craniofacial South Australia Unit protocol and is detailed in

Alveolar bone graft - Bilateral alveolar graft - Autogenous illiac crest bone harvested

10 years

Table 1. Informed consent was gained for this report from the

Cleft-related treatment

5 months

a background medical history of bilateral cleft lip and palate. Her

Dental and orthodontic treatment

13 years

guardian.

15 years

Stage 2 orthodontic treatment -Maxillary and mandibular fixed orthodontic appliances Autotransplantation - General anaesthetic procedure - Extraction of impacted and diminutive upper left first premolar - Upper right second first premolar used as donor tooth

Table 1 Timeline and list of cleft-related procedures for the individual with bilateral cleft lip and palate.

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Figure 1a - photo placement

Clinical examination revealed agenesis of the upper

(Figure 1b). Measurements from the CBCT confirmed that

right and left lateral incisors and upper left second

the upper left first premolar was diminutive, and both left

premolar (Figure 1a). Radiographic evaluation with

and right first premolars were close to maturation of the

orthopantomogram (OPG) and cone beam computed

apical foramen with almost complete root development.

tomography (CBCT) confirmed the presence of the upper

The alveolar bone in the area between the upper left canine

right and left first premolars which were unerupted

and upper left deciduous molar was minimal, with reduced

clinically.

height as well as pneumatisation of the sinus in the area (Figure 1c). Figure 1a top left ; Figure 1b top right; Figure 1c bottom a) Clinical photograph pre-operatively prior to autotransplant with orientations A, anterior; P, posterior; R, right; L; donor tooth (white arrows) and recipient site (black arrows). Pre-operative cone beam computed tomography of the individual’s facial bones in b) axial, and c) reconstructed panoramic view; with orientations of H, head; F, foot. b) Pneumatisation of maxillary sinus (white dashed arrow) adjacent planned recipient site at different vertical cross sections, c) minimal alveolar bone height in planned recipient site due to pneumatisation of sinus.

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Figure 2

A comprehensive treatment plan was formulated for

Intraoral periapical radiograph of the transplanted tooth in the

autotransplantation of the unerupted right first premolar

recipient site at time of surgery (white dashed arrow).

to the upper left premolar site, with extraction of the diminutive left first premolar.The treating orthodontist prepared the site by adjusting the root torque of the adjacent upper left canine and upper left first permanent molar to allow greater alveolar width for placement of the upper right first premolar pre-surgically. Figure 3 a) Clinical photograph and b) intraoral periapical radiograph of the transplanted tooth (white arrow) in the upper left premolar recipient site at 12 months post-operative. Deposition of dentine within the pulp canal space has occurred (black arrows) with continued growth of the apical area and increase in root length.

The procedure was performed under general anaesthesia

Clinical examination one week postoperatively found Grade

with local anaesthesia administered to the surrounding

I mobility after removal of the wound dressing, good oral

tissues of the donor and recipient sites. A Caldwell-Luc

hygiene and appropriate healing of the surrounding soft

access to the maxillary antrum space was used to elevate

tissues. The silk splint was left in situ and a new surgical

the sinus membrane without puncture. The diminutive tooth

wound dressing placed for one week. Review was monthly

was extracted, and the recipient socket was approximately

for three months, followed by review at six months and

formed. The donor tooth was surgically extracted carefully

12 months postoperatively; no tenderness to percussion

without manipulation of the root surface and placed into

or ankylotic tone were observed, and cold and electronic

the recipient site. The gingival tissues were closed around

pulp tests were positive from three months onward. There

the transplanted tooth and silk sutures were used to splint

was no discolouration of the tooth, and no symptoms

in an infraoccluded position. The tooth was covered with a

reported, or radiographic changes observed (Figures 3a,

surgical dressing

3b). Root canal treatment was not initiated and orthodontic

(COE-PAK; GC Corporation, Japan) for 21 days and an

treatment with light wires continued after six months.

immediate post-operative radiograph was obtained (Figure 2). Simple analgesia, oral amoxicillin and instructions for soft diet and good oral hygiene were prescribed.

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DISCUSSION This young individual with bilateral cleft lip and palate had

membrane will directly revascularise the pulp of the

multiple agenic teeth, a diminutive upper left first premolar

autotransplanted tooth, but keeping the membrane intact

and a lack of alveolar bone support, limiting aesthetic

may assist with stability, maturity and revascularisation as

tooth replacement options and leaving a long-span

it does with grafting material.13,14

edentulous space. Autotransplantation of the contralateral first premolar with concurrent sinus lift was successful,

Root development following autotransplantation can be

without root canal treatment and allowed continuation of

expected to continue for an immature tooth with intact

Phase II orthodontic management. Successful pulpal and

Hertwig’s epithelial root sheath.15 Although there was only

bony healing occurred for the transplanted tooth, even

minor growth remaining for the donor tooth in the present

with minimal alveolar support, and provided an aesthetic

study, continuation of the apical area appeared to have

and functional outcome with success at 12 months

taken place radiographically. Increased dentine deposition

postoperative.

within the pulpal canal also occurred, consistent with pulp canal stenosis following autotransplantation, without

Sufficient alveolar bone augmentation is recommended

commencement of root canal therapy.10 The increased

prior to transplantation.1 However, sinus lift without

dentine deposition and positive periodontal ligament space

bone grafting has been successful in implant placement

surrounding the root surface would be considered a positive

in atrophic maxillae or where alveolar bone support is

sign for long-term stability of this tooth. The functional and

minimal. 5-7 Sinus elevation with blood clotting adjacent

aesthetic outcome of the autotransplanted tooth in this

to the membrane has been found to promote alkaline

case provided a positive outcome for the individual. Children

phosphatase (ALP) and osteoclasts, required to enable the

with cleft lip and palate may have aesthetic concerns with

coupling mechanism of osteoclasts and osteoblasts during

their dentition which may negatively impact their lived

bone remodelling. 8 These findings in implantology research

experience, and efforts should be made to improve facial

suggested that augmented bone or additional bone graft

aesthetics.16

in the area of an autotransplantation may not be required. The success of secondary alveolar bone grafting in bilateral

There are no evidence-based guidelines for

cleft lip and palate is dependent on the timing of the graft

autotransplantation in individuals with deficient alveolar

in relation to the eruption of the canine. 9 This individual

bone such as in the case of bilateral cleft lip and palate;

had a secondary alveolar bone graft before the eruption

hence, further research is needed to better understand

of the canine, at 10 years of age, relatively early due to the

the physiology of autotransplantation in the area of a

agenesis of the lateral incisors bilaterally encouraging early

sinus lift to inform clinical care. The management of this

eruption of the canines. On the upper left side, agenesis of

patient with an immature apex and no perforation of the

the second premolar and ectopic eruption of the diminutive

sinus membrane was successful at one year following the

first premolar left a large-span edentulous ridge with an

procedure, avoiding root canal therapy for the transplanted

arch sequence of central incisor, canine and first permanent

tooth and providing a stable outcome in an individual with

molar. Due to the limited height of alveolar bone in the

multiple tooth agenesis.

edentulous area, treatment options were limited.

Acknowledgements Pulpal healing during autotransplantation may occur in immature teeth and is likely to be through

We acknowledge Dr Richard Salmon, Specialist

revascularisation.

Orthodontist, for the planning expertise and orthodontic

10,11

Although the individual in the

present study had a chronologic age of 15 years at the

management of this patient, including the provision of

time of autotransplantation, and apical maturity would

photos for this report. We sincerely thank the individual and

be likely be expected for a second premolar, the apical

their family for their permission to present their case in this

foramen appeared to be greater than 1mm on pre-

report. Their willingness to share their journey is invaluable

operative radiographic investigation. Without histological

in advancing medical knowledge.

examination, it will remain unclear as to the type of cells in the pulp canal, but the tooth responded positively to

Conflict of interest statement

sensibility testing at three months post-operatively and is likely to have blood capillaries to supply nutrients to cells that fill the pulp space.10,12 It is unlikely that the sinus

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The authors declare no conflicts of interest.

Maxillary premolar autotransplantation for a child with bilateral cleft lip and palate References 1.

Martin K, Nathwani S, Bunyan R. Autotransplantation of teeth: an evidence-based approach. Br Dent J 2018;224:861-864.

Haque S, Alam MK. Common dental anomalies in cleft lip and palate patients. Malays J Med Sci 2015;22:55-60.

Newlands L. Secondary alveolar bone grafting in cleft lip and palate patients. Br J Oral Maxill Surg 2000;38:488-491.

Bergland O, Semb G, Åbyholm F, et al. Secondary bone grafting and orthodontic treatment in patients with bilateral complete clefts of the lip and palate. Annals Plastic Surg 1986;17:460-474.

Pérez-Martínez S, Martorell-Calatayud L, Peñarrocha-Oltra D, et al. Indirect sinus lift without bone graft material: Systematic review and metaanalysis. J Clin Exp Dent 2015;7:e316.

Kumar DS, Jayakumar N, Padmalatha O, et al. Effect of maxillary sinus floor augmentation without bone grafts. J Pharm Bioallied Sci 2013;5:176-183.

Zahedpasha A, Ghassemi A, Bijani A, et al. Comparison of bone formation after sinus membrane lifting without graft or using bone substitute “histologic and radiographic evaluation”. J Oral Maxill Surg 2021;79:1246-1254.

Xu H, Shimizu Y, Ooya K. Histomorphometric study of the stability of newly formed bone after elevation of the floor of the maxillary sinus. Br J Oral Maxill Surg 2005;43:493-499.

Jia Y, Fu M, Ma L. Long-term outcome of secondary alveolar bone grafting in patients with various types of cleft. Br J Oral Maxill Surg 2006;44:308-312.

10.

Andreasen J, Paulsen H, Yu Z, et al. A long-term study of 370 autotransplanted premolars. Part II. Tooth survival and pulp healing subsequent to transplantation. Eur J Orthodont 1990;12:14-24.

11.

Skoglund A, Tronstad L, Wallenius K. A microangiographic study of vascular changes in replanted and autotransplanted teeth of young dogs. Oral Surg Oral Med Oral Pathol 1978;45:17-28.

12.

Langová P, Stembirek J, Matalová E, Buchtova M. Tooth autotransplantations--lessons from animal models: a review. Veterinarní medicína. 2015;60(6).

13.

Li X, Chen S-l, Zhu S-X, Zha G-Q. Guided bone regeneration using collagen membranes for sinus augmentation. Br J Oral Maxill Surg 2012;50:69-73.

14.

Artzi Z, Weinreb M, Carmeli G, et al. Histomorphometric assessment of bone formation in sinus augmentation utilizing a combination of autogenous and hydroxyapatite/biphasic tricalcium phosphate graft materials: at 6 and 9 months in humans. Clin Oral Imp Res 2008;19:686-692.

15.

Andreasen J, Kristerson L, Andreasen F. Damage of the Hertwig’s epithelial root sheath: effect upon root growth after autotransplantation of teeth in monkeys. Dental Traumatol 1988;4:145-151.

16.

Jensen ED, Poirier BF, Oliver KJ, et al. Childhood experiences and perspectives of individuals with orofacial clefts: a qualitative systematic review. The Cleft Palate-Craniofac J 2022:10556656221084542.

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P U B L I C A T I O N S SCIENTIFIC ARTICLE

PUBLICATIONS BY AUSTRALIAN AND NEW ZEALAND AUTHORS IN THREE MAJOR ORAL AND MAXILLOFACIAL SURGERY JOURNALS ABSTRACT

Sklavos A (BDS, MD)† Saha A (MBBS, BDS, MS)‡ Beteramia D (DDS)

Introduction:

Hyam D (MBBS (Hons), BDS (Hons), FRACDS (OMS))§ Breik O (BDSc (Hons), MBBS, MClinSc, FRACDS (OMS))

The level of scientific evidence accepted by the major oral and maxillofacial surgery (OMS) journals has increased through time. This study examined how the Oceanic region, represented by Australia and

† La Trobe University, Melbourne, Victoria, Australia

New Zealand (ANZ) has contributed to the major OMS journals.

‡ R oyal Melbourne Hospital, Parkville, Victoria, Australia

Methods:

ustralian National University, A Australian Capital Territory, Australia

We analysed 21 years of publications by ANZ authors in three major

oyal Brisbane and Women’s Hospital, R Herston, Queensland, Australia

subject of the research, and the affiliated institution or surgical unit of

Corresponding Author:

OMS journals. Data was collected on the level of scientific evidence, the first listed ANZ author. Results: A total of 230 publications were identified. The number of publications

ANTON SKLAVOS

by ANZ authors is increasing, and there was a statistically significant

La Trobe University

increase in publications by ANZ authors in one journal (p=.009).

Melbourne, Victoria, Australia

Most of the research was conducted through a university or public

Email: antonsklavos@hotmail.com

hospital (91%). Private practice accounted for 2.6%. Other specialities publishing in these journals included plastic surgery (3.5%), otolaryngology (1.7%) and the dental specialties (1.7%). Conclusion: ANZ authors associated with an OMS hospital unit or university department have steadily increased their contribution to the three major journals through time. There is a relative lack of research in the private practice setting and the most common research topics by ANZ authors have changed over time.

