African Journal of
THORACIC AND CRITICAL CARE MEDICINE VOLUME 24
|
NUMBER 2
|
JUNE 2018
OFFICIAL JOURNAL OF THE SOUTH AFRICAN THORACIC SOCIETY ISSN 2304-0017
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AFRICAN JOURNAL OF
THORACIC AND CRITICAL CARE MEDICINE VOLUME 24 | NUMBER 2 | JUNE 2018
CONTENTS 74
74 75
76
82
87
EDITORIAL
Thoracic endometriosis syndrome at the University of Ilorin Teaching Hospital A Linegar The evaluation and treatment of postintubation tracheal strictures I Schewitz Caregiver education for childhood respiratory conditions in Africa R J Green
ORIGINAL RESEARCH
Assessment of the knowledge of teachers about asthma and the availability of facilities for asthma care in public secondary schools in Lagos, Nigeria O O Adeyeye, Y A Kuyinu, O B Ozoh The evaluation and surgical management of post-intubation tracheal strictures at a thoracic surgery referral centre in South Africa S Ramghulam, R Perumal, D Reddy Thoracic endometriosis syndrome at University of Ilorin Teaching Hospital P O Adeoye, A S Adeniran, K T Adesina, O A Ige, O R Akanbi, A Imhoagene, O O K Ibrahim
AJTCCM EDITOR-IN-CHIEF Prof. K Dheda DEPUTY EDITOR Prof. C Koegelenberg SECTION EDITOR Breath-taking News: Prof. E Irusen EDITORIAL BOARD Prof. G Ainslie, Prof. E Bateman, Prof. R Green, Prof. E Irusen, Prof. M Jeebhay, Prof. P Jeena, Prof. U Lalloo, Prof. A Linegar, Prof. R Masekela, Dr K Nyamande, Dr J O’Brien, Dr R Raine, Prof. G Richards, Dr R van Zyl Smit, Prof. M Wong, Prof. H Zar INTERNATIONAL EDITORIAL BOARD Prof. A Cattamanchi - USA Prof. F Chung - UK Prof. G B Migliori - Italy Prof. S Sharma - India Prof. W W Yew - China Prof B Kirenga, Kampala - Uganda Prof A Katamba, Kampala -Uganda PRESIDENT SA THORACIC SOCIETY Prof. U Lalloo
HMPG
CEO AND PUBLISHER Hannah Kikaya Email: hannahk@hmpg.co.za EXECUTIVE EDITOR Bridget Farham
92 97
99
CASE REPORT
A bronchogenic cyst masquerading as asthma: A case report M D Moremi, N J Maligavhada, N G Tiva, R T Mamogale, S M Risenga Complete lung collapse in a young adolescent T H Ahmed, A Esmail, G Calligaro, K Dheda
BT NEWS
MANAGING EDITORS Naadia van der Bergh, Claudia Naidu TECHNICAL EDITORS Naadia van der Bergh Kirsten Morreira PRODUCTION MANAGER Emma Jane Couzens SENIOR DESIGNER Clinton Griffin CHIEF OPERATING OFFICER Diane Smith | Tel. 012 481 2069 Email: dianes@hmpg.co.za JOURNAL ADVERTISING Reneé Hinze Ladine van Heerden ONLINE SUPPORT Gertrude Fani | Tel. 021 532 1281 Email: publishing@hmpg.co.za
The Editor African Journal of Thoracic and Critical Care Medicine PO Box 13725, Mowbray, 7705 Tel. 021 650 3050 | Fax. 021 650 2610 | Email. admin@pulmonology.co.za The views expressed in individual articles and advertising material are the personal views of the authors and are not necessarily shared by the editors, the advertisers or the publishers. No articles may be reproduced without the written consent of the publishers. The AJTCCM is published by the Health and Medical Publishing Group (Pty) Ltd. Co. registration 2004/0220 32/07, a subsidiary of SAMA. HEAD OFFICE: Block F, Castle Walk Corporate Park, Nossob Street, Erasmuskloof Ext. 3, Pretoria, 0181 EDITORIAL OFFICE: Suite 11, Lonsdale Building, Lonsdale Way, Pinelands, 7405 | Tel. 021 532 1281 All letters and articles for publication must be submitted online at www.AJTCCM.org.za Email: publishing@hmpg.co.za
FINANCE Tshepiso Mokoena HMPG BOARD OF DIRECTORS Prof. M Lukhele (Chair), Dr M R Abbas, Mrs H Kikaya, Dr M Mbokota, Dr G Wolvaardt PRINTED BY TANDYM PRINT
EDITORIAL
Thoracic endometriosis syndrome at the University of Ilorin Teaching Hospital This issue of the AJTCCM includes a retrospective review by Adeoye et al.[1] on their experience with thoracic endometriosis syndrome, as seen over a 3.5-year period at a teaching hospital in Ilorin, Nigeria. Thoracic endometriosis syndrome, which refers to the presence of endometrial tissue in the lung parenchyma or on the pleural surfaces, is an extremely rare condition. Presentation is variable, but patients can be broadly grouped into those who present with signs and symptoms related to catamenial pneumothorax, catamenial haemothorax or intrapulmonary (parenchymal or airway) nodules, which usually present with haemoptysis. In the reported study, pleural effusion was the most frequent presenting sign. As with all rare conditions, diagnosis is often delayed. A diagnosis is based on a high clinical index of suspicion in the first instance. Pointers include: a cyclical presentation of chest pain, dyspnoea, cough or haemoptysis that occurs in relation to the menstrual cycle, cases presenting in women during the productive years and symptoms that affect the right hemithorax. Clinical suspicion should prompt investigation that involves a computed tomography scan and testing CA-125 serum levels. The authors recognise the importance of thoracoscopic techniques for obtaining a histological diagnosis and intrathoracic management of the pleural space. They also mention the inadequacy of basing
the diagnosis on pleural fluid and bronchial lavage cytology. Their observation that chemical pleurodesis alone has a poor success rate in patients with pleural effusions is also supported by the literature. They conclude that early thoracoscopic intervention is desirable, and that pleurectomy should replace pleurodesis when indicated. The authors have provided a concise summary and literature review of various aspects of this interesting and complex condition.
Anthony Linegar Head of Thoracic Surgery, University of Cape Town, Groote Schuur Hospital Affiliated Lecturer, University of the Free State, Universitas Hospital 206 Medical Chambers, N1 City Hospital, Goodwood, Cape Town al@thoracicsurgery.co.za 1. Adeoye PO, Adeniran AS, Adesina KT, et al. Thoracic endometriosis syndrome at University of Ilorin Teaching Hospital. Afr J Thoracic Crit Care Med 2018;24(2):87-91. DOI:10.7196/AJTCCM.2018.v24i2.201
Afr J Thoracic Crit Care Med 2018;24(2):74. DOI:10.7196/AJTCCM.2018.v24i2.213
The evaluation and treatment of postintubation tracheal strictures Tracheal strictures following prolonged ventilation have been well described for at least 50 years. The condition can be successfully treated with resection and end-to-end anastomosis. The use of low-pressure endotracheal cuffs, better attention to nursing detail and advances in invasive ventilation techniques have markedly decreased the incidence. However, strictures following intubation are unfortunately still observed. The paper by Perumal et al.[1] in this issue of the AJTCCM relates the South African experience for a condition that is becoming rare. In a developing country, where many patients are treated by nonspecialists, this paper is important as it highlights a potentially lifethreatening complication associated with invasive ventilation of a patient. Given the high incidence of trauma experienced in the country, patients may receive suboptimal treatment, especially in rural areas where specialists are not available. The misdiagnosis of asthma in a patient who has been ventilated needs to be stressed, as a stricture can develop after a surprisingly short period of ventilation. Suspected tracheal strictures should be referred to a centre of expertise as a matter of urgency. Symptoms
74 AJTCCM VOL. 24 NO. 2 2018
develop only after the diameter of the trachea has reduced by 50%, and are an indication of a significant stricture. The paper is a timely reminder of a potentially life-threatening complication of invasive ventilation, which is seen too often in developing countries and which should be eliminated with good medical and nursing care. Ivan Schewitz Department of Cardio-thoracic Surgery, University of Pretoria, and Linksfield Park Clinic, Linksfield, Johannesburg, South Africa ivan@schewitz.com
Afr J Thoracic Crit Care Med 2018;24(2):74. DOI:10.7196/AJTCCM.2018.v24i2.214 1. Perumal R, Ramghulam S, Reddy S. The evaluation and surgical management of postintubation tracheal strictures at a thoracic surgery referral centre in South Africa. Afr J Thoracic Crit Care Med 2018;24(2):82-86. https://doi.org/10.7196/SARJ.2018. v24i2.193
EDITORIAL
Caregiver education for childhood respiratory conditions in Africa The world has started to realise that both acute and chronic medical conditions are managed best when caregivers (parents and teachers) know something about the condition of the children in their care.[1] Even in developed nations, this process is limited by apathy on the part of medical personnel; in Africa, there is real concern that educational strategies for many conditions are seriously neglected. This problem may arise in part from the lack of government funding for educational programmes, and in part because many respiratory conditions are neglected, because management of HIV and tuberculosis (TB) has enjoyed greater emphasis in medical programmes. Asthma is a common chronic illness affecting children. Despite some educational programmes for management of chronic asthma being in place, very few stress the principles of managing acute exacerbations. In addition, students spend a considerable amount of time in school, and asthma control may be influenced by the knowledge of the teachers, and the facilities available to support them. In this issue of the AJTCCM, Adeyeye et al.[2] report on their study that assessed the knowledge of secondary school teachers in Lagos, Nigeria, regarding asthma, and evaluated the facilities and personnel available in the schools to support asthmatic students during emergencies. Of the 988 teachers sampled in this study, 475 (48.1%) had poor knowledge of managing asthma, 414 (41.9%) had fair knowledge and only 99 (10%) had what the authors describe as ‘good’ knowledge. None of the schools had a nebuliser available for treatment of an asthma emergency. The authors conclude that teachers in secondary schools in Lagos have unsatisfactory knowledge of managing asthma exacerbations. I would not be surprised if this finding were also to apply elsewhere in Africa. The response may be better in South Africa (SA), where the
National Asthma Education Programme has had input into school care for asthmatics, and the Allergy Society of SA has been in dialogue with school governing bodies with regard to the management of food allergies and anaphylaxis.[3,4] However, even in SA, this approach would not be available throughout the country. We need to learn, and learn fast, from our colleagues who manage HIV infection and TB. Those management programmes have had much success in promoting understanding, drug adherence and reporting of symptoms. We cannot stand by while children die of respiratory conditions such as asthma. These conditions are important in Africa. Robin J Green Department of Paediatrics and Child Health, Faculty of Health Sciences, University of Pretoria, Pretoria, South Africa robin.green@up.ac.za
Afr J Thoracic Crit Care Med 2018;24(2):75. DOI:10.7196/AJTCCM.2018.v24i2.215 1. Green RJ, Plit M, Du Plessis W, Rothberg AD. Asthma in South Africa – a long way to go. S Afr Med J 1998;88:1389-1393. 2. Adeyeye OO, Kuyinu YA, Ozoh OB. Assessment of the knowledge of teachers about asthma and the availability of facilities for asthma care in public secondary schools in Lagos, Nigeria. Afr J Thoracic Crit Care Med 2018;24(2):76-81. https://doi. org/10.7196/AJTCCM.2018.v24i2.192 3. Green RJ, Davis G, Price D. Perceptions, impact and management of asthma in South Africa: A patient questionnaire study. Prim Care Respir J 2008;17:212-216. https:// doi.org/10.3132/pcrj.2008.00027 4. Levin M, Van Niekerk A, Katz H, Stuurman C. Allergies in schools. Curr Allergy Clin Immunol 2016;29:272-276.