Keywords: evidence-based surgery | research | levels of evidence | university research | surgery research

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INTRODUCTION

The surgical specialty of Oral and Maxillofacial surgery (OMS) developed from a branch of surgical dentistry.1 From its inception, the specialty has continued to evolve on different fronts. Research in OMS has increased in both the quality of scientific evidence, and quantity of publications across the major journals over time.2-5 As a result there is a corresponding increase in impact factor of each journal which serves to highlight the relative value of the published work in its field.2 The importance of high quality research in OMS has been highlighted as a key area to demonstrate the credentials of the specialty and make a contribution to the particular sub-specialty areas which are shared with other medical and dental specialities.6 This sentiment, and the call for increased involvement in research by OMS training centres and academic institutions has been advocated for decades.7,8 With the increasing standard in the quality of manuscript accepted for publication fewer publications of lower quality scientific evidence, such as case reports, or technical notes are being accepted by the major journals.4,5 This highlights the importance of some level of research training. Exposure to surgical research during the early stages of surgical training has been recognised as a contributing factor to the academic productivity of surgeons in the later stages of their careers. 9 Possessing a formal research degree is also associated with higher research output, when senior surgeons possess a research degree their trainees produce higher volumes of research, and formal training in research attracts greater success in grant applications.10,11 In Australia and New Zealand, training for fellowship in OMS is managed through the Royal Australasian College of Dental Surgeons (RACDS).12 Whilst is has not always been the case, research is now a mandatory component of the training pathway to fellowship. The research requirements may be fulfilled by two pathways. Either by a higher research degree or producing a paper that has been accepted for publication in a peer-reviewed journal. One potential issue which may emerge is that prospective candidates may seek to publish their work in less reputable journals or pay to have their work published. Our hypothesis is that the research output of ANZ authors in three major OMS journals has changed over time. We expected to see an increase in research activity, and expansion from core topics of trauma, orthognathic surgery, and TMJ into other areas such as oncology, reconstruction and craniofacial. The purpose of this study was to provide an overview of the publication trends by Australian and New Zealand authors in three major OMS journals and examine how these trends may have changed over a 21-year period from 1998-2018.

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METHODS Data Source

Results

We analysed 21 years of publications of the three major

The 21-year analysis identified a total of 230 publications

OMS journals: from 1998-2018. The International Journal of

by ANZ authors in the three journals from 1998-2018. This

Oral and Maxillofacial Surgery (IJOMS), the British Journal

included 82 publications in JOMS, 79 in IJOMS and 69 in

of Oral and Maxillofacial Surgery (BJOMS) and the Journal

BJOMS. From 1998-2008 there were 92 publications, and

of Oral and Maxillofacial Surgery (JOMS). Each paper

from 2009-2018 there were 138. A statistically significant

was categorised into one of the following: dentoalveolar

increase in the number of publications was identified in the

(implant, prosthetic and third molar considered as

JOMS (p=.009) (Table 1).

subclassifications), orthognathic, temporomandibular joint, oncology, pathology, radiology, craniofacial implants, craniofacial, trauma, reconstructive, anatomical, pharmacology, aesthetic, sleep related, and technical note. We then examined the authors listed on each publication.

The ANZ authors identified were listed as the first author Journal

Year

No. Publications

1998-2008

2009-2018

1998-2008

2009-2018

1998-2008

2009-2018

BJOMS

Publications were categorised as having the first author

P-value

0.063

as an Australian or New Zealand author, or where there was an Australian or New Zealand author listed on the publication. The nationality of the author was ascertained

JOMS

by the location of the affiliated institution associated with the publication, the affiliated surgical unit was also recorded, noting if the authors were not affiliated with an OMS Unit or department. Where an author was identifiable as an Australian or New Zealand trained OMS, but the

0.009*

IJOMS

0.261

affiliated institution was overseas (during fellowship training or working overseas) these publications were not included in the analysis. Each publication was categorised

Table 1 Total number of publications by ANZ authors over 2 time periods

by a single author (DB) according to the research question. Publications were assigned the corresponding scientific

in 214 publications (93%). ANZ authors affiliated with an

level of evidence outlined by the National Health and

OMS Unit or University department accounted for 209

Medical Research Council (NHMRC) guidelines. Exclusion

publications (91%). Publications with a private practice

criteria included, abstracts, letters to the editor, reflections,

as the affiliated institution accounted for 2.6% of the

and perspective papers. The data collected was available

publications in this study. The remainder were authored

to journal prescribers online and therefore ethical approval

by other surgical and dental specialities including plastic

was not required.

surgery (3.5%), and otolaryngology (1.7%), and other dental specialities (1.7%).

The information was recorded, and descriptive statistics produced. Publications were then considered on two

As a proportion of the total publications for each journal

separate time periods, an early period from 1998-2007

in the given time periods from 1998-2008, ANZ authors

(group 1), and the later period 2008-2018 (group 2). We

accounted for 36 of 1,474 (2.4%) of articles in IJOMS, 28 of

used statistical analysis to account for any statistically

3168 (0.9%) for JOMS and 28 of 1,306 (2.1%) for BJOMS.

significant difference between the two groups. Statistical

During the subsequent period from 2009-2018, ANZ

significance was analysed by an independent t-test for

authors accounted for 43 of 2,271 (1.9%) publications in

each journal with a p-value <0.05 indicating statistically

IJOMS, 54 of 4,061 (1.3%) publications in JOMS and 41 of

significant variation in the mean. Statistical analysis was

2,044 (2%) publications in BJOMS. There was therefore

carried out using jamovi (Version 1.6) software.

a decrease in the proportion of total articles in IJOMS and BJOMS which was not significant, whilst the proportion of ANZ authored papers in JOMS increased.

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Figure 1 Comparison of the categories of publications by ANZ authors over two time periods

BJOMS

JOMS

IJOMS

NHMRC Level

1998-2008

2009-2018

1998-2008

2009-2018

1998-2008

2009-2018

1 (2.4%)

2 (3.7%)

1 (2.3%)

2 (4.9%)

1 (3.6%)

3 (8.3%)

4 (9.3%)

III

11 (39.3%)

13 (31.7%)

8 (28.6%)

29 (53.7%)

7 (19.4%)

14 (32.6%)

4 (14.3%)

5 (12.2%)

1 (3.6%)

6 (11.1%)

6 (16.7%)

7 (16.3%)

8 (28.6%)

13 (31.7%)

9 (32.1%)

6 (11.1%)

10 (27.8%)

12 (27.9%)

V (case report)

5 (17.8%)

7 (17%)

9 (32.1%)

11 (20.4%)

4 (11.1%)

5 (11.6%)

Total

Table 2 Levels of scientific evidence of publications by ANZ authors

The three most frequent subjects from ANZ authors in the

training centre. Outlined in Table 3 are the total numbers

earlier period were TMJ (30 papers), oncology (13) and

of publications based on the geographic location of the

trauma (12). During the subsequent time period the three

authors affiliated institution.

most frequent subjects were trauma (34), pathology (21), oncology (20). Craniofacial research increased from one paper in the first period to six in the subsequent period. TMJ research was the only subject where there was a decrease

Affiliated Training Centre

in the number of publications, from 30 to 17 (Figure 1). The NHMRC level of scientific evidence for each time and journal are displayed in Table 2. The mean difference was analysed using an independent sample t-test. The difference between the means did not reach statistical significance. We considered publications by ANZ authors associated with OMS Units and University departments associated with the regional training centres. Excluded from these numbers were publications by other specialties. There

1998-2008

2009-2018

Victoria

Queensland

South Australia

New South Wales/ACT

Western Australia

New Zealand

125

are six training centres in Australia and New Zealand which includes five training centres in Australia based

Table 3 Publications by training centres in Australia and New Zealand

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DISCUSSION This study highlights the trends in publications by ANZ

research whilst affiliated with an overseas institute.

authors publishing in the major OMS journals. There has

This research has identified an increase in the total number

been an increase in the number of publications over time

of publications by ANZ authors across the three major OMS

across all three journals, this increase was significant for

journals. With a significant increase in articles accepted

the JOMS.

by JOMS. We also identified an increase in the level of scientific evidence for articles accepted in the later period

Maxillofacial trauma has remained the most frequently

considered in the study, and changes in the research

published research topic over time. Topics of craniofacial,

activities of the various training centres.

dentoalveolar surgery, pathology and oncology increased during the most recent period. Research by ANZ authors

Conflict of interest statement

is predominantly undertaken in public OMS Units and University departments, whilst research in private practice

The authors declare no competing interests. No funding

settings is uncommon.

was provided for this study. The data that supports the findings are available upon request. The authors are the

Research in OMS has been a topic which has frequently been addressed in various editorials and publications across the three journals considered here. There have been timely reminders of the importance of research to the specialty of OMS across the three major journals over the study period.6-8, 13-15 Mandating research has been an additional topic of consideration, not necessarily to increase the number of publications, but to encourage critical thinking, and improve the ability to evaluate scientific studies.16 The increasing pipeline to OMS training has also been noted, with some prospective candidates completing research prior to application for training.17 All of these serve to encourage research, and we sought to evaluate the changes in the research output of the ANZ community over a 21-year period in light of the changes which are taking place in the speciality as it evolves. Both IJOMS and JOMS release an issue each month, with BJOMS releasing 10 issues per year. However, it is worth noting that during the study period from 1998-2018 JOMS published 7,229 total articles compared to BJOMS 3350, and IJOMS 3745. However, the total proportion of ANZ authors increased in JOMS compared to the other journals. This may be due to an alignment in the scope of practice and research interest of the journal; however, no conclusions can be drawn from the available data. Whilst the current study provides an overview of the changing research output by ANZ authors over a 21year period, there are some limitations. This study was limited to the three journals and did not include studies in other notable OMS or subspeciality associated journals. Therefore, the sample size remains limited, and may not accurately reflect the research activities of ANZ researchers in OMS. Excluded from the analysis were any publications in which the Australian author conducted 134

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custodians of the data.

Publications by Australian and New Zealand authors in three major oral and maxillofacial surgery journals Referen ce s 1.

Goss AN, Linn R. Extractions to reconstruction: The development of Oral & Maxillofacial Surgery in Australian and New Zealand. Aust Dent J 2018;63 (Suppl 1);S4-S10.

Nabil S, Samman N. Levels of evidence and journal impact factor in oral and maxillofacial surgery: A 15-year follow-up. Int J Oral Maxillofac Surg 2021;50;1394-1399.

Lau SL, Samman N. Levels of evidence and journal impact factor in oral and maxillofacial surgery. Int J Oral Maxillofac Surg. 2007;36;1-5.

Wilson B, Lewis J, O’hare P, Lim C. Following the trend in maxillofacial surgery literature. Br J Oral Maxillofac Surg 2021;59;643-647.

Beteramia D, Sklavos A, Saha A, Hyam D. A 21-year analysis of the publication patterns and level of scientific evidence in three major oral and maxillofacial surgery journals. Int J Oral Maxillofac Surg 2021;50;843-849.

Dimitroulis G. Research - why bother? Int J Oral Maxillofac Surg. 2011;40;1346.

Stoelinga PJ. Research. Int J Oral Maxillofac Surg 1988;17;281.

Laskin DM. Ensuring our future through research. J Oral Maxillofac Surg 1988;46;349.

Ko CY, Whang EE, Longmire WP, Jr., McFadden DW. Improving the Surgeon’s participation in research: is It a problem of training or priority? J Surg Res 2000;91;5-8.

10.

Pausch NC, Neff A, Subbalekha K, et al. Factors affecting scientific productivity of German oral-maxillofacial surgery training centers: a retrospective cohort study. Oral Maxillofac Surg 2015;19(3):259-65.

11.

Han JT, Egbert MA, Dodson TB, Susarla SM. Is formal research training associated with academic success in oral and maxillofacial surgery? J Oral Maxillofac Surg 2018;76;27-33.

12.

Royal Australasian College of Dental Surgeons OMS Training Handbook. https://www.racds.org/documents/Handbooks/OMS_Handbook_%20 2020_v1.0.pdf. 2020

13.

Ramasamy A. Surgical research. Pourquoi pas? (Why Not?). J Oral Maxillofac Surg 2021;79;11-13.

14.

Laskin DM. The need for research in surgical training. J Oral Maxillofac Surg 1990;48;1.

15.

Hupp JR. Making research one of our key priorities. J Oral Maxillofac Surg 2017;75;1573-1574.

16.

Hupp JR. Research during residency; should it be mandated? J Oral Maxillofac Surg 2011;69;2685-2687.

17.

Aghaloo TL. Increasing the pipeline to Oral and Maxillofacial Surgery. J Oral Maxillofac Surg 2022;80;789-790.

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D E N T O A L V E O L A R S U R G E R Y SCIENTIFIC ARTICLE

A SURVEY OF ADELAIDE UNDERGRADUATE DENTAL STUDENTS’ CONFIDENCE IN DENTOALVEOLAR SURGERY Yu E (BDS, BScD (Hons))† Cheng A (MBBS, BDS, GradDipClinDent, FRACDS (OMS))†

ABSTRACT

Sambrook P (MBBS, MDS, FRACDS (OMS), FIBCSOMS)†‡

Objectives:

Goss A (DDSc, FRACDS (OMS))†‡

of dental students in dentoalveolar surgery during their undergraduate

A single centre cohort study to assess the development of confidence training.

† O ral & Maxillofacial Surgery Unit, University of Adelaide, South Australia, Australia ‡ O ral and Maxillofacial Surgery Unit, Royal Adelaide Hospital, South Australia, Australia

Methods: Students enrolled in their third year of study in the Bachelor of Dental Surgery program at the University of Adelaide, Adelaide, South Australia, were longitudinally evaluated in their confidence in extractions during their training in dentoalveolar surgery from November 2019 to December 2020. Surveys were distributed at three

Corresponding Author: ELAINE YU Oral and Maxillofacial Surgery Unit, The University of Adelaide, Adelaide, South Australia, Australia Email: elaine.yu@sa.gov.au

points during this training period and participants were asked to rate their self-confidence in a range of dentoalveolar skills and extractions. Analysis focused on differences in self-rated confidence with degree progression. Comparison was made with students’ assessed grades and also between genders. Results: 95% of eligible students in the cohort responded to the first survey (n=59). All students developed confidence following exposure to undergraduate training (p<0.01). There was no relationship between final student confidence and their assessed grade. Gender differences emerged following preclinical and clinical placement, where males were more confident than females (p<0.05), despite nil correlation with student clinical and theoretical grades. Conclusion: A pyramidal structure of learning starting with theory, followed by preclinical training and clinical placement improves student confidence in dentoalveolar surgery skills.

Keywords: self-confidence | dentoalveolar surgery | undergraduate education | gender

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INTRODUCTION

Extractions are a common requirement in the dental setting.1 The goal of a dental extraction is the intact removal of a tooth, with minimal damage to adjacent structures.2 It is an inherently invasive procedure associated with a multitude of risks and complications. 3 Dental extractions are a source of anxiety for both the patient and practitioner, particularly in the student clinic setting. The anticipation of the procedure can act as a barrier to oral healthcare and exacerbate the cycle of disease. 3,4 Students in the medical and dental field who identify as male exhibited higher levels of confidence compared to female counterparts, although there is no difference in treatment outcomes or quality. 5,6 Across the 10 accredited dental schools in Australia and New Zealand, there have been reports of significant variance in the teaching of dentoalveolar surgery.7 This study showed that Adelaide undergraduates were exposed to the most extensive theoretical, practical and clinical course of any Australasian dental school. This wide discrepancy may be a consequence of brief competency guidelines set by accrediting bodies where only a single sentence is provided. 8 Similarly, on the international scale, 23 European dental schools reported equivalent levels of significant variance in their oral surgery curricula due to brief accreditation guidelines and minimal consensus. 9 This may also be exacerbated by personnel and ethical constraints in the teaching of oral surgery. Compared to other dental specialities, new graduates report low levels of confidence in oral surgery, and it has been identified as an area of stress and insecurity. 9,10 In particular, the wider scope of oral surgery beyond simple extractions, such as surgical extractions and the management of complications and oral pathology, has been identified as an area of low confidence.10,11 It appears that gender may also play a role in the development of confidence in dental students. Male dental students have a tendency for overconfidence and females report decreased levels of self-confidence, which does not correspond with any objective criteria showing poor performance.6 The University of Adelaide’s Bachelor of Dental Surgery (BDS) program is a five-year undergraduate degree. Upon graduation, students are qualified and registered to practise as a general dentist within Australia. The dentoalveolar surgery curriculum is headed by an oral and maxillofacial surgeon and commences at the end of Year Three. The course is pyramidal in structure and runs for 13 months from the end of Year Three until the end of Year Four, where students are assessed in their theoretical understanding and clinical ability. Students are first exposed to a series of online modules over 20 hours in length totalling around 40 lectures which provide detailed theory on general medicine, the principles of surgery and the mechanics of exodontia. Two, three-hour workshops on pig heads are then provided for the purpose of preclinical exposure to flap design, removal of bone, suturing and use of elevators in a simulated environment. As part of this study, an additional 70-minute video series was produced by an oral and maxillofacial surgeon to enhance learning. The series includes a history-taking roleplay, patient/operator positioning, instrument identification and demonstration of exodontia on plastic jaw models, including the removal of fractured roots. These are released prior to commencement of clinical placements, which occur during Years Four and Five. Over these penultimate years, students have a total of three weeks in the Oral and Maxillofacial Surgery Unit at the Adelaide Dental Hospital.

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INTRODUCTION

Year

Length of Block

Aims

Contents

20+ hours online lecture material

Delivery of theoretical content

General medicine, principles of surgery, mechanics of exodontia

70-minute video series

Application of theory

Demonstration of exodontia on plastic models and roleplay scenarios

Two, three-hour workshops

Exposure to surgical techniques and familiarisation with use of instruments on soft and hard tissue

Pig head surgical exercises

• Competence in exodontia under local anaesthetic

• Outpatient clinics

• Experience in surgical extractions

• General theatre sessions

Two, one-week full-day OS block allocations and full year of general dentistry placement

• Exposure to wider OMS

Assessments

Semester 1 and 2 theoretical exam End of year clinical grade

One, one-week full-day OS block allocation and full year of general dentistry placement

Further development of competence and confidence in OS following Year Four

Assessments (Overall – not part of OS curriculum)

Semester 1 and 2 theoretical exam End of year clinical grade

• Local anaesthetic clinics • Elective surgery clinic

Same as in Year Four

Table 1 Outline of the BDS oral surgery curriculum and its aims

Students rotate through clinical sessions in

not resume until the beginning of Semester Two in

outpatient, local anaesthetic extractions,

July 2020. This effectively halved the length of clinical

elective local anaesthetic surgery and

placement from the usual nine months to five. It is also

operating theatre. This is in addition to two full

the observation of senior oral surgery educators that,

years of general dental clinical rotations where

over the last few decades, there has been an increase

students routinely conduct extractions and

in dental student intake, resulting in reduced clinical

basic oral surgery procedures. An outline of

time. Thus, investigation into the impact of preclinical

the BDS oral surgery curriculum and its aims is

and clinical oral surgery experience on student

provided in Table 1.

confidence in extractions is further warranted.

Evaluation of the teaching of oral surgery

The aim of this study was to assess the development

is pertinent, given the emergence of the

of confidence in the skills required to undertake

COVID-19 pandemic during early 2020,

dentoalveolar surgery, in undergraduate dental

and the trend of increasing cohort sizes

students in a single dental school.

across Australian dental schools. Both have resulted in perceived less clinical experience for students in 2020 and 2021.13 On 11 March 2020, the coronavirus COVID-19 was declared a pandemic by the World Health Organization.14 Tertiary education, including the BDS clinical program, was among many affected by this event. In compliance with government restrictions, clinical placements were suspended from 18 March 2020 and did

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Materials and methods

examinations held at two points in the year and practical assessment from oral surgery clinical placements. This was

All research was approved through the University of

obtained following completion, through oral surgery staff.

Adelaide Ethics Committee prior to commencement, under

Single blinding and deidentification occurred through the

approval number H-2019-197.

primary research supervisor, who assigned each unique response with a number from one to 59.

Study design/sample The impact of the COVID-19 pandemic was assessed The present study is a single-centre cohort study of

through obtaining and comparing the clinical outcomes

third-year dental students enrolled in the BDS program at

from student logbooks in 2019, a non-COVID year, with

the University of Adelaide, Adelaide, South Australia, in

2020, the COVID year.

2019. This cohort was selected as they had not previously extracted teeth and were due to commence training in dentoalveolar surgery. Exclusion criteria were students that had completed previous medical degrees, as part of the lateral entry program for Oral and Maxillofacial Surgery, and students who did not progress to Year Four. Lack of progression was due to either taking a leave of absence because of international border closures in the wake of the COVID-19 pandemic, or failure to achieve a passing grade.

Data analyses Statistical analysis was performed using Jupyter Notebook© and analysed through the coding language Python™.17,18 The assumption of normality was checked and confirmed for the data set. Mean differences in confidence between the three surveys were compared with repeated measures ANOVA. Post-hoc pairwise t-tests were performed to confirm any differences. Multiple linear regression models were utilised to compare confidence with final student

Variables

grades. The alpha level was set to 0.05.

The exposure in this study was preclinical training and clinical placement in oral surgery. The outcomes were self-rated levels of confidence and student grades. The covariate was gender.

Data collection methods Three Google Form surveys were distributed through links provided in student emails. The first survey included questions seeking information on participant demographic, including gender identification, and a self-perceived level of confidence in oral surgery skills. For student self-rated confidence, participants were prompted by 22 statements that assessed specific oral surgery skills in three major categories: suturing, mucoperiosteal flap and principles of exodontia. This was based on the learning modules provided to students as part of their oral surgery curriculum. These prompts began with “I feel confident…” and participants were asked to rate on a Likert scale from 0-10, where 0 indicated “not at all confident”, and 10 indicated “totally confident”.15,16 The first survey was distributed prior to preclinical workshops in late 2019. The second was distributed following exposure to preclinical workshops, but prior to clinical placement at the end of 2019 and early 2020. The third and final survey was distributed at the end of clinical placement and the end of fourth year in November 2020. For objective evaluation, the final level of student self-reported confidence was compared with their final oral surgery grade. The final grade consisted of a combination of theoretical

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Results

Characteristics

Sample (n=62) 1

59 (95%)

44 (71%)

42 (68%)

Male

28 (47%)

23 (52%)

23 (55%)

Female

31 (53%)

21 (48%)

19 (45%)

Other

The total number of eligible students was 62 out of a population of 72. Two were excluded as lateral entry students. A further seven were excluded due to lack of

Gender

progression to Year Four. Finally, one more was excluded as the primary investigator in the current project. Fifty-nine students responded to the initial survey (95%), of which 28 (47%) identified as male, 31 identified as female (53%) and none identified as other. For the second survey, 44 students responded (71%), 23 males (52%) and 21 females (48%). For the final survey, 42 students responded (68%), 23 males (55%) and 19 females (45%). The characteristics of the

Table 2 Sample characteristics

For all three dentoalveolar surgical skills, males generally reported higher levels of self-confidence compared to females. This was significant in suturing following clinical placement (p<0.01), and for raising a mucoperiosteal flap

sample are provided in Table 2.

and exodontia following both preclinical training and clinical

For suturing, there was a significant increase in confidence following exposure to both preclinical training and clinical placement (p<0.01). This was confirmed through post-hoc pairwise t-tests (p<0.01) (Table 3, Figure 1).

placement (p<0.05). The linear regression model for comparison of student grades and student confidence generated no significant p-values for theoretical, clinical and final student grades (p>0.05). There was also no significant correlation between

For raising a mucoperiosteal flap, there was a significant difference between the self-rated confidence of students from baseline to post-clinical placement (p<0.01). The posthoc pairwise t-tests indicated that there was significant difference between the confidence levels of baseline

confidence and student grades in males or females (p>0.05). This suggests a lack of any relationship between student confidence and their assessed competency. A summary of the impacts of the COVID-19 pandemic

and preclinical training as well as baseline and clinical

on student clinical outcomes in oral surgery is provided in

placement (p<0.01), however, there was no significant

Table 4. On average, there was a decrease in the number

difference between preclinical training and clinical

extractions and surgical extractions by one third compared

placement (p>0.05) (Table 3, Figure 2).

to the previous non-COVID year.

For the principles of exodontia, there was a significant

Clinical Outcome

2019

2020

Change

Extraction

47(0-105)

33(0-70)

-30

Surgicals

1.3(0-5)

0.8(0-5)

-38%

difference between the confidence of students from baseline to preclinical training and post-clinical placement (p<0.01). This was confirmed through post-hoc pairwise

Table 4 Effect of COVID-19 pandemic on

t-tests (p<0.01) (Table 3, Figure 3).

oral surgery clinical outcomes

Suturing

Mucoperiosteal flap

Exodontia

Survey

Mean

4.6(2.3)

5.9(1.9)

6.8(1.6)

4.5(2.5)

6.0(1.9)

5.9(2.1)

4.4(2.1)

5.1(1.4)

8.3(1.2)

(SD) P

<0.01

5(2.2)

6.2(1.8)

7.4(1.3)

4.8(2.2)

6.3(1.4)

6.7(1.1)

4.7(2.1)

7.7(1.5)

8.8(1.1)

4.2(2.1)

5.5(1.9)

6(1.7)

4.2(2.1)

5.7(1.2)

4.8(1.2)

4.1(2.2)

6.7(1.2)

7.8(1.2)

0.03

<0.01

0.02

<0.01

Pairwise t-test 1

<0.01

0.9

<0.01

Table 3 Mean confidence in oral surgery skills overall

P = Global p-value

1 = Baseline, 2 = Post-preclinical training, 3 = Post-clinical placement

* Significant if p<0.05.

Confidence measured on an 11-point Likert scale

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Figure 1 Confidence in suturing box plot

Figure 2 Confidence in raising a

Figure 3 Confidence in the principles of

(p<0.05 from 1-3, 1-2 and 2-3)

mucoperiosteal flap box plot (p<0.05 from 1-3

exodontia box plot (p<0.05 from 1-3, 1-2 and

and 1-2)

2-3)

DISCUSSION The findings of the present study demonstrate that

to preclinical training and clinical placements. This is

students were more confident following exposure to

consistent with reports in Gilmour et al. (2016) and Haug et

preclinical training and clinical placement in all three

al. (2020) where male dental students possessed greater

surgical skills of suturing, raising a mucoperiosteal flap,

self-confidence in their clinical ability than females. 5,6

and the principles of exodontia. This was despite reduced

Blanch et al. (2008) suggested that the differences in

clinical experience by a third compared to a non-COVID

gender may be due to higher levels of anxiety reported

year.

in female students, despite no clear gender differences in grades or clinical outcomes.15 Females thus have a

The increase in students’ confidence is complemented by

tendency to underestimate their ability whereas males

the observations made by Polychronopoulou and Divaris

tend to overestimate their ability. As oral and maxillofacial

(2010), where confidence levels of students corresponded

surgery tutors and educators, this trend is noted by

with transitions from the didactic baseline to the preclinical

the principal investigators of this project. Often, the

and clinical phases of the curriculum.19 Students were

overconfident but inexperienced student can increase risk

observed to develop further confidence in their oral surgical

of harm to both patient and practitioner. Adjustments to

skills following clinical placement. The only exception was

teaching styles may be required, keeping this in mind.

in raising a mucoperiosteal flap, which is consistent with the reports from Gilmour et al. (2016) where students

Students who did not achieve a passing grade in Year Three

self-scored a high level of confidence in intra-alveolar

and hence did not progress to Year Four were excluded

extractions, but reported the lowest score in raising a flap

from the study, which may be perceived as a source of bias.

comparative to 36 other clinical procedures. 5 Similarly, Yiu

However, this was deemed to have negligible impact as

et al. (2010) and Patel et al. (2006) also noted a lack of

students are not assessed in dentoalveolar surgery until

confidence in complex dentoalveolar surgical procedures

Year Four and only commence preclinical training after

including trans-alveolar exodontia which would, by

the assessment period at the end of Year Three, with the

definition, involve raising a flap.10,11

prerequisite that they have passed Year Three. All students and participants in the current cohort subsequently

A potential influencing factor that could result in

achieved a passing grade for Year Four and progressed to

no significant increase in confidence in raising a

Year Five. Students were also informed that response to the

mucoperiosteal flap following clinical placement may

surveys, including non-response, had nil impact upon their

be that students had limited opportunity to raise a flap

grades, as all responses were deidentified and the principal

during placement compared to the other skills of suturing

investigator was not involved in grading itself.

or principles of exodontia, which are more routine. This is because of the patient and case selection required and the

The cohort study design allowed for longitudinal evaluation

increased complexity of the procedure itself, which was

of students’ confidence as they progressed in their training

exacerbated by the impact of the COVID-19 pandemic on

in dentoalveolar surgery. This meant comparison with a

reduced clinical exposure.

baseline level of confidence could be achieved. Prior studies

Gender differences were evident in the level of self-

also minimises the impact of COVID-19 on reduced clinical

reported confidence in dentoalveolar surgical skills. Males

time on the study cohort, as the focus was on comparative

were significantly more confident following exposure

analysis.

were largely cross-sectional and retrospective. 5,10,11,20 This

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However, it is acknowledged there are limitations in

sample size is required to validate the present findings. One

the current study. The major limitation was the use of

way of achieving this could be through repeating the study

convenience sampling of a single-year student cohort from

method with subsequent year cohorts and with other dental

a single academic institution. Therefore, generalisability

schools. It is in the interest of the Oral and Maxillofacial

is low and is unlikely to represent the wider Australasian

Surgical specialty to ensure that the undergraduate

dental student population. The overall response rate of 78%

students have the optimum exposure and training in

across the three surveys (95% in the initial survey, 71% in

dentoalveolar skills.

the second and 68% in the final) is similar to response rates observed in previous studies that utilised a comparable

Acknowledgements

survey-based method in a dental student cohort.10,19 Although by the third survey 17 participants failed to

Staff at the Oral and Maxillofacial Surgery Unit, Adelaide

respond, the characteristics of the sample population

Dental Hospital, and Adelaide Dental School, University of

remained relatively similar between each survey, only

Adelaide for their cooperation in this project.

differing by ±3%. The exception was gender, where an 8% decrease in female participation was observed, which

Conflict of interest statement

identifies a source of non-response bias – although this may reflect the lower reported levels of self-confidence in

The authors have no conflicts of interest to declare.

female students. Thus, further investigation with a larger

A survey of Adelaide undergraduate dental students’ confidence in dentoalveolar surgery Referen ce s 1.