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RESEARCH
Assessment of the knowledge of teachers about asthma and the availability of facilities for asthma care in public secondary schools in Lagos, Nigeria O O Adeyeye,1 MBBS, MPH, FMCP; Y A Kuyinu,2 MBBS, MPH, FMCPH; O B Ozoh,3 MBBS, FMCP Department of Medicine, Lagos State University College of Medicine, Lagos, Nigeria Department of Community Medicine and Primary Health Care, Lagos State University College of Medicine, Lagos, Nigeria 3 Department of Medicine, College of Medicine, University of Lagos, Lagos, Nigeria 1 2
Corresponding author: O O Adeyeye (olufunkeadeyeye@yahoo.com)
Background. Asthma is a common chronic illness affecting young people. Asthma management at schools may be influenced by teachers’ knowledge of the condition and the availability of treatment facilities. Objective. To assess the knowledge of secondary school teachers in Lagos, Nigeria, regarding asthma and to evaluate management options available at schools. Method. A descriptive cross-sectional study was conducted. Schools were selected by proportional sampling of the educational districts, followed by stratified single-stage cluster sampling. All consenting teachers in the 54 selected schools were recruited. A self-administered questionnaire was used for data collection. A composite score was calculated, with 32 as the maximum possible. Knowledge was regarded as poor if scores were <16, fair for scores between 16 and 21, and good if scores were ≥22. Results. Results show that 475 (48.1%) of the respondents had poor knowledge, 414 (41.9%) had fair knowledge, and only 99 (10%) had good knowledge. Better knowledge about asthma was associated with personal experience (χ2=16.466; p=0.001) or history of a family member with the condition (χ2=6.412; p=0.04). Of the 54 schools surveyed, only 9 (16%) had a school clinic, while a school nurse was available at only 4 (7.41%) of the schools. None of the schools had access to a nebuliser in case of an asthma emergency. Conclusion. Teachers in secondary schools in Lagos have unsatisfactory knowledge about asthma and are not equipped to support affected students during an asthma episode. Afr J Thoracic Crit Care Med 2018;24(2):76-81. DOI:10.7196/AJTCCM.2018.v24i2.192
Asthma is a common chronic condition among children and young adults. It is estimated that 235 million people worldwide have asthma and that the condition was responsible for about 383 000 deaths in 2015.[1] According to the International Study of Asthma and Allergies in Childhood (ISAAC),[2] the prevalence of asthma among 13 - 14-year-olds in Nigeria was 10.7% in 1998 and has increased to 13% in 2002.[3] The management of non-communicable diseases such as asthma is not given priority by policy makers in Nigeria. This may be due, in part, to the high burden of communicable diseases and limited healthcare resources. In an earlier report, skin problems, malnutrition and respiratory tract illnesses were noted as the leading health problems among pupils in Lagos.[4] Although asthma was not specifically mentioned, it is likely that asthma contributed to the burden of respiratory illnesses. Asthma is one of the leading causes of absenteeism from school and work and often limits participation in physical and social activities, including exercise. In the USA, asthma is estimated to cause approximately 14 million absent school days each year.[5] There are no comparable data about absent school days due to asthma in Nigeria. Students with asthma can develop acute emergencies during school hours if exposed to triggers or when participating in exercise during
76 AJTCCM VOL. 24 NO. 2 2018
sporting activities. The competence of teachers with regard to first aid, the availability of treatment facilities and the presence of competent healthcare personnel may influence the outcome of such episodes. Delayed or inappropriate response to asthma emergencies has been reported to lead to asthma deaths in schools.[6] In many parts of the world, teachers’ knowledge regarding asthma has been reported to be only modest (about 40 - 70%) and lower still in disadvantaged communities.[7-10] To our knowledge, Nigerian teachers’ knowledge regarding asthma has not been reported nor have the resources for asthma care in schools been evaluated. Our study therefore investigated the level of knowledge about asthma among teachers in secondary schools in Lagos, Nigeria. It also described the factors associated with the level of knowledge among the teachers and evaluated the availability of facilities and personnel in schools to support asthmatic students during emergencies. As public schools serve a wide range of students, including those with limited access to basic healthcare, the findings from this study may help to make schools safer for students with asthma.
Methods
This was a descriptive cross-sectional study among teachers of selected schools operating under the Lagos State Government Ministry of
RESEARCH Education. The study was conducted between August and December 2016 in 54 public secondary schools in Lagos, which represent 20% of all the urban secondary schools in the city. The six educational districts in Lagos were used as sampling units. We obtained a list of public secondary schools in these districts and stratified them into urban and rural schools. We systematically selected the schools to be sampled from the 252 urban schools (Fig. 1). The schools were numbered per district and the second one in each district list was used as starting point. Every fifth school in a district was subsequently selected until the desired number of schools was obtained. As the districts do not all have an equal number of schools, the number of schools selected in a district was proportional to the total number of urban schools in that district. All the teachers in a selected school were eligible to participate in the study and all those who consented were included. The sample size was calculated according to the formula for determining proportions in a cluster survey of at least 20 000 teachers. [11] The assumption was that 50% of the teachers would have adequate knowledge. The minimum sample size was calculated as 660 teachers. We selected 30 teachers from each of at least 22 schools. A design effect size of 1.5 and a significance level of p<0.05 were used. A 47-item questionnaire, validated through use in previous studies,[12-14] was used to collect information on sociodemographic characteristics and personal history of asthma. Questions explored the teachers’ knowledge of asthma triggers, symptoms, severity, treatment options and associated conditions. The questionnaire also assessed common myths about asthma and teachers’ willingness to participate in an educational intervention programme. The proportion of correct responses was used as outcome. A score of 1 was assigned for a correct response to a question, whereas incorrect responses or ‘don’t know’ answers were assigned a score of 0. A composite score was then calculated, with the maximum attainable score being 32. Knowledge level was rated as poor if the composite score was <16, fair for scores between 16 and 21, and good for scores ≥22. A second questionnaire, completed by school principals, was used to assess the availability of treatment facilities at the school, such as a school clinic, access to a nebuliser, spare inhaler or spacer device, or the availability of a school nurse. 540 Schools
252 Urban
288 Rural
District I = 12
District II = 12
District III = 6
District IV = 6
District V=6
District VI = 12
Number of teachers proportionally selected
408
422
101
121
108
459
Number of teachers in the selected schools
293 (71.8)
250 (59.2)
51 (50.5)
60 (49.6)
74 (68.5)
260 (56.6)
Number of teachers that participated in the study (participation rate)
A total of 988 teachers participated in the study
Fig. 1. Flow chart for the selection of teachers from educational districts.
Statistical analysis
Data were analysed using SPSS version 24 (IBM Corp., USA). Means were calculated for all quantitative data and expressed as mean (standard deviation). The χ2 test was used to compare categorical variables, whereas quantitative data were compared using Student’s t test. A significance level of p≤0.05 was used.
Ethics
Ethical approval for the study was obtained from the Health Research Ethics Committee of the Lagos State University Teaching Hospital (ref. no. LREC/10/06/1022), the authorities at the Lagos State Ministry of Education (ref. no. LED/BES/S.191/T2/248) and the district tutors general. Informed consent was obtained from each school principal and the individual participating teachers.
Results
Of the 1 619 teachers in the districts, 988 completed the questionnaire acceptably. This translates to a participation rate of Table 1. Sociodemographic characteristics of respondents (N=988) Variables Frequency, n (%) Age (years) <20 4 (0.4) 21 - 30 73 (7.4) 226 (22.9) 31 - 40 424 (42.9) 41 - 50 260 (26.3) 51 - 60 1 (0.1) 61+ Mean (SD) age: 44.6 (8.7) Gender Male 355 (35.9) Female 633 (64.1) Marital status Married 845 (85.5) Widowed 32 (3.2) Divorced 2 (0.2) Separated 5 (0.5) Not specified 9 (0.9) Subject taught Art 307 (31.1) Social science 286 (28.9) Pure/applied science 13 (1.3) Post-qualification experience (years) 102 (10.3) ≤5 210 (21.3) 6 - 10 111 (11.2) 11 - 15 16 - 20 170 (17.2) 395 (40) >20 Mean (SD) post qualification experience: 17.2 (8.8) Personal history of asthma Yes 49 (5.0) No 932 (94.3) Family history of asthma Yes 363 (36.7) No 624 (63.2) SD = standard deviation.
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RESEARCH 61%. Sociodemographic characteristics are shown in Table 1. The sample comprised 355 male teachers (35.9%) and 633 female teachers (64.1%). The mean age of the teachers was 44.6 (8.7) years (range: 19 69 years). Most of the teachers had at least a Bachelor’s degree and an average of 17.2 (8.8) years’ post qualification teaching experience. The prevalence of previously diagnosed asthma among the teachers was 5%. A third of the respondents had a first-degree relative with asthma. Responses from the questionnaire showed that 735 (74.4%) of the teachers had heard about asthma before and 350 (35.5%) were aware of a student with asthma in their school. Teachers’ responses to questions about asthma symptoms, severity and comorbidities are shown in Table 2. About 40% of the teachers agreed that asthma is a common respiratory disorder in children and about one-third
(30.9%) knew that asthma can be associated with an allergy. Two-thirds (66.2%) of the respondents thought, incorrectly, that asthma is curable and three-quarters (75.2%) correctly indicated that asthma symptoms can be controlled with the use of proper medication. When asked about the symptoms of asthma, 644 (65.2%) respondents recognised persistent cough as a common symptom and an almost similar proportion (68.7%) knew that speech difficulty may occur during an asthma attack. About 20% of the teachers recognised a recurrent runny nose and an itchy skin rash as possible comorbidities in asthma. Teachers’ responses to questions regarding triggers and treatment are shown in Table 3. A large proportion of the teachers were aware of potential triggers of asthma episodes. Smoke was indicated as a trigger by 841 (85.1%) of the respondents, while 685 (69.3%) and 632
Table 2. Responses of secondary school teachers regarding asthma symptoms, severity and comorbidities (N=988) Yes, No, Survey question n (%) n (%) Asthma is a common respiratory disorder in children. 391 (39.6) 322 (32.6) Allergies are associated with asthma. 305 (30.9) 259 (26.2) Children with asthma have a low intelligent quotient (IQ). 213 (21.6) 375 (38.1) Asthma is curable. 654 (66.2) 84 (8.5) Asthma can be controlled with proper use of medication. 753 (75.2) 54 (5.5) Persistent cough may be a presentation of asthma. 644 (65.2 115 (11.6) Asthma may cause speech difficulty during an attack. 679 (68.7) 75 (7.6) Rapid breathing in a child may result from asthma. 528 (53.4) 108 (10.9) Noisy breathing may occur in a child with asthma. 559 (56.6) 119 (12.0) Chest discomfort may be a complaint in asthma. 505 (57.2) 153 (15.5) Asthma symptoms may develop in an otherwise healthy child. 411 (41.6) 163 (16.5) Agitation and restlessness may signify worsening of a symptom. 469 (47.5) 88 (8.9) Drowsiness and confusion may signify a severe problem in asthma. 526 (53.2) 93 (9.4) Students with asthma may have itchy eyes. 767 (77.6) 59 (6.0) Students with asthma could have a recurrent runny nose. 214 (21.8) 181 (18.3) Students with asthma could have an itchy skin rash. 215 (21.8) 115 (11.6) Students with asthma could exhibit other allergies, including food allergies. 287 (29) 330 (34) Table 3. Responses of teachers regarding asthma triggers and treatment (N=988) Yes Survey question n (%) Exposure to smoke leads to symptoms of asthma. 841 (85.1) Exposure to chalk dust leads to symptoms of asthma. 685 (69.3) Exercise leads to symptoms of asthma. 490 (49.6) Exposure to cold leads to symptoms of asthma. 685 (69.3) Exposure to dust leads to symptoms of asthma. 632 (64) Some over-the-counter medication for pain may lead to an asthma attack. 288 (29.1) Does the use of antibiotics relieve an asthma attack? 439 (44.4) Does a salbutamol/Ventolin inhaler relieve an asthma attack? 786 (79.6) Asthmic children require preventive treatment. 752 (76.1) Blue inhalers are used in an emergency. 600 (60.9) A severe asthma attack should be managed in hospital. 614 (62.1) Asthma can be managed with native medications. 349 (35.3) Children with asthma should not engage in sports. 536 (54.3) Asthmatic students should use their inhalers before exercise. 547 (55.4) Keeping the school free of pets is beneficial to asthmatic students. 767 (77.6) Keeping the classroom dust free is beneficial to an asthmatic child. 671 (67.9) Had previous training on asthma care 377 (38.2)
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No n (%) 55 (5.1) 127 (12.9) 160 (16.2) 72 (7.3) 51 (5.2) 113 (11.4) 92 (9.3) 21 (2.1) 149 (15.1) 27 (2.7) 93 (9.4) 215 (21.8) 148 (15) 117 (11.8) 59 (6.0) 61 (6.2) 611(61.8)
Do not know, n (%) 275 (27) 424 (42.9) 400 (40.5) 250 (25.3) 191 (19.3) 229 (23.2) 234 (23.7) 352 (35.6) 310 (31.4) 329 (33.3) 414 (41.9) 431 (43.6) 369 (37.3) 160 (16.2) 593 (60) 658 (66.6) 365 (36.9)
Do not know n (%) 92(9.3) 171 (17.8) 338 (34.2) 231 (23.4) 305 (30.8) 587 (59.4) 447 (44.3) 181 (18.3) 86 (8.8) 361 (36.5) 281 (284) 424 (42.9) 304 (30.8) 323 (32.8) 160 (16.2) 256 (25.9)
RESEARCH (64%) of respondents indicated that chalk dust and cold, respectively, can trigger an asthma episode. The majority of the respondents knew about the reliever medication Ventolin. About 90% of the teachers recognised the need to train teachers about asthma, while 909 (92%) were willing to participate in such training. A mean knowledge score of 15.31 (5.74) was achieved by the sample. Of the total number of respondents, 475 (48.1%) achieved a score below 15, indicating poor knowledge, while 414 (41.9%) achieved a score between 16 and 22, indicating fair knowledge. Only 99 (10%) of the teachers showed good knowledge (Table 4). Personal history of asthma was associated with better knowledge (χ2=16.466, p=0.001), as was a family history of asthma (χ2=6.412;
p=0.04) and having a student with asthma (Table 4). None of the other factors listed in Table 4 were found to have a significant association with knowledge level (p>0.05). Only 9 (16%) of the schools visited had a clinic on the premises and a school nurse was available at only 4 (7.4%) of the schools. None of the schools had access to spare reliever medication, spacers or a nebuliser.
Discussion
Our results show that knowledge about asthma was poor among secondary school teachers in Lagos, Nigeria. This may have serious consequences, as an unrecognised asthma attack may prove potentially fatal. It is surprising that although more than three-quarters of the teachers
Table 4. Association between sociodemographic characteristics of secondary school teachers in Lagos, Nigeria, and their knowledge level about asthma (N=988) Knowledge level, n (%) Poor (n=475) Fair (n=414) Good (n=99) p-value Sociodemographic characteristics χ2/t Age (years) 16.428 0.172 <20 1 (0.2) 3 (0.6) 0 (0) 23 (5.6) 8 (8.1) 21 - 30 42 (8.8) 31 - 40 119 (25.1) 86 (20.8) 21 (21.2) 43 (43.4) 41 - 50 209 (44.0) 172 (41.5) 104 (21.9) 129 (31.2) 27 (27.3) 51 - 60 61+ 0 (0) 1 (0.2) 0 (0) Gender Male 184 (51.8) 138 (38.9) 33 (9.3) 3.128 0.20 Female 291 (46) 276 (43.6) 66 (10.4) Level of education National Certificate of Education Bachelor’s degree Diploma in Education Master’s degree Doctorate Not specified Marital status Single Married Widowed Divorced Separated Not specified Post-qualification experience (years) ≤5 6 - 10 11 - 15 16 - 20 >20 Personal history of asthma Yes No Relative with asthma Yes No Student(s) with asthma Yes No
32 (6.7) 344 (72.4) 24 (5.1) 67 (14.1) 1 (0.2) 7 (1.5)
27 (6.5) 282 (68.1) 24 (5.8) 70 (16.9) 3 (0.3) 8 (1.9)
7 (7.1) 58 (58.6) 6 (6.1) 22 (22.2) 1 (0.1) 5 (5.1)
13.292
0.208
52 (10.9) 402 (84.6) 15 (3.2) 1 (0.2) 1 (0.2) 4 (0.8)
38 (9.2) 355 (85.7) 13 (3.1) 1 (0.2) 3 (0.7) 4 (1.0)
5 (5.1) 88 (88.9) 4 (4.0) 0 (0) 1 (1.0) 1 (1.0)
5.490
0.856
56 (11.8) 109 (22.9) 58 (12.2) 92 (19.4) 160 (33.7)
36 (8.7) 85 (20.5) 45 (10.9) 59 (14.3) 189 (45.7)
10 (10.2) 16 (16.3) 8 (8.2) 19 (19.4) 45 (45.9)
17.760
0.023
13 (2.8) 459 (97.2)
24 (5.8) 387 (94.2)
12 (12.2) 86 (87.8)
16.466
0.001
188 (40.6) 275 (59.4)
148 (36.7) 255 (63.3)
27 (27.3) 72 (72.7)
6.412
0.04
146 (32.2) 308 (67.8)
169 (41.9) 234 (58.1)
35 (36.8) 60 (63.2)
8.778
0.012
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RESEARCH have heard about asthma before, most were unaware of students being affected by asthma. This may lead to their failing to recognise symptoms and inadequate responses to the need of the students. The mean score in this survey was 15.31 (5.74) out of a possible 32. This is similar to the findings from a Bahraini study, in which teachers scored an average of 5.16 (2.18) out of 10.[15] The finding is also similar to that of a Spanish study, in which the mean score from a sample of 7 000 teachers recruited from 9 cities, was 16.0 (4.8) out of 31.[16] Only 10% of the teachers in our study showed good knowledge about asthma, which is unlike the findings from a Pakistani study in which more than half the teachers surveyed had good knowledge.[17] It should be noted, however, that the sample size in that study was only 330 teachers, which may render this comparison contentious. The low knowledge score in our study may be a reflection of the general lack of awareness about asthma in Nigeria and possibly be related to the chronic nature of asthma not being widely recognised. The prevalence of physician-diagnosed asthma among the teachers in our study was 5%, which compares well with the 5.8% found in a Spanish study.[16] As in some other studies, our results also showed that a personal history of asthma or having a relative with asthma was associated with better knowledge about the condition.[15-18] This was an expected finding, as exposure to health education in the course of their illness or as carers to affected family members may have translated to better knowledge and increased awareness among these teachers. In our study, neither gender nor teachers’ level of qualification affected the scores. This is different from the Pakistani study,[17] which found female teachers and those with a Bachelor’s degree or higher to have better knowledge about asthma. There was no evidence of formal training opportunities to equip teachers with knowledge to deal with students’ health concerns. However, about a third of the teachers indicated having had exposure to some informal education initiatives, mostly obtained to guide personal care or care of a first-degree relative with asthma. Our study revealed some misconceptions and knowledge gaps about the symptoms, triggers and management of asthma. Many teachers indicated that they thought asthma is a curable condition. This assumption could influence the type of advice teachers give to parents and so may further propagate the misconception, as teachers are respected opinion leaders in their communities. As they provide advice to parents, teachers with poor knowledge about asthma may provide wrong advice about seeking help, which may negatively affect the health of a student. It is therefore important that knowledge gaps with regard to treatment options be addressed through effective educational intervention. Some teachers indicated that they consider poor academic performance of students with asthma to be a result of asthmatic students inherently having a low intelligent quotient; this is not supported by any evidence. The poor scholastic performance may be due to frequent absenteeism. Good asthma control can be achieved if appropriate support is available at school to deal with episodes and create an asthma-friendly school environment. This will reduce absenteeism and promote learning. Questions about potential triggers of asthma revealed variable knowledge. Many respondents recognised exposure to smoke as a trigger, but not that some over-the-counter analgesics may also trigger
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an attack. This may lead to an unnecessary asthma attack if a student were to receive an analgesic for pain relief at school. Several teachers also did not show satisfactory knowledge about exercise-induced asthma. This is similar to the findings of Aqeel et al.[17] but different from those of Hussey et al.,[18] who reported about 80% of their teachers knowing about exercise-induced asthma. Asthmatic students may subsequently be excluded from sporting activities, which may, in turn, promote an inactive lifestyle and unsatisfactory management of the condition. Only half of the respondents in our study could recognise symptoms of an asthma attack. This is different from the findings of Aqeel et al.,[17] who reported that about 80% of their respondents showed good knowledge about the symptoms of an asthma attack. The high level of knowledge seen in that study was attributed to teachers having access to diverse information sources about asthma. We could not find evidence that teachers had access to additional information about asthma other than what was provided in the case of a personal history of asthma or if an asthmatic relative had to be cared for. We also found poor knowledge among respondents about other conditions that may coexist with asthma. Training teachers to recognise when medical assessment would be needed may prove valuable, particularly in resource-poor settings where only limited healthcare professionals are available. Although about 80% of the respondents in our study knew that an inhaler could be used to relieve the symptoms of asthma, we did not explore whether the teachers had experience in using an inhaler.