Oral Health and Dental Care in Australia 2015. Australian Institute of Health and Welfare (AIHW). 2016.

Dym H, Weiss A. Exodontia: Tips and techniques for better outcomes. Dent Clin North Am 2012;56(1):245-266.

Sambrook P, Goss A. Contemporary exodontia. Aust Dent J 2018;63(S1):S11-S8.

Caltabiano ML, Croker F, Page L, et al. Dental anxiety in patients attending a student dental clinic. BMC Oral Health 2018;18(1).

Gilmour ASM, Welply A, Cowpe JG, et al. The undergraduate preparation of dentists: Confidence levels of final year dental students at the School of Dentistry in Cardiff. Br Dent J 2016;221(6):349-354.

Haug SR, Linde BR, Christensen HQ, et al. An investigation into security, self-confidence and gender differences related to undergraduate education in Endodontics. Int Endod J 2021;54(5):802-811.

7. 8.

Goss A. The teaching of oral & maxillofacial surgery to dental students in Australia & New Zealand. Aust Dent J 2018;63(S1):S114-S7. Australian Dental Council. Professional competencies of the newly qualified dentist. Clinical Treatment and Evaluation. Australian Dental Council Ltd. Victoria, Australia. 2016.

Henk SB, Carlijn CJVDC, Sophie MER, Jacques AB. Tooth extraction education at dental schools across Europe. BDJ Open 2015;1(1).

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Yiu CKY, McGrath C, Bridges S, et al. Self-perceived preparedness for dental practice amongst graduates of The University of Hong Kong’s integrated PBL dental curriculum. Eur J Dent Educ 2012;16(1);e96-e105.

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Patel J, Fox K, Grieveson B, Youngson CC. Undergraduate training as preparation for vocational training in England: a survey of vocational dental practitioners’ and their trainers’ views. Br Dent J 2006;Suppl:9-15.

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Arena G, Kruger E, Holley D, et al. Western Australian dental graduates’ perception of preparedness to practice: A five‐year follow‐up. J Dent Ed 2007;71(9):1217-22.

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Thorpe ARDS, Hsu J, Carter EF, et al. Dental student oral surgery training –Comparing the impact of COVID-19 and cohort sizes. Eur J Dent Educ 2023;27(1):63-68.

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World Health Organization. WHO Director-General’s opening remarks at the media briefing on COVID-19 – 11 March 2020. Accessed from https:// www.who.int/director-general/speeches/detail/who-director-general-s-opening-remarks-at-the-media-briefing-on-covid-19---11-march-2020. World Health Organization. 2020.

15.

Blanch DC, Hall JA, Roter DL, Frankel RM. Medical student gender and issues of confidence. Patient Educ Couns 2008;72(3):374-381.

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Gavinski K, Cleveland E, Didwania AK, et al. Relationship between confidence, gender, and career choice in internal medicine. J Gen Intern Med 2021;36(3):662-667.

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Jupyter Notebook. Project Jupyter 2021. Available at: https://jupyter.org

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Python. Python Software Foundation 2021. Available at: https://www.python.org

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Polychronopoulou A, Divaris K. A longitudinal study of Greek dental students’ perceived sources of stress. J Dent Ed 2010;74(5):524-530.

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Honey J, Lynch CD, Burke FM, Gilmour ASM. Ready for practice? A study of confidence levels of final year dental students at Cardiff University and University College Cork. Eur J Dent Ed 2011;15(2):98-103.

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I N F E C T I O N CASE REPORT

MYCOTIC ANEURYSM SECONDARY TO ODONTOGENIC INFECTION CAUSING BILATERAL CAVERNOUS SINUS THROMBOSIS

A rare but important complication Jensen ED (BDS, BScDent (Hons), DClinDent (Paed))†‡ Candy K (MBBS, MDS, FRACDS (OMS))†‡ Candy NG (MBBS, MS)§¶ Nguyen T (MBBS, FRANZCR)†† Cheng A (MBBS, MDS, FRACDS (OMS))†‡ Sambrook P (MBBS, MDS, FRACDS (OMS), FIBCSOMS)†‡

† O ral & Maxillofacial Surgery Unit, University of Adelaide, South Australia, Australia ‡ O ral & Maxillofacial Surgery Unit, Royal Adelaide Hospital, South Australia, Australia §

epartment of Otolaryngology, D Head and Neck Surgery, University of Adelaide, South Australia, Australia epartment of Neurosurgery, D Royal Adelaide Hospital, Adelaide, South Australia, Australia

†† Department of Radiology, Royal Adelaide Hospital,

ABSTRACT Mycotic (infected) aneurysms of the carotid artery are rare and prone to lethal rupture. The aetiology of mycotic aneurysms is evolving with early interceptive management of potentially causative conditions such as infective endocarditis. We report an unusual case of mycotic aneurysm from a severe odontogenic infection in an otherwise healthy 32-year-old female intravenous drug user. Following a diagnosis of an odontogenic infection accompanied by multi-organ dysfunction, immediate surgical intervention encompassed dental clearance, incision, drainage, and tissue debridement. Despite intensive care, minimal improvement was observed. Subsequent investigations found bilateral cavernous sinus thrombosis and skull base osteomyelitis. Neurological deterioration occurred, revealing 100% occlusion of the right internal carotid artery, a 20mm mycotic aneurysm of the left internal carotid artery, which was managed endovascularly. This case underscores the complexity of managing severe odontogenic infections and their potential systemic complications, necessitating a multidisciplinary approach for optimal outcomes.

Adelaide, South Australia, Australia Corresponding Author: ANDREW CHENG Oral & Maxillofacial Surgery Unit Royal Adelaide Hospital Adelaide, South Australia, Australia Email: ahacheng@hotmail.com

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INTRODUCTION

Intracranial mycotic aneurysms represent a rare subset of intracranial vascular pathology, accounting for a minority proportion (0.7-6.5%) of all intracranial aneurysms.1 These anomalous vascular formations are distinctive not only for their infrequency but also for their predilection for younger individuals, with a median age of 35.1 years. Their aetiology is intricately linked to infections both within and beyond the vascular system, stemming from a diverse range of pathogens including bacteria, fungi, mycobacteria, spirochetes, viruses, and toxoplasma.2 Viridans streptococci and Staphylococcus aureus are predominant causative organisms.

The pathogenesis of intracranial mycotic aneurysms is multifaceted, with the most common aetiology stemming from haematogenous spread to the arterial wall with subsequent infection of the tunica media and vessel adventitia. 3 Less commonly, mycotic aneurysms can develop from direct extravascular infection in cases of parameningeal infection.

Diagnosis of these mycotic aneurysms is by advanced imaging modalities, with computed tomography (CT) and magnetic resonance imaging (MRI) serving as the primary diagnostic tools. However, the gold standard for definitive diagnosis remains digital subtraction angiography. 3 Establishing a diagnosis of mycotic aneurysm relies upon identifying an aneurysm in the context of a well-established underlying infection. While infective endocarditis remains the most frequent inciting clinical condition (accounting for 65% of cases), other less common precipitating factors such as cavernous sinus thrombosis and poor dental hygiene have also been reported. 3 Interestingly, the size of the aneurysm does not consistently correlate with the risk of rupture.

The management of intracranial mycotic aneurysms encompasses a triad of approaches: medical, surgical, and/or endovascular therapy. However, due to the rarity of this pathology there have been no randomised controlled trials to inform evidence-based practice. Management is individualised to the patient, with treatment recommendations only informed by case reports and case series.

Medical therapy has a high rate of failure, with contemporary series demonstrating slightly over 50% rate of successful treatment.4 However, the role of antibiotic therapy is significant, given patients that require salvage treatment with surgery or endovascular management have high rates of long-term morbidity and mortality. This case report aims to illustrate the application of flow diversion therapy in the management of an intracranial mycotic aneurysm of odontogenic origin, emphasising the importance of individualised treatment strategies in the absence of evidence-based guidelines for this uncommon pathology.

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CASE REPORT A 32-year-old female presented to a general medical practitioner in August 2011 with a five-day history of rightsided facial swelling. She was otherwise fit and healthy with a history of intravenous drug abuse and a neglected dental state. She was prescribed a three-day course of oral amoxycillin and metronidazole, but was not advised to see a dentist. Her facial swelling worsened and she developed chest pains on expiration. She presented to the Emergency Department of a local hospital. They promptly transferred her to the Royal Adelaide Hospital where she was admitted under the Oral & Maxillofacial Surgery Unit, with consultation from the Infectious Diseases Unit. She was diagnosed with lower right buccal vestibular space odontogenic infection with necrotising fasciitis of the perioral mucosa. In the coming days, further diagnoses were confirmed of multi-organ dysfunction including encephalopathy, acute respiratory distress syndrome, acute renal failure, disseminated intravascular coagulation and metabolic acidosis. Immediate surgical management of the individual included general anaesthesia for a full dental clearance (Figure 1a), intraoral incision and drainage of the lower right buccal vestibular space, and debridement of the necrotic peri-oral tissue. Wound swabs and blood cultures grew Staphylococcus aureus and Candida tropicalis. Management of the bilateral pleural effusion and widespread pulmonary abscesses (Figure 1b) included commencement on intravenous (IV) antibiotics of metronidazole, gentamicin, ceftriaxone and meropenem. Postoperative medical management in the intensive care unit (ICU) had minimal clinical improvement and new lower limb hemiparesis developed. Magnetic resonance image (MRI) of the individual’s brain (Figure 1c) showed bilateral cavernous sinus thrombosis, encephalopathy and subcortical infarcts for which she was treated with warfarin and enoxaparin sodium. SPECT (Single Photon Emission Computed Tomography)/CT facial bone scan revealed osteomyelitis in her skull base with increased radiotracer uptake in the right sphenoid bone, ethmoid bone and right frontal bone, as well as left maxilla. She continued to have partial blindness with light perception only for the right eye.

Figure 1 Radiographs obtained at three different time points, including OPG (a) immediately upon admission with facial swelling, with multiple periapical radiolucencies and grossly carious and brokendown dentition with orientations of R, right; L, left; H, head and F, foot. A computed tomograph of the chest (b) in the axial plane with orientations A, anterior and P, posterior, taken within a day of admission found multiple widespread pulmonary abscesses (black arrows) and repeated MRI of the brain in axial plane (c) at day two post-surgical incision and drainage of facial abscess and debridement of necrotic tissue found bilateral cavernous sinus thrombosis (white arrows).

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Further neurological deterioration occurred at 66 days post-admission with increasing left upper motor neuron weakness and hemiparesis of the left side with impaired speech and swallow. A subsequent brain MRI found 100% occlusion of the right internal carotid artery (ICA) with subcortical infarcts and a 20mm mycotic aneurysm of the left ICA with 95% stenosis (Figure 2a-d). This developed within two months following an initial MRI which demonstrated normal vasculature. Digital subtraction angiography confirmed that perfusion of the Circle of Willis was maintained by the remaining 5% left ICA flow and posterior circulation. Neuropsychiatry assessment found cognitive impairment and a loss of ability to consent, leading to a delay in stenting the mycotic aneurysm. At 140 days postadmission, the left ICA aneurysm was treated with a Pipeline Embolisation Device (Medtronic Neurovascular, Irvine, CA) by interventional radiology after loading with aspirin and clopidogrel. The stenosis was improved from 95% to 50% and an MRI was taken 10 days post-stenting which showed a reduction in size of the left ICA mycotic aneurysm. Clinical stability with independent mobility and ability to perform simple activities of daily living allowed discharge after a 210-day admission to a rehabilitation centre and subsequently low-level nursing care facility. She remained a nursing care resident until she died aged 43. No autopsy was carried out and thus the cause of death

Figure 2 Left mycotic aneurysm (white arrows) of the intracavernous internal carotid artery visualised by a) a T2 magnetic resonance image in the axial plane with orientations R, right; L, left; A, anterior and P, posterior, b) digital subtraction angiography. Time of flight magnetic resonance angiogram maximum intensity projection at c) initial presentation with mild luminal narrowing of the left cavernous internal carotid artery (dashed arrow) and d) subsequent image two months post-presentation with marked narrowing of the cavernous internal carotid artery with the left mycotic aneurysm visualised.

remains unknown.