Conclusion
Owing to the poor knowledge of teachers about asthma and the lack of appropriate facilities, schools in Lagos will unlikely be able to offer satisfactory support to students with asthma. It is recommended that teachers be skilled appropriately through training initiatives to address the situation and that school clinics be equipped with basic emergency medication to deal with an asthma episode. Acknowledgements. We acknowledge the support and cooperation of the permanent secretary of the Lagos State Ministry of Education, the tutor generals and the school principals. Author contributions. All authors participated in drafting the questionnaire, analysis of results and writing the manuscript. Funding. None. Conflicts of interest. None. 1. World Health Organization. Asthma. http://www.who.int/news-room/fact-sheets/ detail/asthma (accessed 01 June 2018). 2. International Study of Asthma and Allergies in Childhood (ISAAC) Steering Committee. Worldwide variation in prevalence of symptoms of asthma, allergic rhinoconjunctivitis, and atopic eczema. Lancet 1998;351(9111):1225-1232. https:// doi.org/10.1016/s0140-6736(97)07302-9. 3. Mallol J, Crane J, Von Mutius E, Odhiambo J, Keil U, Stewart A. The International Study of Asthma and Allergies in Childhood (ISAAC) Phase Three: A global synthesis. Allergol Immunopathol 2013;41(2):73-85. https://doi.org/10.1016/j.aller.2012.03.001. 4. Lagos State Ministry of Education. 2011 and 2012 State of Education Reports in Lagos State. http://www.lasgmoed.com/wp-content/uploads/2014/02/2011-12-SoER-Final6-1-corrected-final_PF-2014.pdf (accessed 26 July 2016). 5. Healthy Schools Campaign. Five health-related causes of chronic absenteeism. https:// healthyschoolscampaign.org/education/five-health-related-causes-of-chronicabsenteeism (accessed 01 June 2018). 6. Greiling AK, Boss LP, Wheeler LS. A preliminary investigation of asthma mortality in schools. J Sch Health 2005;75(8):286-290. https://doi.org/10.1111/j.1746-1561.2005. tb07345.x
RESEARCH 7. Unikel LH, Evans D, Bornstein L, Surrence K, Mellins RB. Asthma knowledge and asthma management behavior in urban elementary school teachers. J Asthma 2010;47 (2):185-191. https://doi.org/10.3109/02770900903519908 8. Getch YQ, Neuharth-Pritchett S. Teacher characteristics and knowledge of asthma. Public Health Nurs 2009;26(2):124-133. https://doi.org/10.1111/j.15251446.2009.00763.x 9. Szczepanski R, Brockmann G, Friede G. [Dealing with asthma for teachers – needs and possibilities]. Pneumologie 2001;55(11):512-519. https://doi. org/10.1055/s-2001-18500 10. Henry RL, Gibson PG, Vimpani GV, Francis JL, Hazell J. Randomized controlled trial of a teacher-led asthma education program. Pediatr Pulmonol 2004;38(6):434-442. https://doi.org/10.1002/ppul.20095 11. Dean AG, Sullivan KM, Soe MM. OpenEpi: Open Source Epidemiologic Statistics for Public Health. Version Version 2.2.1. www.OpenEpi.com (accessed 05 May 2017). 12. Pitstick C. Asthma knowledge among primary and secondary school teachers in rural northern Costa Rica. UNED Research J 2014;7(1):25-32. https://doi.org/10.22458/ urj.v7i1.858 13. Govender D, Gray A. Knowledge of primary school teachers about asthma: A cross-sectional survey in the Umdoni sub-district, KwaZulu-Natal. S Afr Fam Pract 2012;54(4):347-351. https://doi.org/10.1080/20786204.2012.10874247
14. Brookes J, Jones K. Schoolteachers’ perceptions and knowledge of asthma in primary schoolchildren. Br J Gen Pract 1992;42(365):504-507. 15. Alnasir FAL. Bahraini school teachers’ knowledge of asthma. Middle East J Fam Med 2004;2(2) 16. Varela AL, Esteban SR, Díaz SP, et al. Knowledge of asthma in school teachers in nine Spanish cities. Pediatr Pulmonol 2016;51(7):678-687. https://doi.org/10.1002/ ppul.23363 17. Aqeel T, Akbar N, Dhingra S, Haq N-U. Assessment of knowledge and awareness regarding asthma among school teachers in urban area of Quetta, Pakistan. J Pharm Pract Comm Med 2015;1(1):18-23. https://doi.org/10.5530/jppcm.2015.1.5 18. Hussey J, Cahill A, Henry D, King AM, Gormley J. National school teachers’ knowledge of asthma and its management. Ir J Med Sci 1999;168(3):174-179. https:// doi.org/10.1007/bf02945848.
Accepted 9 January 2018.
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The evaluation and surgical management of post-intubation tracheal strictures at a thoracic surgery referral centre in South Africa S Ramghulam,1 MB ChB, MMed, FC Cardio; R Perumal,2,3 MB ChB, MPH, MMed, FCP; D Reddy,1 MB ChB, MMed, FC Cardio Department of Cardiothoracic Surgery, School of Clinical Medicine, Nelson R Mandela School of Medicine, University of KwaZulu-Natal, Durban, South Africa Centre for the AIDS Programme of Research in South Africa, Nelson R Mandela School of Medicine, University of KwaZulu-Natal, Durban, South Africa 3 Department of Pulmonology and Critical Care, University of Cape Town, South Africa 1 2
Corresponding author: R Perumal (rubeshanperumal@gmail.com)
Background. The surgical treatment of tracheal stenosis following endotracheal intubation or tracheostomy is well described in the developed world. Objectives. To describe our surgical experience with this pathology, and highlight the nuances of its diagnosis and management in South Africa. Methods. We reviewed the clinical records and archived imaging of all patients who underwent tracheal resection and reconstruction for post-intubation tracheal stenosis between 1 July 2003 and 31 July 2014 in the Department of Cardiothoracic Surgery at Inkosi Albert Luthuli Central Hospital, Durban, South Africa. Results. During the study period, 42 patients underwent tracheal resection. We evaluated the preoperative bronchoscopic characteristics of the tracheal stricture in all patients, and computed tomography (CT) was used as an adjunct in 28 (66%) patients. The stricture lengths determined by CT and intraoperative measurement were strongly correlated (r (27)=0.506, p=0.006), and the stricture lengths determined by bronchoscopy and intraoperative measurement were weakly correlated (r (41)=0.201, p=0.209). A total of 36 patients (85.7%) underwent surgery via a cervical approach and 6 (14.3%) via a right thoracotomy approach. There was no early mortality, and surgery was complicated by vocal cord palsy in 4 cases, restenosis in 2 cases, infection in 1 case and paraparesis in 1 case. Conclusions. Tracheal resection for the treatment of post-intubation tracheal stenosis can be undertaken safely with minimal complications in the developing world, with the vast majority of lesions approached via a cervical approach. A preoperative evaluation of the stricture using CT is an accurate technique for planning tracheal resection and reconstruction. Afr J Thoracic Crit Care Med 2018;24(2):82-86. DOI:10.7196/SARJ.2018.v24i2.193
Despite progressive advances in critical care medicine with regard to invasive ventilation, tracheal strictures that follow endotracheal intubation or tracheostomy remain the most common nonmalignant indication for tracheal resection and reconstruction worldwide.[1,2] In 1996, Hermes Grillo stated that â&#x20AC;&#x2DC;any patient who has received ventilatory support in the recent past or even not-so-recent past, who develops signs and symptoms of upper airway obstruction, has an organic lesion until proved otherwise.â&#x20AC;&#x2122;[1] Despite the use of high-volume low-pressure endotracheal tube cuffs, the reported incidence of post-intubation tracheal strictures is estimated to be up to 11% in critically ill ventilated patients, and associated risk factors include systemic hypotension, long duration of ventilation and tracheostomy.[2,3] It has also been suggested that tracheal stenosis following percutaneous tracheostomy occurs earlier than with surgical tracheostomy.[4] The progression of symptoms of stridor in patients with an evolving tracheal stricture is variable, and often leads to misdiagnosis as asthma until the stridor is evidently not responsive to bronchodilator therapy, and the tracheal lumen significantly reduced to approximately 30% of the normal diameter.[5] Spirometry generally illustrates the characteristic flow-volume loop appearance of fixed obstruction to maximal inspiration and expiration, whether intrathoracic or extrathoracic.[6]
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Diagnostic investigations include plain chest radiography and computed tomography (CT) scanning of the tracheal air column, while bronchoscopy serves as the ideal evaluation of the trachea, as well as the glottis and vocal cords. Rigid bronchoscopy may also play a therapeutic role in dilating the area of stenosis, to temporarily relieve symptoms of stridor prior to definitive surgical management. When indicated, the surgical approach to a post-intubation tracheal stricture is based on the level and complexity of the lesion. The most common scenario encountered is an end-to-end resection with anastomosis, undertaken using a cervical approach, usually producing satisfactory results. Despite the high incidence of post-intubation tracheal stenosis referred to tertiary thoracic surgery centres for tracheal resection, there have been no reports of the South African (SA) surgical experience with this pathology. All tracheal resections at our centre are undertaken by thoracic surgeons, as we are equally comfortable with both cervical and intrathoracic surgery, and tailor our approach to the patientâ&#x20AC;&#x2122;s pathology. This contribution highlights the nuances of diagnosing and treating this pathology in the context of a cardiothoracic surgical referral centre in SA.
Methods
The Department of Cardiothoracic Surgery at Inkosi Albert Luthuli Central Hospital is the sole provider of cardiothoracic surgical care
RESEARCH for the province of KwaZulu-Natal and the eastern seaboard of SA, encompassing a referral base of approximately 14 million patients. We undertook a retrospective cohort study of all patients with postintubation tracheal stenosis who underwent tracheal resection and reconstruction during the 11-year period from 1 July 2003 to 31 June 2014. The data were collected from electronic case records and perioperative imaging studies, including intraoperative photographic images. The study excluded patients with post-intubation tracheal stenosis treated by definitive endoscopic methods or surgical procedures other than tracheal resection and reconstruction. All patients referred with suspected post-intubation tracheal stenosis underwent plain film chest radiography to exclude extrinsic airway compression and to evaluate the lung fields; rigid bronchoscopy was undertaken for diagnostic and therapeutic purposes. Computer tomography has been performed routinely in all patients since 2008, and prior to this, when clinically indicated. At bronchoscopy the vocal cords and the lesion were assessed, and the dimensions and nature of the tracheal stricture were documented to plan future surgical treatment. In patients with significant airflow obstruction, the tracheal stricture was progressively dilated using a series of paediatric and adult rigid bronchoscopes. This approach served to relieve symptoms and to allow time for further imaging modalities and the healing of associated injuries, and to assist with planning definitive surgical treatment on an elective basis. All patients with cicatricial concentric tracheal strictures were considered for definitive treatment by tracheal resection and end-to-end reconstruction. Associated comorbidities, including HIV infection, were managed by appropriate medical therapy. As a general principle, tracheal surgical intervention is viewed as the final planned operation in all patients, and this occasionally requires weeks to months of preparing patients with healing injuries such as burns or skin grafting, or patients requiring other surgical procedures in the interim, such as orthopaedic treatment of fractures. The surgical approach to each patient was individualised based on the endoscopic data derived from rigid bronchoscopy, and CT was used to assist in surgical decision-making (Fig. 1). All patients underwent tracheal stricture resection and end-to-end reconstruction, either via a cervical approach (Figs 2 and 3) or via a thoracic approach (Figs 4 and 5), using techniques of tracheal reconstruction. Median sternotomy and cardiopulmonary bypass were not employed in any patient, and the use of either a suprahyoid release procedure or a hilar release procedure was at the discretion of the surgeon, to ensure a tension-free anastomosis. All patients were maintained in neck flexion for at least 10 days after surgery, assisted by the use of a customised neck flexion harness and a chin-to-manubrium suture (guardian suture). Repeat bronchoscopy was not undertaken unless recurrence of symptoms necessitated intervention, and following hospital discharge, early outpatient follow-up was undertaken at 6 weeks and 6 months, and annually thereafter. Data analysis was undertaken using SPSS software version 23.0 (IBM Corp., USA). For all statistical comparisons, a significance level of p<0.05 was used; correspondingly, 95% confidence intervals (CIs) were used to describe effect size. All data were assessed for normality, and nonparametric tests were used where necessary. Medians and interquartile ranges (IQRs) were used to describe data not amenable to parametric description. Pearson product-moment correlation coefficients were used to describe linear correlation between variables of interest.
Fig. 1. The two modalities used to evaluate post-intubation tracheal stenosis. A. The endoscopic appearance of a complex fibrous tracheal stricture that followed prolonged endotracheal intubation and tracheostomy. B. The reconstructed tracheal image from a CT scan, illustrating the length and location of the lesion in relation to the larynx and the carina. (CT = computed tomography.)
Fig. 2. A. The postero-anterior plain chest radiograph and the coronal section of a CT scan. B demonstrating a cervical post-intubation tracheal stricture distorting the tracheal air column. This lesion would be amenable to surgical treatment via the cervical approach. (CT = computed tomography.)
Fig. 3. The intraoperative images for the patient depicted in Fig. 2. A. With the neck extended, a cervical collar incision is made and subplatysmal flaps are created. B. The 2 cm concentric fibrous stricture in the cervical trachea is exposed, with circumferential dissection restricted to the region of the stricture. C. The stricture is excised and the proximal and distal trachea are extensively mobilised, preserving the lateral vascular pedicles. D. Our preferred technique of cross-field ventilation while placing the interrupted, circumferential absorbable sutures prior to approximating the proximal and distal trachea. E. The reconstituted trachea with the neck in a flexed position to maintain a tension-free anastomosis.
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Fig. 4. A. The postero-anterior plain chest radiograph, and B, the coronal section of a CT scan of a patient with an intrathoracic post-intubation tracheal stricture, which was resected via thoracotomy. (CT = computed tomography.)
Table 1. Preoperative patient characteristics Characteristic (N=42) Age, mean (SD), years 22 (9.94) Male, n (%) 29 (69) Female, n (%) 13 (31) Haemoglobin, mean (SD), g/L 13.08 (1.41) Albumin, mean (SD), g/L 42.6 (5.78) Reason for intubation: non-airway 42 (100) trauma, n (%) Duration of ventilation, median 12 (8 - 21) (IQR), days Duration to definitive surgery, 49.5 (19.5 - 79.5) median (IQR), days Dilations before surgery, median 5.5 (2.75 - 9.00) (IQR), days SD = standard deviation; IQR = interquartile range.
Table 2. Tracheal stenosis lesion characteristics Characteristic Median (IQR), mm Bronchoscopy (n=42) Distance to vocal cords 40 (30 - 50) Stricture length 20 (20 - 25) Distance to carina 50 (40 - 60) Computed tomography (n=28) Stricture length 20 (14 - 20) Intraoperative evaluation (n=42) Stricture length 20 (20 - 25) IQR = interquartile range.
Fig. 5. The intraoperative images of the patient depicted in Fig. 4. A. Via a right posterolateral thoracotomy, the azygous vein is divided and the mediastinal pleura opened, allowing access to the entire length of the intrathoracic trachea. B. Following resection of the stenotic lesion, cross field ventilation of the left lung is achieved while interrupted absorbable sutures are placed circumferentially. C and D. The significant gap between the ends of the trachea is anastomosed in a tension-free fashion using hilar release techniques if required. E and F. The excised tracheal stricture with typical cicatricial features. The study was conducted under the oversight of the Biomedical Research Ethics Committee at the Nelson R Mandela School of Medicine (ref. no. BE468/14).
Results
A total of 42 patients with tracheal stenosis were included in this study (Table 1), with a median follow-up of 114 (IQR 43 - 210) days. Twentynine patients were male (69%), and the mean (standard deviation; 95% CI) age was 22 (9.94) years. Significant preoperative laboratory
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Table 3. Operative technique and outcomes Characteristic (N=42) Surgical approach Cervical Thoracic Suprahyoid release Guardian stitch, n Duration to discharge, median (IQR), days Complications Vocal cord paralysis Restenosis Infection Paraparesis
n (%) 36 (85.7) 6 (14.3) 2 (4.8) 42 (100) 10.5 (10 - 14) 4 (9.5) 2 (4.8) 1 (2.4) 1 (2.4)
IQR = interquartile range.
parameters reflected a mean haemoglobin of 13.08 (1.41) g/L and a mean serum albumin of 42.6 (5.78) g/L. The primary indication for intubation and ventilation in all 42 patients was trauma unrelated to the airway. The median duration of ventilation was 12 days (IQR 8 - 21), and the median time from presentation to definitive surgery was 49.5 days (IQR 19.5 - 79.5). Prior to tracheal resection, the median number of therapeutic tracheal dilations undertaken using rigid bronchoscopy was 5.5 dilations (IQR 2.75 - 9) per patient.
RESEARCH The tracheal stricture characteristics on bronchoscopy demonstrated a median (IQR) distance to the vocal cords of 40 (30 - 50) mm, median (IQR) stricture length of 20 (20 - 25) mm and median (IQR) distance to carina of 50 (40 - 60) mm (Table 2). The median (IQR) proportion of tracheal involvement was 18.2% (15.7 - 21.4). Computed tomography was performed in 28 cases, with the lesion characteristics determined by CT demonstrating a median (IQR) stricture length of 20 (14.25Â -Â 25), and the median stricture length determined at surgery was 20 (20 - 25) mm. The stricture lengths determined by CT and intraoperative measurement were strongly correlated (r (27)=0.506; p=0.006). The stricture lengths determined by CT and bronchoscopy were moderately correlated (r (27)=0.472; p=0.01), and the stricture lengths determined by bronchoscopy and intraoperative measurement were weakly correlated (r (41)=0.201; p=0.209). Upper tracheal lesions were resected via a cervical approach in 36 (85.7%) patients, with the remaining 6 (14.3%) with lower tracheal lesions undergoing right thoracotomy. In 2 cases a suprahyoid release was employed to ensure a tension-free anastomosis (Table 3). The median (IQR) period to discharge from definitive surgery was 10.5 (10 - 14) days. There was no perioperative mortality, and morbidity included vocal cord palsy in 4 patients, restenosis in 2 patients, infection in 1 patient and transient paraparesis in one patient.
Discussion
We have described a cohort of patients with post-intubation tracheal stenosis treated by tracheal resection, the majority of whom were male (69%) and young (mean age 22 years), where the primary indication for intubation was predominantly trauma unrelated to the airway. The median duration of ventilation of 12 days is consistent with the need for adjuvant surgical procedures, including relook laparotomies in patients with an open abdomen, and recovery following head injuries. Plain chest radiography remains the primary imaging modality for patients with suspected post-intubation tracheal strictures, often to examine the lung fields, as impaired clearing of secretions may result in bronchopneumonia. Obscuration of the tracheal air column may be noted, but the absence of this feature is not helpful. Following plain radiographic evaluation, rigid bronchoscopy is a key component in the primary diagnostic and therapeutic management of patients with post-intubation tracheal strictures. Although bronchoscopy remains the gold standard for the identification and characterisation of airway lesions, it fails to provide adequate visualisation of the post-stenotic trachea in cases of severe luminal narrowing. In addition, bronchoscopy is an invasive procedure that may cause patient discomfort, requires sedation and is associated with a 0.8% morbidity.[8] Of critical value is the therapeutic role of rigid bronchoscopy in dilating severely stenotic lesions, as a resuscitative manoeuvre prior to intubation, or as a temporising measure while surgery is being planned. In rare instances, tracheal dilatation of an early stricture may suffice as definitive therapy, without the need for late intervention. However, limited direct control of bleeding distal to the lesion is possible, following diagnostic or interventional bronchoscopy. In this study, patients required a median of 5.5 serial tracheal dilatations prior to definitive surgical therapy, with surgery only undertaken once all associated injuries and open wounds were healed. In addition to assessing vocal cord function and obtaining bronchoalveolar lavage
specimens, detailed evaluation of the tracheal stricture is made, including the nature (fibrous v. granulation tissue), the configuration (concentric v. eccentric) and the diameter, length and location of the stricture/s. Despite the widespread use of bronchoscopy as a tool to determine stricture length, we found that the overall correlation with findings at surgery were poor. This may be related to operator/observer variability, or the fact that strictures may evolve with repeated dilatation, resulting in the external scarring noted at surgery being inconsistent with the length of the intraluminal lesion measured at bronchoscopy.[8] CT was performed to improve the accuracy of our evaluation of tracheal stricture length and location in patients with tracheal strictures necessitating surgical intervention, with a specific interest in evaluating the airway using virtual bronchoscopy airway reconstruction techniques.[9] CT is a non-invasive procedure and well tolerated by all patients. Taha et al.[10] reported that the detection rate of post-intubation tracheal stenosis was 94% by CT, and 88% by rigid bronchoscopy, and CT provided additional information on the morphology of the tracheal wall. Tracheostomy tubes were removed prior to chest CT, thereby avoiding radiographic artifacts, and high-resolution imaging of the lung fields allowed exclusion of any chronic pulmonary sequelae in patients with a prolonged ventilatory course. The non-invasive nature of this imaging modality made it complementary to rigid bronchoscopy, and a strong correlation between the evaluation of tracheal stricture length on CT and findings at surgery was found.[11] The combined use of CT and endoscopic evaluation has allowed for extremely accurate preoperative planning, including the surgical approach, the need for adjunctive release manoeuvres and the prediction of postoperative complications. The definitive surgical treatment of short segment tracheal strictures is resection with end-to-end anastomosis.[12] While an approach involving primary emergency tracheal resection upon diagnosis may be favoured by some centres,[13] it is widely held that tracheal reconstruction should be the final act in foreseeable tracheal instrumentation. The median duration from presentation to definitive surgery of approximately 7 weeks in this study generally reflected the need for optimisation of patients prior to definitive surgery. For example, one patient with extensive burns to the chest and face underwent multiple reconstructive surgeries during his 20-week admission prior to tracheal resection, during which time his stridor was palliated by weekly dilatation of the tracheal stricture with rigid bronchoscopy. Similarly, patients with open wounds underwent skin grafting prior to definitive tracheal surgery. The core principles of tracheal resection and reconstruction are well established, and the surgical technique was consistent across all surgeons in this study.[14,15] The vast majority of patients presented with tracheal strictures in the upper trachea, as seen at bronchoscopy and confirmed on CT scan, and therefore underwent tracheal resection and end-to-end reconstruction via a cervical collar incision. In six patients the stricture was situated in the lower trachea, necessitating an approach via right posterolateral thoracotomy. Following tracheal dilatation via rigid bronchoscopy, oral intubation is performed with a single lumen reinforced endotracheal tube, with cross-field ventilation maintained by the surgeon after division of the trachea. The tracheal anastomosis is undertaken with interrupted 3-0 Vicryl
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RESEARCH sutures (Ethicon Inc., USA). A tension-free anastomosis is ensured by maintaining the patient’s neck in a flexed position, aided by a suture from chin to manubrium (guardian stitch). In a minority of patients, additional release manoeuvres are necessary, the most common being a suprahyoid release, as described by Montgomery.[16] All patients are extubated after the procedure and remain in a neck flexion position for 10 days, and are discharged after release of the guardian suture. The complications encountered after tracheal resection are well described, with four patients in this study having experienced varying degrees of laryngeal dysfunction, with unilateral vocal cord palsy. Both the patients who underwent suprahyoid release complicated with vocal cord palsy, which is known to be a risk of this technique.[16] Two patients presented with restenosis after tracheal resection. In one patient, the cause was granulation tissue at the anastomosis, and this was dealt with by rigid bronchoscopy, with no subsequent recurrence. The second patient had undergone long-segment tracheal resection following tracheostomy-site stenosis, and after a second attempt at tracheal resection, she was ultimately managed with Montgomery T-tube insertion. Previous tracheostomy, long-segment tracheal resection and prior tracheal resection are documented risk factors for surgical failure and postoperative complications.[17-19] Montgomery T-tube placement affords normal airway humidification and speech, and less damage to the stomal area than with tracheostomy.[20] An alternative is the use of a fenestrated tracheostomy tube, which has similar benefits.[21] In exceptional circumstances, tracheal stents may be considered to manage complex lesions, but the associated complications of stent migration and mucosal overgranulation limit their effectiveness.[22,23] Other complications encountered included superficial cervical wound infection, and transient paraplegia probably related to hyperflexion of the neck in the postoperative period.[24,25] The outcomes of tracheal resection for post-intubation tracheal stenosis in this study are consistent with those of other published series.[26,27] Ventilation for trauma in young patients remains a significant cause of post-intubation tracheal stenosis, and such patients often require adjuvant surgical procedures prior to definitive airway surgery. This study suggests that CT scan is an accurate preoperative investigation method to estimate the length of the segment to be excised, although rigid bronchoscopy remains a cornerstone of early management. A combination of endoscopic and CT imaging allows the surgical approach to be planned, but ultimately the intraoperative findings upon transection of the trachea dictate the technique of reconstruction and the potential need for adjunctive release procedures. Utilising standard principles of tracheal resection and end-to-end reconstruction as described by others, successful outcomes were achieved in 95.2% of patients undergoing surgery. Acknowledgements. The authors thank the medical staff and administration at Inkosi Albert Luthuli Central Hospital for their participation, and for permission to undertake this study. Author contributions. SR and DR conceptualised the study. SR performed the data capture. RP performed the data analysis. All authors contributed equally to writing the manuscript. Funding. None. Conflicts of interest. None. 1. Grillo HC, Donahue DM. Post-intubation tracheal stenosis. Semin Thorac Cardiovasc Surg 1996;8(4):370-380.