DISCUSSION This case reports the intricate clinical course and

Advancements in airway intervention, surgical techniques,

management of a mycotic aneurysm in the context of a

and medical care have reshaped the landscape of

severe odontogenic infection, highlighting the importance

managing severe odontogenic infections. This infection

of timely diagnosis, multidisciplinary intervention, and the

progressed from dental structures to facial soft tissues,

potential far-reaching consequences of dental infections.

eventually infiltrating the base of the skull and the

Odontogenic infections can extend beyond local tissue

cavernous sinus. The subsequent cavernous sinus

destruction, exerting profound systemic and regional

thrombosis from contiguous spread of infection is

effects.

likely the cause of the left-sided internal carotid artery mycotic aneurysm. The interplay between aggressive

The Oral & Maxillofacial Surgery Unit of the Royal Adelaide

microorganisms and the anatomy of the facial venous

Hospital has maintained a database of admissions for

system facilitated the infection’s migration, leading to

severe odontogenic infections.4-6 Most patients fully

cavernous sinus involvement and subsequent neurological

recover, but less than 1% die, mainly from spreading

sequelae. These sequalae were most likely cause by

sepsis and organ failure. Another small group may develop

ischaemia created by the bilateral carotid stenosis as well

craniofacial necrotising fasciitis. The reported case is an

as turbulent flow induced by the left-sided cavernous

unusual outlier, given that they survived but with a poor

segment aneurysm. The emergence of ophthalmic and

functional outcome. This outcome was despite intensive

neurological symptoms prompted thorough investigations

multidisciplinary treatment over many weeks with an

to ensure timely control of infection spread. The integration

estimated cost of over $350k in 2011 dollars.

of multidisciplinary care, encompassing medical, surgical, and interventional approaches, is imperative for curbing the devastating potential of odontogenic infections.

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This case highlights the value of serial imaging in patients

This case serves as a reminder of the systemic and regional

with rapidly progressive skull base osteomyelitis. An

effects of severe odontogenic infections, complicated by

MRI completed during initial workup demonstrated

the development of a mycotic aneurysm from adjacent

right cavernous sinus thrombosis with normal left-sided

infection. In rare cases of fulminant odontogenic infection

cavernous sinus and normal arterial structures. Within six

associated with skull base osteomyelitis and/or cavernous

weeks the individual had developed bilateral cavernous

sinus thrombosis, serial imaging with CTA or MRI might

sinus occlusion with new right-sided cavernous segment

be reasonable to monitor for the development of mycotic

internal carotid artery occlusion and a new left-sided

aneurysms.

cavernous segment internal carotid aneurysm with associated stenosis proximally.

Finally, prevention is better than treatment. A healthy mouth does not develop severe odontogenic infection.

Alawieh et al. (2023) published management and long-term outcomes for 24 patients with mycotic aneurysms.7 They

Acknowledgements

monitor individuals with serial vascular imaging every 2-4

We acknowledge with gratitude the valuable contribution of

weeks, then 3-6 months, and then annually. The presented

the individual and their medical records, which will serve to

case was different given the index imaging demonstrated

advance medical knowledge.

no evidence of an aneurysm. Given the rapidly progressive nature of the individual’s infection, it is possible that earlier surveillance imaging may have identified the developing

Conflict of interest statement The authors declare no conflicts of interest.

aneurysm at an earlier stage and facilitated a better outcome.

Mycotic aneurysm secondary to odontogenic infection causing bilateral cavernous sinus thrombosis: A rare but important complication References 1.

Kannoth S, Thomas SV. Intracranial microbial aneurysm (infectious aneurysm): current options for diagnosis and management. Neurocritical Care 2009;11;120-129.

2. 3.

Deipolyi AR, Rho J, Khademhosseini A, Oklu R. Diagnosis and management of mycotic aneurysms. Clin Imaging 2016;40(2);256-262. Ducruet AF, Hickman ZL, Zacharia BE, Narula R, Grobelny BT, Gorski J, et al. Intracranial infectious aneurysms: a comprehensive review. Neurosurg Rev 2010;33;37-46.

Bayetto K, Cheng A, Sambrook P. Necrotizing fasciitis as a complication of odontogenic infection: a review of management and case series. Aust Dent J 2017;62(3);317-322.

5. 6.

Bayetto K, Cheng A, Goss A. Dental abscess: A potential cause of death and morbidity. Aust J Gen Pract 2020;49(9);563-567. Han J, Liau I, Bayetto K, May B, Goss A, Sambrook P, Cheng A. The financial burden of acute odontogenic infections: the South Australian experience. Aust Dent J 2020;65(1);39-45.

Alawieh AM, Dimisko L, Newman S, Grossberg JA, Cawley CM, Pradilla G, et al. Management and long-term outcomes of patients with infectious intracranial aneurysms. Neurosurg 2023;92(3);515-523.

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C O V I D - 1 9 SCIENTIFIC ARTICLE

TRENDS IN ORAL AND MAXILLOFACIAL SURGERY DURING THE COVID-19 PANDEMIC

The Australian experience Mian M (MD, BDS, MPH)† Subhashaan Sreedharan S (MD, MPH)† Sklavos A (BDS, MD)† Austin S (MBBS, GdipDent, MPhil, FRACDS (OMS))‡ † The Royal Melbourne Hospital, Melbourne, Victoria, Australia ‡ Department of Oral & Maxillofacial Surgery, Western Health, Melbourne, Victoria, Australia Corresponding author: ANTON SKLAVOS The Royal Melbourne Hospital, Parkville, Victoria, Australia

ABSTRACT Objectives: The aim of the study was to investigate the impact of COVID-19 on Medicare rebatable specialist OMS services (consultations and treatment) in Australia during the period January 2020 to December 2020. Methods: A smoothing model was applied to public datasets between 2016 to 2019, in order to predict the number of quarterly consultations and treatments between January 2020 and July 2021 with 95% confidence (p<0.05). Predicted and observed consultations and treatments were compared.

Email: antonsklavos@hotmail.com

Results: There was a significant decrease in the number of consultations in the second quarter of 2020 (30.3%, 95% CI: 23.0% to 36.3%). This was followed by subsequent increases in third (13.7%, 95% CI: 2.0% to 28.4%) and fourth quarters of 2020 (16.6%, 95% CI: 0.8% to 38.4%). Telehealth consultations made up only 5.1% of total consultations during the pandemic. There was a significant decrease in the number of OMS treatments in the second quarter of 2020 for both dualqualified (33.8%, 95% CI: 27.9% to 38.9%) and single qualified surgeons (42.2%, 95% CI: 35.4% to 47.7%). Total treatments for dual-qualified rebounded in the third (7.9%, 95% CI: 0.2% to 16.9%) and fourth quarters of 2020 (9.5%, 95% CI: 1.7 to 18.6%). This was associated with a change in treatment types, including sustained reductions in orthognathic surgery and an increase in preprosthetic and implant surgery. Conclusion: These findings confirm that the most significant impact of COVID-19 Keywords:

on OMS services in Australia occurred early in the pandemic. Total

coronavirus | healthcare delivery |

services recovered during the second half of 2020 to reach pre-

healthcare systems | maxillofacial surgery |

pandemic levels. The relative proportion of treatments subtypes is

SARS-CoV-2

changing.

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INTRODUCTION

Coronavirus disease 2019 (COVID-19) continues to have a profound impact on healthcare systems and economies worldwide. In the international context, Australia has fared relatively well, with 115,802 cases of COVID-19 resulting in 1,357 deaths as of October 6th, 2021.1 However, aggressive suppression strategies and lockdowns across the country have had significant impacts on healthcare delivery, including surgical services. Throughout the pandemic, oral and maxillofacial surgery (OMS) has been particularly affected, in part due to the perceived risk of COVID-19 transmission associated with instrumentation of the upper aerodigestive tract.2 As part of the response to the COVID-19 pandemic, national and state governments implemented a range of measures aimed to reduce the risk of transmission in healthcare settings and to ensure adequate hospital capacity and personal protective equipment (PPE). Restrictions were placed on non-urgent elective surgery and dental services, first beginning nationally in March 2020, and implemented at state and local government levels during subsequent waves. Surgical consultations in public outpatient and private practice settings were also limited. In this context, the Australian Government also funded temporary COVID-19 telehealth services. 3 In Australia, OMS services are provided predominately through the private healthcare system.4 Government rebates for specified services are available through the national universal healthcare scheme (Medicare). This study used an interrupted time series model to analyse publicly available Medicare Benefits Schedule (MBS) data for OMS consultations and treatments. 5 The aim of this study was to investigate the impact of COVID-19 on rebatable specialist OMS services (consultations and treatments) in Australia during the period January 2020 to July 2021.

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METHODS Data Source Statistical Analysis Category 3 and 4 of the Australian Medicare Benefits Schedule (MBS) outline the range of coded surgical services

Triple exponential smoothing (TES) is a commonly used

utilised by OMS surgeons that attract a Medicare rebate.

forecasting method used to model and predict interrupted

Category 3 services are used by most OMS clinicians

time series data. TES uses weighted averages of past

(dentally and medically qualified) and are shared with

observations, with weights decreasing exponentially as

other medical specialists. Category 4 applies exclusively

observations get older. TES modelling decomposes the

to clinicians who obtained their FRACDS (OMS) or

data series into a level component, a trend component and

equivalent qualifications prior to 1 November 2004

a seasonal component, thus accounting for long term trend

(dentally qualified). This study utilised data from both

and seasonal or holiday related changes.7

the OMS section of Category 3 of the MBS as well as all Category 4 data. Category 3 services are divided into

In the present study, additive TES was used to model the

orthognathic surgery, TMJ surgery, oral and maxillofacial

quarterly number of (1) consultations, (2) total treatments

pathology surgery, preprosthetic and dental implants

and (3) each OMS treatment subtype between January

and treatment of facial fractures. Category 4 services are

2016 and December 2019. Models were used to predict

divided into face-to-face consultations, general surgery

consultations and treatments between January 2020 and

(including oral and maxillofacial infections and pathology

July 2021. Observed and predicted number of quarterly

surgery), plastic and reconstructive surgery (including

consultations and treatments between January 2020 to

orthognathic surgery), preprosthetic surgery and dental

July 2021 were compared to calculate residual differences.

implants, neurosurgical surgery, ear-nose-throat surgery,

A 95% confidence interval for predictions was also

temporomandibular joint surgery and treatment of facial

calculated in order to determine statistically significant

fractures. Quarterly MBS data for rebatable OMS services

residual differences (p<0.05). All statistical analyses were

provided between January 2016 to December 2019 was

performed using SPSS version 22 (IBM SPSS, Amronk,

extracted from MBS datasets and grouped into face-to-

NY, USA) and LKS-CHART Forecasting Tool (St. Michael’s

face consultations or treatments. In addition, the number

Hospital, Toronto, ON, Canada).

of telehealth consultations, using the temporary COVID-19 telehealth item numbers for OMS (Category 4), was extracted and included in consultation data. This analysis was Human Research Ethics exempt as only publicly available, de-identified, composite statistical data was considered.

RESULTS Consultations There was no significant difference between the number

There was no significant difference between the number of

of observed and predicted consultations in the first quarter

observed and predicted total quarterly consultations during

of 2020. There was a significant decrease in the number

2021 (Figure 1, Table 1). There were 3,519 telehealth

of consultations in the second quarter of 2020 (4247,

consultations (Category 4) between January 2020 and

30.3%, 95% CI: 23.0% to 36.3%). This was followed by

July 2021 making up 5.1% of total consultations during the

a significant increase in consultations in the third (1893,

period implemented.

13.7%, 95% CI: 2.0% to 28.4%) and fourth quarter of 2020 (2082, 16.6%, 95% CI: 0.8% to 38.4%).

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Figure 1 Total oral and maxillofacial surgery consultations (Category 4) performed in Australia during COVID-19. Panel A shows observed consultations between January 2017 and June 2020, modelled consultations between January 2017 and December 2019 and predicted (with 95% CI) consultations between January 2020 and June 2020. Panel B shows observed and predicted (with 95% CI) consultations between January 2020 and June 2020.

Jan-Mar 2020

Apr-Jun 2020

Jul-Sep 2020

Oct-Dec 2020

Jan-Mar 2021

Apr-Jun 2021

-1.6 (-9.6, 7.9)

-33.8* (-38.9, -27.9)

7.9* (0.2, 16.9)

9.5* (1.7, 18.6)

0.4 (-6.6, 8.5)

2.4 (-4.5, 10.4)

Orthognathic

-15.6* (-24.0, -5.2,)

-46.8* (-53.8, -37.4)

-10.5 (-25.0, 11.1)

-27.0* (-41.2, -3.9)

-33.3* (-48.3, -5.7)

-41.7* (-57.4, -7.8)

TMJ

5.9 (-21.0, 60.3)

-37.8* (-51.8, -12.6)

30.6* (2.8, 79.3)

12.6 (-9.5, 49.0)

13.4 (-11.2, 57.1)

-11.4 (-28.7, 17.0)

Pathology

-6.3 (-12.7, 1.1)

-32.7* (-37.0, -27.8)

-3.4 (-2.9, 10.5)

5.6 (-0.9, 13.1)

-4.6 (-10.4, 2.0)

-7.2* (-12.5, -1.1)

Preprosthetic and Implant

0.0 (-8.4, 10.0)

-37.0* (-41.9, -31.2)

7.3 (-0.5, 16.5)

8.4* (1.1, 16.9)

9.9* (1.8, 19.3)

16.8* (8.7, 26.1)

Fractures

-20.3 (-44.9, 44.1)

-35.7 (-53.5, 4.0)

-24.4 (-47.3, 27.4)

-20.1 (-46.6, 58.5)

6.1 (-29.6, 115.5)

-5.5 (-34.0, 66.4)

Total Treatments Treatment Subtypes

* indicates statistical significance Table 1 Percentage differences between observed and predicted oral and maxillofacial treatments (Category 3)

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Figure 2 Oral and maxillofacial surgery treatments (Category 3) performed in Australia during COVID-19. Panel A shows observed treatments between January 2017 and June 2020, modelled treatments between January 2017 and December 2019 and predicted (with 95% CI) treatments between January 2020 and June 2020. Panel B shows observed and predicted (with 95% CI) treatments between January 2020 and June 2020.