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2. Stauffer JL, Olson DE, Petty TL. Complications and consequences of tracheal intubation and tracheostomy: A prospective study of 150 critically ill adult patients. Am J Med 981;70(1):65-76. https://doi.org/10.1016/0002-9343(81)90413-7 3. Mathias DB, Wedley JR. The effects of cuffed endotracheal tubes on the tracheal wall. Br J Anaesth. 1974;46(11):849-852. 4. Raghuraman G, Rajan S, Marzouk JK, Mullhi D, Smith FG. Is tracheal stenosis caused by percutaneous tracheostomy different from that by surgical tracheostomy? Chest 2005;127(3):879-885. https://doi.org/10.1378/chest.127.3.879 5. Spittle N, McCluskey A. Tracheal stenosis after intubation. BMJ 2000;321(7267):10001002. https://doi.org/10.1136/bmj.321.7267.1000 6. Pinsonneault C, Fortier J, Donati F. Tracheal resection and reconstruction. Can J Anesth 1999;46(5):439-455. https://doi.org/10.1007/BF03012943 7. Grobbelaar J, Daniller T. Tracheal resection for laryngotracheal stenosis: A retrospective analysis. S Afr J Surg 2014;52(3):79-81. https://doi.org/10.7196/sajs.1409 8. Herth F, Becker HD, LoCicero J, Ernst A. Endobronchial ultrasound in therapeutic bronchoscopy. Eur Respir J 2002;20(1):118-121. https://doi.org/10.1183/09031936.0 2.01642001 9. Koletsis EN, Kalogeropoulou C, Prodromaki E, et al. Tumoral and non-tumoral trachea stenoses: A valuation with three-dimensional CT and virtual bronchoscopy. J Cardiothorac Surg 2007;2:18. https://doi.org/10.1186/1749-8090-2-18 10. Taha MS, Mostafa BE. Spiral CT virtual bronchoscopy with multiplanar reformatting in the evaluation of post-intubation tracheal stenosis. Eur Arch Otorhinolaryngol 2009;266(6):863-966. https://doi.org/10.1007/s00405-008-0854-y 11. Morshed K, Trojanowska A, Szymanski M, et al. Evaluation of tracheal stenosis: Comparison between computed tomography virtual tracheobronchoscopy with multiplayer reformatting, flexible tracheofiberoscopy and intraoperative findings. Eur Arch Otorhinolaryngol 2011;268(4):591-597. https://doi.org/10.1007/s00405-010-1380-2 12. Grillo HC, Donahue DM, Mathisen DJ, Wain JC, Wright CD. Postintubation tracheal stenosis. Treatment and results. J Thorac Cardiovasc Surg 1995;109(3):486-492. https://doi.org/10.1016/S0022-5223(95)70279-2 13. Cordos Ioan, Bolca C, Paleru C, Posea R, Stoica R. Sixty tracheal resections – single centre experience. Interact Cardiovasc Thorac Surg 2009;8:63-66. https://doi. org/10.1510/icvts.2008.184747 14. Grillo HC. Development of tracheal surgery: A historical review. Part 1: Techniques of tracheal surgery. Ann Thorac Surg 2003;75(2):610-619. http://doi.org/10.1016/ S0003-4975(02)04108-5 15. Grillo HC. Development of tracheal surgery: A historical review. Part 2: Treatment of tracheal diseases. Ann Thorac Surg 2003;75(3):1039-1047. https://doi.org/10.1016/ S0003-4975(02)04109-7 16. Montgomery WW. Suprahyoid release for tracheal anastomosis. Arch Otolaryngol 1974;99(4):255-260. https://doi.org/10.1001/archotol.1974.0078003026500517 Abbasidezfouli A, Shadmehr MB, Arab M, et al. Postintubation multisegmental tracheal stenosis: Treatment and results. Ann Thorac Surg 2007;84(1):211-215. https:// doi.org/10.1016/j.athoracsur.2007.03.050 17. Wright CD, Grillo HC, Wain JC, et al. Anastomotic complications after tracheal resection: Prognostic factors and management. J Thorac Cardiovasc Surg 2004;128(5):731-739. http://doi.org/10.1016/j.jtcvs.2004.07.005 18. Bibas BJ, Terra RM, Oliveira Junior AL, et al. Predictors for postoperative complications after tracheal resection. Ann Thorac Surg 2014;98(1):277-282. http:// doi.org/10.1016/j.athoracsur.2014.03.019 19. Saghebi SR, Zangi M, Tajali T, et al. The role of T-tubes in the management of airway stenosis. Eur J Cardiothorac Surg 2013;43(5):934-939. https://doi.org/10.1093/ejcts/ezs514 20. Hess DR, Altobelli NP. Tracheostomy tubes. Respir Care 2014;59(6):956-973. https:// doi.org/10.4187/respcare.02920 21. Tsakiridis K, Darwiche K, Visouli AN, et al. Management of complex benign posttracheostomy tracheal stenosis with bronchoscopic insertion of silicon tracheal stents, in patients with failed or contraindicated surgical reconstruction of trachea. J Thorac Dis 2012;4(S1):S32-S40. https://doi.org/10.3978/j.issn.2072-1439.2012.s002 22. Chen G, Wang Z, Liang X, et al. Treatment of cuff-related tracheal stenosis with a fully covered retrievable expandable metallic stent. Clin Radiol 2013;68(4):358-364. https:// doi.org/10.1016/j.crad.2012.08.022 23. Pitz CC, Duurkens VA, Goossens DJ, Knaepen PJ, Siegers P, Hoogenboom LJ. Tetraplegia after a tracheal resection procedure. Chest 1994;106(4):1264-1265. http:// doi.org/10.18203/issn.2454-5929.ijohns20181877 24. Dominguez J, Rivas JJ, Lobato RD, Diaz V, Larru E. Irreversible tetraplegia after tracheal resection. Ann Thorac Surg 1996;62(1):278-280. https://doi.org/10.1016/00034975(96)00246-9 25. Sharpe DA, Moghissi K. Tracheal resection and reconstruction: A review of 82 patients. Eur J Cardiothorac Surg 1996;10(12):1040-1046. 26. Negm H, Mosleh M, Fathy H. Circumferential tracheal resection with primary anastomosis for post-intubation tracheal stenosis: Study of 24 cases. Eur Arch Otorhinolaryngol 2013;270(10):2709-2717. https://doi.org/10.1007/s00405-0132367-6 Accepted 26 February 2018.
RESEARCH
Thoracic endometriosis syndrome at University of Ilorin Teaching Hospital P O Adeoye,1 MBBS, FWACS, Cert CTS; A S Adeniran,2 MBBS, FWACS, FMCOG; K T Adesina,2 MBBS, FWACS, FMCOG DMAS, O A Ige,3 MBBS, FWACS; O R Akanbi,4 MB ChB; A Imhoagene,5 MBBS; O O K Ibrahim,6 MBBS, FMC (Path); G G Ezeoke,2 MBBS, FWACS Division of Thoracic and Cardiovascular Surgery, Department of Surgery, College of Health Sciences, University of Ilorin, Nigeria Department of Obstetrics and Gynaecology, College of Health Sciences, University of Ilorin, Nigeria 3 Department of Anaesthesia, College of Health Sciences, University of Ilorin, Nigeria 4 Department of Surgery, University of Ilorin Teaching Hospital, Nigeria 5 Department of Obstetrics and Gynaecology, University of Ilorin Teaching Hospital, Nigeria 6 Department of Morbid Anatomy, College of Health Sciences, University of Ilorin, Nigeria 1 2
Corresponding author: P O Adeoye (poadeoye@yahoo.ca)
Background. Endometriosis is defined as the presence of endometrial tissue (stroma and functional glands) outside the uterine cavity in women of reproductive age. Ectopic sites are frequently located in the pelvis; extrapelvic sites have been reported in the gastrointestinal tract and thoracic cavity. Thoracic manifestation of endometriosis constitutes thoracic endometriosis syndrome (TES). Objectives. To examine the presentation pattern and outcome of in the management of TES. Methods. This study is a retrospective review of medical records of patients diagnosed with endometriosis at the University of Ilorin Teaching Hospital over a 3.5-year period from January 2014 to June 2017. Results. A total of 21 patients presented with endometriosis, of whom 8 (38.1%) presented with TES. The most common variety of TES was catamenial pleural effusion (CPE) accounting for 75%, followed by catamenial chest pain (37.5%). Two patients (25%) each presented with catamenial pneumothorax and catamenial haemoptysis, while 1 (12.5%) had catamenial surgical emphysema. Closed thoracostomy tube drainage plus chemical pleurodesis was the most frequent intervention technique, accounting for 62.5%. Conclusion. TES remains an uncommon entity, despite being the most common extrapelvic manifestation of endometriosis. CPE appeared to be the most common variant of TES in our environment. Currently available treatment options need to be improved, and more use made of video-assisted thoracoscopic surgery. Afr J Thoracic Crit Care Med 2018;24(2):87-91. DOI:10.7196/SARJ.2018.v24i2.201
Endometriosis is defined as the presence of endometrial tissue (stroma and functional glands) outside the uterine cavity. It is therefore a situation when endometrial tissue is present in ectopic locations instead of its eutopic site in the uterus, a condition first described by Maurer et al.[1] in 1958. It is found among women of reproductive age, with an incidence varying between 5% and 15%, as documented in the literature.[1-5] The majority of ectopic sites are in the pelvic organs, with only 8.9% - 12% reported as extrapelvic in location.[2,3] While the thoracic cavity is one of the more common extrapelvic sites,[6,7] a report documented gastrointestinal endometriosis as the most common site (32.3%). Another site is the urinary tract (5.9%), while together, the lungs, umbilicus, abdominal scars, liver, gall bladder, pancreas, breast and extremities constitute 61.8%.[2] Endometriosis of the central nervous system and the heart has also been documented.[4] Thoracic manifestation in endometriosis is varied, and collectively referred to as thoracic endometriosis syndrome (TES).[3,5,6] Various theories have been propounded to explain this condition.[2-6] The theory of coelomic metaplasia is premised upon the common origin of endometrial and mesothelial cell from coelomic epithelium. Appropriate pathologic stimuli (probably refluxed menstrual blood) then trigger metaplastic change. The retrograde menstruation or
migration theory postulates that diaphragmatic endometrial implants result from shedding of eutopic endometrial tissue through the patent fallopian tube into the pelvis, and thence into the peritoneal fluid. The physiologic hypothesis suggest that high circulating levels of prostaglandin F2 present during menstruation cause vasoconstriction and bronchospasm, which predisposes to alveolar rupture, hence pneumothorax. The transgenital-transdiaphragmatic passage of air theory explains how pneumothorax develops from air movement from the vagina through the fallopian tube into the peritoneum and through congenital or acquired diaphragmatic defects into the pleural cavity. Transgenital movement of air is aided by deficiency of mucus plug during menses. The concept of clockwise peritoneal circulation, starting from the pelvis and upwards through the right paracolic gutter to the right hypochondrium, may facilitate the migration theory. Lastly, microembolisation of endometrial cells into the lungs through venous or lymphatic circulation have been postulated in the metastatic theory. Though none of these theories individually completely explains the TES phenomenon, there may be interplay between the various mechanisms. TES remains an uncommon condition, with the literature mostly documenting case reports or case series, and the manifestation is often varied. Furthermore, there is a paucity of reporting of this
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RESEARCH condition from the African continent. We therefore constituted an endometriosis study group and present a report of this rare condition from North Central Nigeria.