Treatments There was no significant difference between the number of

There were significant changes to the relative proportions

observed and predicted total treatments in the first quarter of

of Category 3 treatment subtypes in 2021. There was a

2020 for either Category 3 or 4 items. There was a significant

sustained reduction in orthognathic surgery during the first

decrease in the number of OMS treatments in the second

(734, 33.3%, 95% CI: 5.7% to 48.3%) and second quarters of

quarter of 2020 for both Category 3 (1761, 33.8%, 95% CI:

2021 (931, 41.7%, 95% CI: 7.8% to 57.4%). There was also a

27.9% to 38.9%) and Category 4 (1971, 42.2%, 95% CI:

reduction in treatment for head and neck pathology in 2021,

35.4% to 47.7%). This was followed by significant increases

which reached statistical significance in the second quarter

in total treatments for Category 3 in the third (440, 7.9%, 95%

of 2021 (335, 7.2%, 95% CI: 1.1% to 12.5%). In contrast,

CI: 0.2% to 16.9%) and fourth quarters of 2020 (532, 9.5%,

Category 3 preprosthetic and implant surgery increased in the

95% CI: 1.7 to 18.6%), (Figure 2). In contrast, there was no

first (213, 9.9%, 95% CI: 1.8% to 19.3%) and second quarters

significant difference in observed and predicted treatment

of 2021 (389, 16.8%, 95% CI: 8.7% to 26.1%).

levels in the third and fourth quarters of 2020 for Category 4 treatments (Figure 3). Decreases were observed across most OMS treatment subtypes during the second quarter of 2020 (Table 1, Table 2).

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Figure 3 Oral and maxillofacial surgery treatments (Category 4) performed in Australia during COVID-19. Panel A shows observed treatments between January 2017 and June 2020, modelled treatments between January 2017 and December 2019 and predicted (with 95% CI) treatments between January 2020 and June 2020. Panel B shows observed and predicted (with 95% CI) treatments between January 2020 and June 2020.

Jan-Mar 2020

Apr-Jun 2020

Jul-Sep 2020

Oct-Dec 2020

Jan-Mar 2021

Apr-Jun 2021

Total Consultations

-7.5 (-14.6, 0.8)

-30.3* (-36.3, -23.0)

13.7* (2.0, 28.4)

16.6* (0.8, 38.4)

12.8 (-5.4, 39.6)

14.7 (-7.8, 51.8)

Total Treatments

-5.6 (-14.8, 5.8)

-42.2* (-47.7, -35.4)

8.7 (-1.4, 21.2)

-0.1 (-9.2, 11.0)

8.2 (-2.2, 20.9)

1.3 (-8.2, 13.0)

-8.5 (-20.2, 7.2)

-33.6* (-41.7, -22.8)

1.4 (-10.7, 17.3)

-0.3 (-12.9, 16.6)

8.4 (-5.7, 27.6)

2.3 (-10.5, 19.3)

- Infection†

-2.1 (-20.2, 26.5))

-25.6 (-42.5, 5.3)

6.6 (-18.9, 55.7)

-9.8 (-34.0, 42.7)

-1.3 (-29.5, 64.4)

0.1 (-31.6, 86.5))

- Pathology†

-10.5 (-22.5, 6.0)

-43.9* (-50.9, -34.6)

-12.8 (-23.5, 1.4))

-11.3 (-22.9, 4.2))

-3.1 (-16.3, 15.1)

-12.9 (-23.9, 1.8)

-5.0 (-14.6, 6.9)

-34.3* (-40.8, -26.2))

24.9* (13.0, 39.4)

4.4 (-5.3, 16.1)

12.5* (1.8, 25.8)

8.0 (-2.1, 20.5)

-0.7 (-20.4, 31.9))

-53.8* (-64.8, -32.9)

-21.8 (-38.8, 8.5)

3.1 (-17.5, 37.5)

-0.1 (-20.3, 33.8))

-4.4 (-27.7, 41.0))

Preprosthetic and Implant

2.1 (-15.3, 28.6)

-40.5* (-51.6, -22.6)

26.9 (-2.0, 80.0)

23.7 (-9.0, 93.0)

50.5 (-2.0, 224.0)

55.5 (-6.3, 357.3)

Neurosurgical

-4.3 (-41.0, 152.1)

-45.7 (-65.8, 30.9)

4.6 (-39.0, 266.8)

6.8 (>-46.5)

47.4 (>-34.7)

17.7 (>44.5)

Ear, Nose and Throat

14.9 (-4.3, 43.8)

-37.6* (-47.3, -23.6)

13.3 (-4.2, 38.6)

1.1 (-14.2, 23.2)

26.4* (4.8, 59.3)

-2.9 (-18.3, 19.6)

Temporomandibular Joint

5.1 (-24.3, 71.9)

-35.6 (-53.2, 3.5)

-14.7 (-36.5, 29.8)

15.9 (-15.7, 85.6)

12.5 (-20.2, 90.7)

12.2 (-19.8, 86.4)

Fractures

-4.3 (-46.1, 328.5)

-42.7 (-63.5, 32.8)

-45.4 (-62.5, 0.6)

64.0 >-22.9

10.1 >-47.4

37.3 (-20.2, 391.9)

Treatment Subtypes

General

Plastic and Reconstructive - Orthognathic‡

* indicates statistical significance † treatments related to infections and pathology are a subgroup of general treatments ‡ treatments related to orthognathic surgery are a subgroup of plastic and reconstructive treatments

Table 2 Percentage differences between observed and predicted oral and maxillofacial consultations and treatments (Category 4)

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DISCUSSION This study aimed to investigate changes in government

orthognathic surgery. In summary, the most significant

subsidised OMS consultations and surgical treatment

impact of COVID-19 on OMS services occurred during the

during the COVID-19 pandemic in Australia. There was a

first wave of the pandemic. Total services recovered during

30% reduction in consultations during the second quarter

the second half of 2020 to reach pre-pandemic levels,

of 2020. This corresponded to the peak of the first wave

however the relative proportion of treatments subtypes is

of the pandemic and national lockdown (Figure 4), with

changing.

patients avoiding face-to-face attendances with all medical personnel. 8 Non-emergency dental treatment was also

There was limited uptake of telehealth services by oral

heavily restricted during this period. Given that dentists

and maxillofacial surgeons, making up only 5.1% of total

and dental specialists represent the source of 80% of OMS

consultations since implementation. In contrast, telehealth

referrals in Australia, dental service restrictions likely had

consultations made up 35.6% of total consultations for

a direct effect in further reducing OMS consultations.4

general practitioners and 38.5% of total consultations

Consultations rebounded in the second half of 2020, with

for non-GP specialists in Australia. 8 A limitation of this

increases of 14% and 17% in the third and fourth quarters

report is that it did not break down into surgical and non-

respectively, before stabilising to pre-pandemic levels in

surgical non-GP specialist services, therefore the impact

2021. Similarly, OMS treatments were reduced by over

on surgical specialists is not clearly defined. 8 There are

one third during the second quarter of 2020. Category

obvious barriers to the use of telehealth services specific to

3 treatments (dual-qualified surgeons) rebounded with

OMS, such as difficulties in adequate intraoral examination,

increases of 8% and 10% in the third and fourth quarters

inability to palpate important structures, as well as

of 2020, respectively. This was associated with a change

inadequate staffing and facilities at the patient’s location

in the type of treatments delivered, including an increase

to assist in examination. 9-10 Despite this, there is evidence

in pre-prosthetic and implant surgery and a decrease in

to suggest a high level of concordance between face-to-

Figure 4 Cases of COVID-19 in Australia. Daily (left axis) and cumulative (right axis) COVID-19 cases diagnosed in Australia are shown with key time points for elective surgery procedures indicated.

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Figure 5 Patient information sheet in preparation for a telemedicine consultation. Reprinted with permission from Prasad et al, 2020.14

face and telehealth consultations for triage and treatment

and to prepare patients for the encounter. Patients may be

planning in OMS.

advised to organise a flashlight and retraction device to

10-12

Indeed, acceptance of telehealth by

oral and maxillofacial surgeons during COVID-19 has been

assist in examination. Providing written information may

high elsewhere.13 In this study, OMS consultation data

also help in optimising the consultation (Figure 5).15

was only available for clinicians utilising Category 4 of the MBS. They are likely to be older and may be less inclined

Following service restrictions on non-urgent elective

to use telehealth services, which may help to explain the

surgery and dental treatment the Australian and New

low uptake of telehealth. Telehealth services are likely to

Zealand Association of Oral and Maxillofacial Surgeons

have an expanding role in oral and maxillofacial surgery in

(ANZAOMS) provided guidelines for appropriate procedure

the future. In particular for patient populations who reside

categorisation to reduce the impact of COVID-19 on

in regional and rural areas who face the burden of travel

time-sensitive surgery. This included management of

distance when seeking OMS services. Increased travel

suspected or existing head and neck cancers, craniofacial

distance has been found to be associated with more severe

and dentoalveolar trauma and infections not responding to

odontogenic infections, and higher rates of loco-regional

non-surgical measures.16 Despite this, this study showed

failure from oral tongue squamous cell carcinoma.

a large reduction in the treatment of oral and maxillofacial

Telehealth consultations could play an important role for

pathology which persisted even when total treatment

regional and rural patients in accessing timely care and oral

volume returned to pre-pandemic levels. Concerningly, there

cancer screening. A number of strategies are important

is emerging international evidence of delayed diagnoses of

to help increase uptake and effectiveness of telehealth

head and neck cancers resulting from system-wide health

services. This can include administrative staff contacting

service disruptions due to COVID-19.18-20 It is likely that this

patients prior to the consultation to test video technology

is also the case in Australia, where 430 cases of head and

14,15

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neck cancer are diagnosed per month, with one third of

knowledge of the infectious disease; during the COVID-19

these cases referred to oral and maxillofacial surgeons.21,22

pandemic this was directed toward minimising non-

For example, in Victoria, from April 2020 to October 2020,

essential contact with patients, and minimisation of

there was a 15% reduction in head and neck cancer

aerosol producing procedures, and utilisation of telehealth

notifications, representing 105 undiagnosed tumours.23 The

consultations where appropriate27.

delayed diagnosis and management of these patients may result in a higher tumour burden associated with greater

This study demonstrated significant differences between

morbidity and mortality. Ongoing monitoring of service

single qualified and dual qualified OMS surgeons in

levels, in particular of diagnosis and treatment of important

Australia. Over the five-year study period, item numbers

oral and maxillofacial pathology, should inform evidence-

used by single qualified surgeons remained stable or

based strategies to reduce the detrimental secondary

decreased, while those for dual qualified surgeons has

public health impacts of COVID-19.

steadily increased, reflecting the professional requirements of new OMS surgeons in Australia since 2004 to be

The reduction in services during the peak of the pandemic

qualified in both medicine and dentistry. Following the

has a number of secondary implications for the speciality.

first wave of the pandemic, treatment levels for dual-

This includes disruption of surgical education for OMS

qualified surgeons rebounded with significant increases

trainees, which is similar to the experience from trainees

in the third and fourth quarter of 2020. A similar rebound

around the world.24,25 The impacts of government

was not observed for single qualified surgeons. This may

restriction and reduction in patient volume on private OMFS

reflect differences in the scope of practice between these

surgeons also warrants further investigation. This will

groups.28

allow practices to better predict and prepare for ongoing disruptions related to staff employment, pre-emptive

This study quantified the effect of COVID-19 on OMS

purchase of equipment, and planning for access to private

services at a national level using robust modelling methods,

general anaesthetic facilities for future pandemics.

however these findings should be interpreted in the context of a number of limitations. OMS specific consultation data

Reductions in orthognathic surgery persisted in 2021,

was only available from Category 4 of the MBS. These

when total services returned to pre-pandemic levels. This is

clinicians will be registered as dental practitioners only.

likely to be due to the elective nature of most orthognathic

They will also be older and have worked for longer.27 This

procedures, as well as intermittent restrictions on dental

may affect the pattern of consultations and the proportion

services which deferred or extended essentially all

of consultations delivered via telehealth. Finally, data

orthodontic treatment during the pandemic. Delays in

presented here does not represent all oral and maxillofacial

orthodontic treatment during the pandemic have already

surgery services in Australia. The MBS does not include

been noted elsewhere.26 In contrast, there were increases

services provided to inpatients at public hospitals or

in Medicare-subsidised preprosthetic and implant surgery

services that are entirely private and do not attract a

following the national lockdown in early 2020. The specific

Medicare rebate, this includes most dentoalveolar surgery

drivers of this change are unclear. One explanation is that

(including most dental implants and third molar surgery).

clinicians may be shifting practice to facilitate the oral

These procedures are classified as aerosol generating and

rehabilitation of existing patients who have previously

therefore are likely to also have been significantly affected.

undergone head and neck cancer resection. Ongoing

Despite these limitations, these findings are still largely

monitoring of service levels will be important to better

reflective of the overall impact of the of COVID-19 on oral

understand how the pandemic is changing the type of

and maxillofacial surgery services in Australia.

treatments being provided by OMS surgeons and prepare the speciality to meet these demands. The current study has demonstrated that there were significant changes in the practice of OMS during the COVID-19 pandemic period between January 2020 to December 2020. Significant decreases in elective surgical output were demonstrated early in the pandemic response, and the uptake of telehealth consultations was noted throughout this period. Preparation for future pandemics can be divided into general and specific preparedness.27 General preparation is based on principles of minimising risk, appropriate training of staff, and minimising nonessential services until specific knowledge of the disease threat is known.27 Specific preparation is guided by 156

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CONCLUSIONS The most significant impact of COVID-19 on OMS services

treatment subtypes is changing. The surgical needs of the

in Australia occurred during the first wave of the pandemic.

public should be balanced against the risk of COVID-19

Reduction in OMS services during this time likely had

transmission and the need to protect hospital capacity.

significant implications for patients, especially those

Ongoing surveillance of service levels is important to

requiring non-emergency surgery. Despite the profession’s

respond to and reduce the impact of the pandemic on OMS

attempt to protect access to care for patients requiring

patients, trainees and surgeons.

time sensitive treatment, system-wide reductions in services may have resulted in a delay of diagnosis and treatment of significant pathologies such as head and neck malignancies. While total services recovered to reach pre-pandemic levels in 2021, the relative proportion of

Conflict of interest statement No conflict of interest or funding sources are declared. Ethics approval was not required for this study.