Methods
We conducted a retrospective review of medical records of patients diagnosed with endometriosis at the University of Ilorin Teaching Hospital over a 3.5-year period (January 2014 - June 2017). All patients presented through either the obstetrics and gynaecology or thoracic and cardiovascular surgery department. Identification of cases managed was from the databases of the departments as portals of entry, as well as the hospital medical records database. The index of suspicion used in identifying possible TES patients included chest symptoms and signs related to the menstrual cycle, with or without a background diagnosis of pelvic endometriosis or chronic pelvic pain. The diagnostic approach included a history and physical examination at presentation, review of gynaecological history/gynaecologist review, management of emergency needs plus sample collection, other procedures and biopsy as required and necessary supportive treatments. Also documented were their demographic characteristics, mode of presentation and diagnosis, the treatment offered and their outcomes. Data were collected and descriptive statistics presented using Excel (Microsoft, USA).
Results
We documented a total of 21 patients presenting with endometriosis, representing 1.27% of all gynaecological admissions during the study period. Of these, 8 patients (38.1%) presented with TES. This represented 1.2% of admissions for thoracic disorders during the study period. The characteristics of these patients are presented in Table 1. Twelve patients (57.1%) had extrapelvic presentation, of whom the TES 8 comprised 66.7%; the others were 2 gastrointestinal (GIT) (25%) and 2 umbilical (16.7%) cases (2 patients had combined TES and GIT presentation). All patients had had multiple episodes of symptomatology for TES before presentation and diagnosis at our facility. The age range for TES was 14 - 38 years (median 31, mean 28.7 (standard deviation (SD) 8.63), and 87.5% were nulliparous. The most common variety of TES among the patients was catamenial pleural Table 1. Presentation and intervention in patients with TES Manifestations Patient Age (yrs) Parity Thoracic Other 1 27 0 Rt CCP, CPE Pelvic 2 3
35 35
0 0
Rt CPE, CHp Rt CPE
Pelvic Pelvic, GIT
4
38
2
Rt CPE
Pelvic, GIT
5
35
0
Rt CCP, CPT, CPE
Pelvic
6
27
0
Rt CPE
Pelvic
7 8
14 19
0 0
CSE Rt CCP, CPT, CHp
Pelvic -
effusion (CPE) in six (75%) patients; three (37.5%) had catamenial chest pain (CCP), two (25%) each presented with catamenial pneumothorax (CPT) and catamenial haemoptysis (CHp) while one (12.5%) had catamenial surgical emphysema (CSE). In addition, four (50%) of the patients had multiple thoracic manifestations, while right-sided TES occurred in seven (87.5%). The patient with CSE had bilateral diffuse thoracic, nuchal and facial surgical emphysema. Seven patients (87.5%) had concomitant extrathoracic manifestation, all involving the pelvis, of whom two (28.6%) had an additional GIT manifestation; thus 25% of all patients with TES had GIT manifestation (Table 1). Table 2 shows that diagnosis was based on strong clinical grounds in all patients, establishing catamenial relationship to presentation. In three (37.5%) patients who had a chest tube in situ prior to onset of menstruation, the effluent increased in volume and became haemorrhagic with menstruation. Histological confirmation was obtained in four (50%) cases, while serum CA-125 was performed in three patients, with elevated results found in two of them. Closed thoracostomy tube drainage (CTTD) plus chemical pleurodesis was the most frequent intervention technique, accounting for 62.5% (five patients), while diagnostic video-assisted thoracoscopic surgery (VATS) was performed on two (25%) patients. Another two (25%) had thoracotomy with parietal pleurectomy after failed chemical pleurodesis. Fig. 1 shows endometriotic nodules on the diaphragm at thoracotomy of patient 5 on the list, while Fig. 2 shows the photomicrograph of the histology of the same patient. However, Table 2. Modality of diagnosis of TES Clinical Patient Histological presentation 1 No Yes 2 No Yes 3 Yes Yes 4 Yes Yes 5 Yes Yes 6 Yes Yes 7 No Yes 8 No Yes
Intervention VATS + excision of lung cyst, CTTD + chemical pleurodesis CTTD, home ambulatory system VATS + pleural biopsy, CTTD + chemical pleurodesis CTTD + chemical pleurodesis, laparotomy CTTD + chemical pleurodesis, thoracotomy + pleurectomy CTTD + chemical pleurodesis, thoracotomy + pleurectomy Conservative Conservative
Serum CA-125 No No No No No Yes Yes Yes
Remarks Recurrence, repeat chemical pleurodesis Recurrence, declined further intervention Failed chemical pleurodesis Failed chemical pleurodesis -
TES = thoracic endometriosis syndrome; Rt = right; CCP = catamenial chest pain; CPE = catamenial pleural effusion; CPT = catamenial pneumothorax; CSE = catamenial surgical emphysema; CHp = catamenial haemoptysis; VATS = video-assisted thoracoscopic surgery; CTTD = closed thoracostomy tube drainage; GIT = gastrointestinal tract.
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Fig. 1. Endometriotic nodules on the diaphragm.
Fig. 2. Photomicrograph of histology on endometrioma (haematoxylin and eosin staining, magnification ×400) showing a dilated endometrial gland surrounded by endometrial stroma, including mononuclear inflammatory cells (mostly macrophages). two patients were managed non-operatively. In addition, two patients had recurrent pleural effusion during subsequent menstruation following an initial successful treatment. Both were counselled for thoracotomy for parietal pleurectomy, but one opted for repeat chemical pleurodesis, while the other chose expectant management. No mortality was recorded, and patients were transferred to the gynaecology service for hormonal therapy, apart from two patients who had had hormonal treatment prior to presentation with thoracic manifestations.
Discussion
TES refers to a constellation of manifestations resulting from growth of endometrial-like glands and stroma in the lungs, on the pleural surfaces or in the airway.[3,5-7,9,10] It is an uncommon condition, documented in the literature mostly in case reports and case series.[5-7,9-12] An analysis of 110 case reports/series published in English was conducted by Joseph and Sahn in 1996, [13] and Channabasavaiah and Joseph, [4] in 2010, conducted a similar review with the same number of patients, covering a 6.5-year period. Haga et al.,[14] in Tokyo, Japan, reported 84 cases of CPT
while evaluating 570 cases of spontaneous pneumothorax in women. Over a 7-year period, Hwang et al.[2] documented 15 cases at a single centre in Seoul, Korea. Forty cases were reported by Dvorakovskaya et al. [15] from St Petersburg Hospital, but the duration of collation was not stated. Reports from Africa are even rarer: there is a report of 3 cases by Ekpe et al. [16] from South South Nigeria, and a case report each from Ghana[17] and Uganda. [18] There is one review article each from Nigeria and Zimbabwe. [3,19] TES is a rare but important extrapelvic manifestation of endometriosis. We therefore document these eight cases of TES seen over 3.5 years at a tertiary hospital in North Central Nigeria. Manifestations constituting TES include CCP, CPT, CHt/CPE, catamenial haemoptysis (CHp), pulmonary nodules and CPM.[2-8] These presentations result from the presence of endometrial tissue on the pleural surfaces (CCP, CPT, CPE, CPM), in the lung parenchyma (pulmonary nodule) or the airway (CHp). Pleural involvement is more common, accounting for about 83% of TES, while parenchymal and airway involvement account for 17%,[9] and our study found a similar distribution. An even rarer occurrence of endometriosis on the thoracic aorta has been reported.[20] The term catamenial is derived from the Greek word Katamenios, which means ‘monthly occurrence’. Thus development of these manifestations in a temporal relationship to menstruation is vital to clinical diagnosis. The expected interval reported between symptoms and menstruation varies, but a 72-hour period before onset and after cessation of menstruation is considered acceptable.[13] Our study reflects a higher presentation of extrapelvic lesions, of 57% (12 of 21 patients with endometriosis), compared with the 8.9% - 12% documented in the literature.[2,3] The higher incidence of TES (66.7%) in extrapelvic sites has been reported in some studies;[5,6] however, contrary to reports of GIT dominance by others,[2] in our study this constituted only 25%. The mean age of occurrence of TES in our patients was 28.7 years (SD 8.63); this is younger than the ~35 years reported in the literature.[2-4,8,10,13,21] The presentation of CHp in one of our youngest patients (19 years) may support the postulate that this manifests at a relatively younger age than other forms of manifestation.[4] An interesting finding from this study is the predominance of CPE, accounting for 75% of cases, while CCP was second, with 37.5%. We also identified a rare occurrence of CSE without associated pneumothorax. Most reports present either CPT (up to 73%)[3-7] or CCP (80% - 90%)[3,7,21] as the most common manifestation of TES. In a review of 15 cases by Hwang et al.,[2] CHt accounted for 53%, while the remaining 47% were CPT cases. The 87.5% predominance on the right side in this study is consistent with previous reports of the vast majority of TES (above 85%) occurring in the right hemithorax.[2,3,5,6,10,13,21] This predilection has been attributed to clockwise peritoneal circulation by some authors.[5] Multisite involvement in extragenital endometriosis is rare. However, TES is often associated with pelvic endometriosis with an incidence varying from 18% to 84%.[2,6,13,21,22] In this study, 87.5% had concomitant TES and pelvic endometriosis. Studies have suggested that pelvic endometriosis usually occurs about 5 years earlier than onset of thoracic manifestation.[13] Of the six patients who wished to become pregnant, only one had children, with the remaining 83.3% being infertile. The association
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RESEARCH between endometriosis and infertility has been well documented, with 30% - 50% of patients with endometriosis estimated to be infertile, while about 20% - 50% of infertile women are said to have endometriosis.[23,24] The presence of endometrioma in the pelvis, with resultant adhesions, has been coined the â&#x20AC;&#x2DC;pelvic factorâ&#x20AC;&#x2122;. The distortion that arises causes tubo-ovarian dis-co-ordination, and affects tube patency.[23,24] Other possible mechanisms include endocrine and ovulatory abnormalities, altered peritoneal function and altered endometrial hormonal and cell-mediated function.[24] Diagnostic criteria in the establishment of thoracic endometriosis include clinical and histological factors, and the use of a biomarker. The establishment of cyclical symptoms in temporal relationship with menstruation, as seen in all patients in this study, is pivotal to diagnosis.[6,16,21] However, a confirmatory diagnosis is established when endometrial stroma and glands are identified histologically, as was seen in 50% of our patients.[2,6] Obtaining tissue for histological diagnosis may not always be feasible, and pleural fluid or bronchial lavage cytology is often negative. The identification of endometriotic lesions by VATS or bronchoscopy may be easier during menstruation. Bronchoscopy performed within 2 days of onset of menses may improve localisation, especially in patients with CHp.[3,5] The appearances of lesions on radiological imaging techniques are nonspecific.[2,5,10] Focal areas of consolidation, ill-defined opacities or bullous disease on the lung, or hypo-attenuated areas on the diaphragm seen on chest computed tomography scans are not pathognomonic, and the sensitivity on magnetic resonant imaging may be superior.[2,3,5,10] The biomarker serum CA-125 is now used to improve the diagnosis of endometriosis. However, an elevated serum or pleural fluid level is nonspecific, as it is associated with any process causing irritation of mesothelial cells.[3,10] We have only recently included this in the investigation protocol for our patients, and two of the three who had the test showed an elevated level. Two patients from our study did not require surgical intervention. CSE in patient 7 resolved on intranasal oxygen supplementation while she was nursed in semi-Fowlerâ&#x20AC;&#x2122;s position. CPT in patient 8 was mild and allowed for reabsorption. Of the remaining 6 patients, CTTD was the initial line of treatment in order to relieve raised intrapleural pressure. Two patients (1 and 3) had diagnostic VATS, but since our centre is not equipped for therapeutic VATS, parietal pleurectomy could not be conducted. Patient 2 did not achieve lung re-expansion for chemical pleurodesis and declined thoracotomy for parietal pleurectomy. She had a modified ambulatory home drainage system instituted. The other five patients all had chemical pleurodesis; in one (20%) this was successful at first application, another one (20%) at second application, and two (40%) failed and proceeded to have thoracotomy and parietal pleurectomy. One patient with recurrence declined further intervention. Our observation that chemical pleurodesis alone has a poor success rate in patients with CPE is supported by the literature.[9,10,16] This is expected, as continuous activity from cyclical proliferation of endometrial implants, and also migration through patent diaphragmatic defects, predisposes to recurrence. We recorded no mortality, however, and patients were referred to the gynaecologist for hormonal therapy and further management. Despite individual case requirements, we propose a systematic approach to the management of TES, by a multidisciplinary team,
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consisting of a gynaecologist, cardiothoracic surgeon, pulmonologist, histopathologist, radiologist and anaesthesiologist.[3,11] A high index of suspicion is needed on clinical assessment and radiologic evaluation. Initial supportive oxygen, observation and rest are instituted for small collections. Patients in respiratory distress need immediate relief by either thoracocentesis or tube thoracostomy, with fluid specimens obtained for microscopy, Ziehl Neelsen stain, chemistry, cytology and CA-125 assay. When available, VATS should be employed early. This is currently the gold standard, as both diagnosis and treatment (including resection of implants, closure of diaphragmatic fenestrations and pleurectomy with abrasive pleurodesis) can be effected with the attendant benefit of minimal access. [3,5,11,21] Combined VATS and video-assisted laparoscopy is recommended by some researchers [10] Conventional thoracotomy should be utilised where VATS is unavailable, in cases of recurrence after VATS or failure of the minimally invasive technique. Hormone therapy using gonadotrophin-releasing hormone analogue is recommended in the immediate postoperative period and for 6 - 12 months afterwards.[2,6,10] Two limitations of this study are its retrospective nature, and the small sample size. We have therefore constituted an endometriosis study group to collate prospectively collected data for future presentation.