Trends in Oral and Maxillofacial Surgery during the COVID-19 pandemic: The Australian Experience

Referen ce s 1.

World Health Organization. WHO Coronavirus Disease (COVID-19) Dashboard (2020). https://covid19.who.int

Givi B, Schiff BA, Chinn SB, et al. Safety recommendations for evaluation and surgery of the head and neck during the COVID-19 pandemic. JAMA Otolaryngol Head Neck Surg 2020;146:579-584.

Australian Government Department of Health. COVID-19 Temporary MBS Telehealth Services (2020). http://www.mbsonline.gov.au/internet/ mbsonline/publishing.nsf/Content/Factsheet-TempBB

Singh KA BD, Spencer AJ, Goss AN. Practice patterns of oral and maxillofacial surgeons in Australia. Australian Institute of Health and Welfare (Population Oral Health Series No 3; 2004). https://www.aihw.gov.au/reports/dental-oral-health/practice-patterns-oral-maxillofacial-surgeons/ contents/table-of-contents

Services Australia. Medicare Item Reports (2020). http://medicarestatistics.humanservices.gov.au/statistics/mbs_item.jsp

Australian Government Department of Health. Medicare Benefits Schedule Book: Category 4. Canberra. 2020.

Coghlan A. A Little Book of R For Time Series (2018). https://buildmedia.readthedocs.org/media/pdf/a-little-book-of-r-for-time-series/latest/a-littlebook-of-r-for-time-series.pdf

Scott A. The impact of COVID-19 on GPs and non-GP specialists in private practice (2020). https://melbourneinstitute.unimelb.edu.au/__data/ assets/pdf_file/0003/3436014/UoM-MI-ANZ_Brochure-FV.pdf

Wood EW, Strauss RA, Janus C, Carrico CK. The use of telemedicine in oral and maxillofacial surgery. J Oral Maxillofac Surg 2016;74:719-728.

10.

Wood EW, Strauss RA, Janus C, Carrico CK. Telemedicine consultations in oral and maxillofacial surgery: A follow-up study. J Oral Maxillofac Surg 2016;74:262-268.

11.

Brownrigg P, Lowry JC, Edmondson MJ, Langton SG. Telemedicine in oral surgery and maxillofacial trauma: A descriptive account. Telemed J E Health 2004;10:27-31.

12.

Rollert MK, Strauss RA, Abubaker AO, Hampton C. Telemedicine consultations in oral and maxillofacial surgery. J Oral Maxillofac Surg 1999;57:136138.

13.

Al-Izzi T, Breeze J, Elledge R. Following COVID-19 clinicians now overwhelmingly accept virtual clinics in Oral and Maxillofacial Surgery. Br J Oral Maxillofac Surg 2020;10:290-295.

14.

15.

Sklavos A, Lee K, Masood M. The association of travel distance and severity of odontogenic infections. Oral Maxillofac Surg. 2022. doi: 10.1007/ s10006-022-01135-1. Online ahead of print.

16.

Prasad A, Brewster R, Newman JG, Rajasekaran K. Optimizing your telemedicine visit during the COVID-19 pandemic: practice guidelines for patients with head and neck cancer. Head Neck. 2020;42:1317-1321.

17.

Australian and New Zealand Association of Oral and Maxillofacial Surgeons. ANZAOMS Guidelines for Elective Procedure Categorisation – Oral

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and Maxillofacial Surgery. https://www.anzaoms.org/documents/item/523. 2020. 18.

Arduino PG, Conrotto D, Broccoletti R. The outbreak of Novel Coronavirus disease (COVID-19) caused a worrying delay in the diagnosis of oral cancer in north-west Italy: The Turin Metropolitan Area experience. Oral Dis 2021;27 (Suppl 3):742-743.

19.

Gilligan G, Lazos J, Piemonte E, Criado E, Pánico R. Delays in the diagnosis of oral cancer due to the quarantine of COVID-19 in Córdoba, Argentina. Spec Care Dentist 2020;40:618-620.

20.

Sud A, Torr B, Jones ME, et al. Effect of delays in the 2-week-wait cancer referral pathway during the COVID-19 pandemic on cancer survival in the UK: A modelling study. Lancet Oncol 2020;21:1035-1044.

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Australian Institute of Health and Wellbeing. Cancer data in Australia (2020). https://www.aihw.gov.au/reports/cancer/cancer-data-in-australia/ contents/summary

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22.

Kaing L, Manchella S, Love C, Nastri A, Wiesenfeld D. Referral patterns for oral squamous cell carcinoma in Australia: 20 years progress. Aust Dent J 2016;61:29-34.

23.

Te Marvelde L, Wolfe R, McArthur G, Blake LA, Evans SM. Decline in cancer pathology notifications during the 2020 COVID-19-related restrictions in Victoria. Med J Aust. 2021;214:281-283.

24.

Brar B, Bayoumy M, Salama A, Henry A, Chigurupati R. A survey assessing the early effects of COVID-19 pandemic on oral and maxillofacial surgery training programs. Oral Surg Oral Med Oral Pathol Oral Radiol 2021;131:27-42.

25.

Huntley RE, Ludwig DC, Dillon JK. Early effects of COVID-19 on oral and maxillofacial surgery residency training—Results from a national survey. J Oral Maxillofac Surg 2020;78:1257-1267.

26.

Abed Al Jawad F, Alhashimi N. Orthodontic treatment pause during COVID-19 outbreak: Are we overlooking potential harms to our patients and their treatment outcomes? Dental Press J Orthod. 2021;26:e21ins2.

27.

Nesbitt I. Pandemic planning and its relevance to surgeons. Surgery (Oxf). 2021;39:444-448.

28.

Ricciardo P, Bobinskas A, Vujcich N, Nastri A, Goss A. Survey of Australasian oral and maxillofacial surgeons 2011 - scope and workforce issues. Int J Oral Maxillofac 2015;44:1569-1573.

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C O V I D - 1 9 SCIENTIFIC ARTICLE

UTILISATION OF ORAL AND MAXILLOFACIAL SPECIALIST TELEHEALTH SERVICES DURING COVID-19 IN AUSTRALIA

A trend analysis

ABSTRACT

Masongo C (BSc (Hons))† Chih H (BSc (Hons), MBiostat, PhD)† Daire J (BSc (Nurs), MA (HlthPolPlan), PhD)†

Objectives:

Estai M (MBBS), MSc, PhD)‡ Gebauer D (BDSc, MBBS, FRACDS (OMS), MClinRes)‡ § Smith L (BDSc, MBBS, PGradDip (OMS), FRACDS (OMS)

To investigate the trends in the use of MBS (Medicare Benefits Schedule) specialist telehealth services by patients and their potential implications for oral and maxillofacial surgery.

† Curtin School of Population Health, Curtin University, Bentley, Western Australia, Australia

Methods:

‡ School of Human Sciences, University of Western Australia, Crawley, Western Australia, Australia

telehealth services (video conference and telephone) from May

Department of Oral & Maxillofacial Surgery, Royal Perth Hospital, Perth, Western Australia

A retrospective evaluation of deidentified Medicare Benefits Schedule data from Services Australia of claims made under specialist 2020 to May 2022, a billing method used by oral and maxillofacial surgeons was obtained. The uptake of each specialist telehealth service item was reported across Australian states and territories by gender and age groups. Descriptive statistics were used to compare uptake among groups, and the most and least number of claims were identified.

Corresponding Author: HUIJUN CHIH Curtin School of Population Health Bentley, Western Australia, Australia Email: h.chih@curtin.edu.au

Results: Over 2.5 million services were billed under specialist telehealth items from the MBS. Video conferencing usage was five times greater than telephone telehealth services during the study period of 2020 to 2022. Across all telehealth items, Victoria had the highest uptake, whereas the Northern Territory had the lowest uptake per capita. Females were 18% more likely than males to undergo a specialist telehealth consultation. Video conferencing consultations were more popular with those aged 55 to 74, with 30% of claims filed under these items, while telephone consultations were more popular with those aged 35 to 54, with 39% of claims reported for the items. The subsequent telehealth services were utilised twofold over the initial telehealth services. Conclusion: The trends in specialist telehealth services uptake for oral and maxillofacial surgery demonstrate a clear demand for such services

Keywords:

post-COVID pandemic. Hence, providing practice guidelines,

telehealth | COVID-19 | specialist care access |

educational materials, and training tailored to the needs of specific

barriers | oral and maxillofacial care |

populations has the potential to enhance both the patient and provider

patient uptake

experience of using telehealth. ANZAOMS PREVIEW VERSION |

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INTRODUCTION

Australian specialist telehealth services are available through the Medicare Benefits Schedule, ensuring equitable access to healthcare services and financial coverage.1 These services have proven successful in screening, diagnosis, treatment management, follow-up, general assessment, and treatment planning across specialties requiring extensive physical examination, such as oral and maxillofacial surgery.2 Telehealth services have been especially critical during the COVID-19 pandemic, helping patients access healthcare while avoiding unnecessary physical contact. 3 In March 2020, new funding was introduced under the Medicare Benefits Schedule (MBS) for telehealth services in response to the COVID-19 pandemic.1 As part of the response to COVID-19, telehealth services for oral and maxillofacial surgery were introduced in the form of videoconferencing item codes 54001 (initial) and 54002 (subsequent) and telephone telehealth item codes 54003 (initial) and 54004 (subsequent).4 This funding has expanded the scope of specialist telehealth services to videoconferencing services and patients living in rural and remote areas.1 Providing specialist services in this manner proved safer and more cost effective. 5 This policy response contributed significantly to the adoption of telehealth during COVID-19. Barriers have been identified as the overuse of services and the build-up of existing limitations. 5 This evidence created an understanding of telehealth services used in Australia during COVID-19 and insight into potential future implementation. There has been an emphasis on determining future policy measures that will not increase risk, but ensure continuity of quality and safety of care while alleviating social barriers. 5,6 This affirms the importance of telehealth policies that are both effective and beneficial to all stakeholders. 5 An individual’s ability to benefit from telehealth is determined by factors such as accessibility and availability of resources, relationships with providers, the individual’s condition and health, digital literacy, and provider operating conditions.7,8 To determine the best direction for the implementation of telehealth, these factors should be considered when developing telehealth policies. 9 Optimising telehealth’s potential requires an understanding of these determinants in particular clinical specialties. 5,6 An increase in mobility difficulties among older adults as due to physical and cognitive limitations they age makes it difficult for them to visit their healthcare providers, and more women than men are affected by these limitations.10,11,12 Access to transportation, long waiting times for appointments, and financial burden as a result of multiple health conditions requiring extensive healthcare are a few of these issues.10,11 Another common issue has been technology literacy.13 A significant proportion of the elderly population has difficulty understanding and utilising technology for information and services13. In a study of older Australians’ digital behaviour, males report being more proficient at performing basic and advanced digital tasks than females.13 Furthermore, older age groups (80 and over) reported having less access to digital technology than younger older adults (50-69).13 To meet the needs of the aging population, interventions must balance meeting demand and meeting challenges

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while life expectancy and the proportion of older people

To explore the future implications of MBS specialist

increase.10,11 Hence, advancement in technology has great

telehealth used in oral and maxillofacial surgery, further

potential to not only provide accessible healthcare services

research is required. By leveraging telehealth, there is

to wider population groups it also offers the opportunity to

a greater capacity to bridge the gap in care inequalities

diversify healthcare to meet the preferences of patients as

and widen the scope of oral and maxillofacial healthcare

well as providers. 5

services available to patients. For this reason, this study aims to examine trends in the uptake of specialist

Researchers previously examined the uptake of COVID-19

telehealth services by patients of different characteristics

telehealth item codes for oral and maxillofacial surgical

and discusses the future expectations of specialist

services provided by clinicians with a single dental/

telehealth services for oral and maxillofacial telehealth in

fellowship qualification. A significant increase in uptake

Australia based on the trends in patient uptake. There is a

was observed during the rollout of these services. There

focus on the following key objectives:

is, however, limited information available on the uptake and use of oral and maxillofacial specialist telehealth services used for billing purposes by dual-qualified oral and maxillofacial surgeons in Australia during COVID-19. Considering that the use of telehealth is expected to

increase in the future, there is a need for telehealth

To explore the trends in telehealth use during the COVID-19 pandemic in Australia by age and gender groups; and

policies tailored towards specific specialists to guarantee successful outcomes.7

Identify the trends in the uptake of telehealth service use across Australian States and Territories by age and gender groups.

METHODS Study design and data collection A retrospective descriptive study examined the trends in

These items are used by dual-qualified oral and

telehealth use from specialist MBS telehealth consultations

maxillofacial surgeons. Item numbers used by single dental

claimed during COVID-19 (May 2020 to May 2022). To

degree-qualified surgeons, who represent a decreasing

capture data on uptake during the COVID-19 peak in

proportion of older surgeons (10%), are not captured in this

Australia, a two-year period was chosen. The sampling

analysis.

frame was the open-access Medicare Statistics Database (Services Australia, 2022). The data were collected

To identify trends in how telehealth was utilised during

from a purposive sample of patients who had received a

this period, the per capita rates for utilisation of telehealth

telehealth consultation billed under a specialist telehealth

service items were compared between states and

MBS item number between May 2020 and May 2022.

territories. This was done to determine trends in the uptake

The relevant data were deidentified, processed and

of telehealth services. The overall patterns of uptake across

made available through Services Australia, an executive

gender and age groups were measured by aggregating

agency of the Australian Government.14 Inclusion criteria

the number of monthly claims made during the observation

included patients billed by specialist clinicians for the four

period by gender and age groups for each item.

items (91822, 91823, 91832, 91833) in Australia. The numbers, which include a mix of consultations and post-

Ethics approval for this study was granted by the Curtin

operative follow-up, are used for initial telephone (91822),

Human Research Ethics Committee (Approval number:

subsequent telephone (91823), initial videoconferencing

HRE2022-0317-02).

(91832), and subsequent videoconferencing (91833).

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MBS activity data for each specialist telehealth item (codes 91822, 91823, 91832, 91833) were extracted as comma-separated values (CSV) files. Summary statistics of frequency and proportion of telehealth consultations (items 91822, 91823, 91832, 91833) claimed from May 2020 to May 2022 were performed. The data were also reported by year of the observation period (May 2020- April 2021; May 2021-May 2022) across all Australian states and territories by gender (males and females) and age (from 0-14 to 75+ in intervals of 15 years) groups. Reports for the overall trends in specialist telehealth services uptake were also generated per capita amongst states and territories. Item report statistics were compiled, arranged, and analysed using Excel spreadsheets (Microsoft 2023).

Consultation Services Claimed N (Thousands)

Data analysis

70 60 Male

50 Female

40 30 20 10 0

ACT

NSW

QLD

TAS

VIC

States and Territories Figure 1 Claims made for male and female patients under item 91822

Comparing claims made for male and female patients under item 91822, 30% more consults were claimed for female patients than male patients. The median uptake

Results

among female patients was 3,578 (IQR 1,539-17,180),

There were more than 2.5 million specialist telehealth

while the median uptake among male patients was 2,491

services claimed under the MBS between May 2020 and

(IQR 920-10,017) (Figure 1). Female patients were more

May 2022. The first year of the observation period saw the

likely to receive an initial telephone consultation than their

submission of approximately 1.2 million claims (May 2020

male counterparts, with a difference of 1,087 consults.

to April 2021). There was an increase in the number of

Over the two-year observation period, female patients

claims made in the second year of the observation period

from New South Wales reported the highest uptake of the

(May 2021 to May 2022), with over 1.3 million (53%) claims

initial telephone item, which represented 15% of all initial

being made.

telephone claims. Contrary to this, male patients from the Northern Territory had the lowest uptake of the initial

Demographic results revealed that 59% of claims were

telephone item.

submitted for female patients, while 41% were submitted for male patients. Amongst age groups, 34% of claims

A total of 418 (IQR 136-1858.75) median claims were

were recorded for patients aged 55 to 74. This was

reported under the initial telephone service across all age

followed by patients aged 35 to 54 (30%), patients aged

groups. In total, 42% of all claims billed under the initial

15 to 34 (18%), patients aged 75 and over (16%), and the

telephone item were for patients 35 to 54 years of age

youngest age group with ages 0 to 14 (2%).

(Figure 2). More individuals aged 35 to 54 years from New South Wales opted for the initial telephone item than the

In this period, 69.7% of the total claims were submitted

other age groups across Australia. The lowest uptake

under the subsequent videoconferencing item (91833).

of initial telephone service was in the Northern Territory

The initial videoconferencing item was the second most

among individuals aged 75 and older, accounting for less

reported item, accounting for 12.5% of all claims. The

than 1% of claims under item 91822 (Figures 1-2).

third-most reported item was subsequent telephone item, which accounted for 11.5% of all claims. Over the period of

Item 91823 (Subsequent telephone)

observation, the initial telephone item had the least number of reported claims, at 6.3%.

The uptake for the subsequent telephone items were similar across the States and Territories. Majority of claims of this

Item 91822 (Initial telephone)

item 91823 were filed by patients in Victoria (44%), with 1942 claims reported per capita. The Northern Territory

The number of claims made under the initial telephone

had the lowest reported uptake of claims under this item

item indicates a substantial difference between states

with 264 claims made per capita.

and territories. Results revealed that 41% of consultations were claimed in Victoria, with 990 claims made per capita. The lowest number of claims was recorded the Northern Territory, with 219 claims made per capita during the observation period.

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Consultation Services Claimed N (Thousands)

VIC

TAS

QLD

NSW

0 - 14

15 - 34

35 - 54

55 - 74

ACT

75 +

Age Group (Years)

Figure 2 Claims under item 91822 made for all patients

There had been 24% more consultations claimed for female

Item 91832 (Initial video conferencing)

patients under the subsequent telephone item (median: 6,919 (IQR 2,233-31,791) than male patients, who claimed

It was reported that most claims were submitted in Victoria

4,859 consultations (IQR 1,280 - 18,954) (Figure 3).

(38%), where 1,833 claims were made per capita, this

Victorian females reported the highest proportion of claims,

reflected the population’s significant uptake of the initial

representing 14% of claims. Females had a higher reported

videoconferencing service. The Northern Territory had the

uptake compared to males across all States and Territories.

lowest number of claims at this time. However, the lowest per capita uptake was reported in Queensland at 565 claims per capita.

Across the age groups, the highest uptake of item 91823 was observed among patients 35 to 54 years old, with a median uptake of 2,131. The lowest uptake was observed

Australia experienced a high uptake of the initial

among 0-14-year-olds, with a median of 314 claims. A

videoconferencing telehealth service, particularly among

comparison of the uptake across States and territories

females, who had a 16% higher uptake than males

showed that New South Wales had the highest rate of

(Figure 5). The median number of reported claims for

uptake by age group (Figure 4) for those aged 35 to 54 who

female patients was 10,984 (IQR 2,030-29,612), and for

received a telephone consultation within the second year

male patients, 10,773 (IQR 2,239-20,999).

of the observation period (9% of all claims were reported under this category). The Northern Territory reported the

It was observed that individuals aged 55 to 74 had the

lowest number of consultations among those aged 75 and

highest uptake of the initial video conferencing item, with

older (Figures 3-4).

a median of 4320 (IQR 675-8056), representing 34% of all claims (Figure 6). Victorians aged 55 and 74 had the highest reported number of claims during the observation period. The Northern Territory’s 0 to 14 year-olds were the least likely to use the initial video conferencing item

Consultation Services Claimed N (Thousands)

140

120 Male 100 Female 80

Consultation Services Claimed N (Thousands)

(Figures 5-6).

WA 120 VIC 100 TAS 80 SA 60 QLD 40 NT 20 NSW 0

0 ACT

NSW

QLD

TAS

VIC

States and Territories

Figure 3 Claims made by male and female patients under item 91823

0 - 14

15 - 34

35 - 54

55 - 74

ACT

75 +

Age Group (Years)

Figure 4 Claims under item 91823 made for all patients ANZAOMS PREVIEW VERSION |

163

Consultation Services Claimed N (Thousands)

140

120 Male 100 Female 80

0 ACT

NSW

QLD

TAS

VIC

States and Territories

Figure 5 Claims made by male and female patients under item 91832

first year of the observation period, representing 11% of all

Item 91833 (Subsequent video conferencing)

the uptake for the item. It was found that Victorian patients were most likely to use the subsequent video conferencing item, where 10,240

There was a high uptake of the subsequent video

consultation claims were recorded per capita. In the

conferencing item among individuals aged 55 to 74, with

Northern Territory, 1,959 claims were reported per capita;

a median uptake of 25,256 (IQR 5,240-57,488) (Figure 8).

this was the lowest uptake reported under this item across

The lowest uptake was observed in the 0 to 14-year-old

all States and Territories.

age group, where the median uptake was 1,721 (IQR 3014,665).

The number of claims for the subsequent video conferencing item involving women was 18% higher than

The highest uptake by age group across all states and

those involving men (Figure 7). For female patients, the

territories was seen among Victorians aged 55 to 74, which

median number of telehealth consultations claimed under

was reported between May 2020 and April 2021. This

item 91833 was 74,301 (IQR 14,091-185,514). Male

group accounted for approximately 7% of the total use

patients’ median claim number was 58,354.5 (IQR 13,824-

of the item by age group across all states and territories

131,567). Uptake by gender across States and Territories

(Figures 7-8).

Consultation Services Claimed N (Thousands)

was highest among Victorian females; this occurred in the

WA 120 VIC 100 TAS 80 SA 60 QLD 40 NT 20 NSW 0 0 - 14

15 - 34

35 - 54

55 - 74

75 +

ACT

Age Group (Years) Figure 6 Claims under item 91832 made for all patients

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Consultation Services Claimed N (Thousands)

700

600 Male 500 Female 400

300

200

100

0 ACT

NSW

QLD

TAS

VIC

States and Territories Figure 7 Claims made by male and female patients under item 91833

700

Consultation Services Claimed N (Thousands)

WA 600 VIC 500

TAS

400

300

QLD

200

100

NSW

ACT

0 0 - 14

15 - 34

35 - 54

55 - 74

75 +

Age Group (Years) Figure 8 Claims made under item 91833 made for all patients

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DISCUSSION Telehealth services were welcome across Australia during

The analysis of item claims revealed that females are more

the COVID-19 pandemic, with uptake trends remaining

likely to use specialist telehealth services, irrespective of

steady across States and Territories, as reflected in MBS

the telehealth consult type. Female patients tend to be

claims made under a specialist telehealth item between

more likely to receive medical treatment than males.22

May 2020 and May 2022. The current study revealed that

However, they also experience significant difficulties

the uptake of all follow-up items was significantly higher

accessing healthcare.12,22 The higher female uptake in

than the initial items. As patient needs and demands

specialist telehealth services suggests that telehealth

evolve, and higher level of patient satisfaction is met, it is

is advantageous to female patients22. It is speculated

likely that future use of MBS specialist telehealth services

that more women are taking advantage of specialist

will increase. The future integration of telehealth into

telehealth services, which may enable them to remain more

specialties such as oral and maxillofacial surgery is

autonomous and in control of their healthcare.22

expected to increase as a result. As such, it is important to 7

plan and ensure that the supply and demand of services,

Across all age groups, it was observed that those aged

and providers in specialties such as oral and maxillofacial

35 to 54 used telephone services more frequently.

surgery are not at odds. 5,17

Comparatively, videoconferencing items were more popular among those aged 55 to 74. These findings are

All states and territories demonstrated a higher uptake

consistent with findings by the Australian Bureau of

of follow-up items than initial items. As evidenced by the

Statistics concerning patient experiences with telehealth

higher number of claims submitted towards the subsequent

services, showing that older age groups are more likely to

items, there is a potential need and demand for these

use telehealth services than younger age groups.23 This

items18 . This finding implies that patients and providers

trend supports the assertion that older age groups favour

are increasingly seeking out or providing these services,

the convenience of telehealth. 8 As discussed in previous

which could be due to various factors, such as improved

literature, factors related to accessibility and disease

access to healthcare services, higher levels of awareness,

burden play a crucial role in this higher use of telehealth

or provider preferences as prior studies demonstrated the

among these population groups.10 As older adults

effectiveness of telehealth in oral and maxillofacial surgery

experience high morbidity, telehealth becomes increasingly

in reducing accessibility issues for patients.6, 18,19,20

convenient, particularly for managing ongoing treatments on a self-directed management plan. 8,25 Taking into account

State and territory trends in specialist telehealth services

the needs of an aging population and female patients, the

further reinforce the need and demand to provide equitable

use of telehealth is expected to continue to increase in these

access9. Across most telehealth items, the Northern

groups. For future telehealth implementation, it is critical

Territory had the lowest rate of claim submissions per

to understand how gender and age uptake of specialist

capita. This can be explained by differences in population

telehealth services are interdependent with accessibility,

sizes, but also by the fact that the Northern Territory has

technology infrastructure, and digital health literacy.25

the highest proportion of Indigenous residents among its

Policymakers and providers may need to tailor to the needs

population who are faced with significant accessibility

of these patients accordingly.

barriers to healthcare. 9, 21 It has been noted in prior studies that minority populations are less likely to use telehealth.22

The trends in specialist telehealth claims during the

This phenomenon could have contributed to the lower rate

COVID-19 pandemic between May 2020 and May 2022

of telehealth claim submissions in the Northern Territory,

provide insights into the integration of telehealth in

highlighting the importance of developing strategies to

specialties fields such as oral and maxillofacial surgery.

increase telehealth use among minority populations.

Developing policies in response to these trends may

These findings highlight the potential for telehealth to

facilitate efficient resource allocation and contribute to

significantly enhance access to oral and maxillofacial

meeting the demand for oral and maxillofacial services. 5,17

services in Australia, thereby increasing equitable access to

As telehealth uptake varies between gender and age

care for all Australians. Oral and maxillofacial consultants

groups across states and territories, to maximise

have cited an increase in accessibility as a significant

the impact, telehealth strategies should be designed

contributor to providing telehealth services to their

considering population needs, particularly concerning

patients.20

accessibility.

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Following the findings of this study, future implementation

Limitations

of specialist telehealth services will focus on the development and planning of solutions based on areas

The MBS data were aggregated; therefore, it is not possible

of greatest need. Telehealth solutions that improve

to identify and provide information about the experiences

accessibility, ensure adequate technology infrastructure,

and outcomes of individual outcomes. For instance, the

and address privacy and quality concerns from both

number (multiple) of telehealth consultations attended

patients and providers should be prioritised. Incorporating

by a single patient for any item could not be established,

features that allow for remote monitoring and follow-up

which would be useful for health professionals to assess

into telehealth solutions will ensure better communication

the effectiveness of telehealth services accurately. Further

by fostering a stronger patient-clinician relationship.

research should ensure that the telehealth policy for oral

Providing education and training to patients experiencing

and maxillofacial surgery is based on sound logic and is

the greatest inequalities in access can be essential to

reflective of current reality. Qualitative research methods

ensuring the highest level of healthcare post-pandemic.25,26

can facilitate a deeper understanding of the motivations

In addition, establishing practice guidelines based on the

and barriers associated with MBS specialist telehealth

specific needs of a particular population can facilitate

items.

telehealth services implementation. Nevertheless, it 26

is important also to examine the influence of oral and maxillofacial consultants. Their perceptions of use and decisions to offer these telehealth services post-pandemic may assist in providing better healthcare for oral and maxillofacial patients of different needs.

CONCLUSION This study illustrated variations in the uptake of different

Acknowledgements

specialist telehealth services by patients of different age and gender groups. In order to identify the most effective

This study has been generously funded by ANZAOMS

integration policies and initiatives for telehealth availability

Research and Education Foundation and the Trust.

and accessibility that can benefit both patients and providers, it is important for future studies to examine the consultants’ perceptions of use and their decisions to offer telehealth services for oral and maxillofacial surgery and further the patient’s perceptions of using the services.

Conflict of interest statement There are no declared competing interests.

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