Conclusion
TES remains an uncommon condition despite being the most common extrapelvic manifestation of endometriosis. Its association with pelvic presentation is further strengthened by this study. There is some variability in the modes of manifestation of TES. Recurrent chest symptoms in a woman of childbearing age with a history of infertility should raise a high index of suspicion. Despite most literature reporting CPT as the most common TES manifestation, we found CPE to account for the majority of cases in our study. We also present a rare manifestation of CSE. Being a developing country with paucity of facilities, the utilisation of VATS as a treatment option is limited in Nigeria. Chemical pleurodesis and conventional thoracotomy with parietal pleurectomy are therefore the most common intervention modes. However, we found that chemical pleurodesis was generally unsuccessful in patients with TES; therefore, performing thoracotomy without attempting chemical pleurodesis may be a more beneficial in absence of VATS. Acknowledgements. None. Author contributions. POA: conceptualisation, literature review, data collection and analysis, manuscript drafting and review, corresponding author; ASA: data collection, manuscript review; KTA: manuscript review; OAI: drafting of abstract, manuscript review; ORA: data collection and analysis, manuscript review; AI: data collection; OOKI: development of micrograph, manuscript review; GGE: manuscript review. Funding. None. Conflicts of interest. None. 1. Maurer ER, Schaal JA, Mendez FL. Chronic recurring spontaneous pneumothorax due to endometriosis of the diaphragm. JAMA 1958;168:2013-2014. https://doi. org/10.1001%2Fjama.1958.63000150008012c 2. Hwang SM, Lee CW, Lee BS, Park JH. Clinical features of thoracic endometriosis: A single center analysis. Obstet Gynecol Sci 2015;58(3):223-231. https://doi.org/10.5468/ ogs.2015.58.3.223
RESEARCH 3. Kesieme EB, Prisadov G, Welcker K, Abubakar U. Thoracic endometriosis syndrome: Current concept in pathophysiology and management. Orient J Med 2016;28(1-2):1-10. 4. Channabasavaiah AD, Joseph JV. Thoracic endometriosis: Revisiting the association between clinical presentation and thoracic pathology based on thoracoscopic findings in 110 patients. Medicine 2010;89(3):183-188. https://doi.org/10.1097%2Fmd.0b013 e3181df67d5 5. Azizad-Pinto P, Clarke D. Thoracic endometriosis syndrome: Case report and review of the literature. Perm J 2014;18(3):61-65. https://doi.org/10.7812%2Ftpp%2F13-154 6. Visouli AN, Zarogoulidis K, Kougioumtzi L, et al. Catamenial pneumothorax. J Thor Dis 2014;6(S4):S448-S480. https://doi.org/ 10.3978/j.issn.2072-1439.2014.08.49 7. Fang HY, Jan CI, Chen CK, Chen WTL. Catamenial pneumothorax due to bilateral pulmonary endometriosis. Respir Care 2012;57(7):1182-1185. https://doi. org/10.4187%2Frespcare.01256 8. Bridge-Cook P. Thoracic endometriosis. Hormones matter, 2016. https://www. hormonesmatter.com/thoracic-endometriosis/ (accessed 13 April 2017). 9. Sevinc S, Unsal S, Ozturk T, et al. Thoracic endometriosis syndrome with bloody pleural effusion in a 28-year-old woman. J Pak Med Assoc 2013;63(1):114-116. https:// doi.org/ 10.1016/j.ijscr.2017.02.052 10. Nair SS, Nayar J. Thoracic endometriosis syndrome: A veritable Pandoraâ&#x20AC;&#x2122;s box. J ClinDiag Res 2016;10(4):QR04-QR08. https://doi.org/10.7860%2Fjcdr%2F2016%2F17668.7700 11. Nemes RM, Paleru C, Danaila O, et al. Thoracic endometriosis with long delay in diagnosis. Rom J Morphol Embryol 2015;1:295-300. 12. Joseph J, Reed CE, Sahn SA. Thoracic endometriosis: Recurrence following hysterectomy with bilateral salpingo-oophorectomy and successful treatment with talc pleurodesis. Chest 1994;106(6):1894-1896. https://doi.org/10.1378%2Fchest.106.6.1894 13. Joseph J, Sahn SA. Thoracic endometriosis syndrome: New observation from an analysis of 100 cases. Am J Med 1996;100(2):164-170. https://doi.org/10.1016%2 Fs0002-9343%2897%2989454-5 14. Haga T, Kumasaka T, Kurihara M, Kataoka H, Miura M. Immunohistochemical analysis of thoracic endometriosis. Path Int 2013;63(9):429-434. https://doi. org/10.1111%2Fpin.12089
15. Dvorakovskaya I, Platonova I, Pechennikova V, et al. Intrapulmonary endometriosis is rare thoracic pathology. Eur Resp J 2016;48(S60):PA579. https://doi. org/10.1183%2F13993003.congress-2016.pa579 16. Ekpe EE, Bassey EA, Umanah IN. Thoracic endometriosis syndrome, not so rare; report of 3 cases. Case Study Case Report 2013;3(2):95-102. 17. Gumanga S, Takyi G, Ofosu H, Imogie S, Koomson B. Catamenial pneumothorax masquerading pulmonary tuberculosis? A case report of a 28 year old woman who presented with recurrent haemothorax and empyema thoracis. Am J Med Case Rep 2015;3(11):362-366. https://doi.org/10.12691/ajmcr-3-11-4 18. Byanyima RK. Menstruation in an unusual place: A case of thoracic endometriosis in Kampala, Uganda. Afr Health Sci 2001;1(2):97-98. 19. Ngwenya S. Pulmonary endometriosis: A review. Pulm Res Resp Med Open J 2016;3(2):30-32. https://doi.org/10.17140%2Fprrmoj-3-128 20. Notzold A, Moubayed P, Sievers HH. Endometriosis in the thoracic aorta. N Engl J Med 1998;339(14):1002-1003. https://doi.org/10.1056%2Fnejm199810013391413 21. Nezhat C, Main J, Paka C, Nezhat A, Beygui RE. Multidiciplinary treatment of thoracic and abdominopelvic endometriosis. J Soc Laparoendos Surg 2014;18(3):e2014.00312. https://doi.org/10.4293%2Fjsls.2014.00312 22. Hilaris GE, Payne CK, Osias J, Cannon W, Nezhat CR. Synchronous rectovaginal, urinary bladder and pulmonary endometriosis. J Soc Laparoendos Surg 2005;9(1):78-82. 23. Woodward PJ, Sohaey R, Mezzetti TP. Endometriosis: Radiologic pathologic correlation. Radiographics 2001;21(1):193-216. https://doi. org/10.1148%2Fradiographics.21.1.g01ja14193 24. Bulletti C, Coccia EM, Battistoni S, Borini A. Endometriosis and infertility. J Assist Reprod Genet 2010;27(8):441-447. https://doi.org/10.1007%2Fs10815-010-9436-1
Accepted 24 January 2018.
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A bronchogenic cyst masquerading as asthma: A case report M D Moremi, BSc, MB ChB, DCH, FCPaed, Dip Allergology, Cert Pulm (SA); A L Motene, MB ChB, FCPaed, Cert Pulm (SA); N J Maligavhada, BSc, MB ChB, DCH, FCPaed, Dip Allergology, Cert Pulm (SA); N G Tiva, MB ChB, FCPaed, Cert Pulm (SA); R T Mamogale, MB ChB, MMed Rad Diag; S M Risenga, BSc, MB ChB, DCH, MMed, Dip Allergology, Cert Pulm (SA) 1 2
Department of Paediatric Pulmonology and Allergy, University of Limpopo and Pietersburg Provincial Hospital, Polokwane, South Africa Department of Diagnostic Radiology, University of Limpopo and Pietersburg Provincial Hospital, Polokwane, South Africa
Corresponding author: S M Risenga (sam.risenga@gmail.com)
Wheezing in infants and under-five children may present a diagnostic problem as there are various aetiologies for this symptom. Diagnosis of asthma is often made as it is one of the causes of wheezing in children. It is however important to have taken a complete history, including allergy and appropriate diagnostic investigations. If the child’s symptoms do not improve despite appropriate therapy, a different diagnosis must be pursued. We report the case of a child who presented to us with wheezing and who did not respond to therapy. Afr J Thoracic Crit Care Med 2018;24(2):92-96. DOI:10.7196/SARJ.2018.v24i2.169
Bronchogenic cysts (BCs) of the mediastinum are uncommon congenital malformations arising from the primitive foregut.[1] Mediastinal congenital bronchogenic cyst, or those found anywhere in the body are usually asymptomatic.[2] They are at times discovered as an incidental finding, becoming a source of diagnostic confusion.[2] Symptoms develop when they compress adjacent structures, become infected or increase in size.[2] Most BCs are asymptomatic at birth.[3] The majority of BCs are mediastinal and some can occur in the lung parenchyma.[4]
Case
A 27-month-old boy was referred from one of our district hospitals to the Pietersburg tertiary hospital with an assessment of an acute asthmatic attack. The child was in severe respiratory distress and not responding to therapy, which included salbutamol nebulisations and oral prednisone. The child did not have a history of haemoptysis, fever, dysphagia, diarrhoea or weight loss. There was no history of tuberculosis contact in the household. He was fully immunised with normal milestones and good nutrition. Previous admissions could not be elicited although he was treated on a few occasions by his general practitioner for recurrent chest infections and noisy breathing prior to this admission. The therapy at the time included salbutamol syrup, which is not standard therapy for asthma, oral prednisone and antibiotics; with no improvement. Two months before this presentation he was diagnosed with asthma at a district hospital where he was treated with budesonide metred dose inhaler (MDI) 100 µg twice a day and 2 puffs of salbutamol MDI 100 µg when needed. However, there was minimal clinical response to this therapy, with persistence and worsening of symptoms. Clinical examination on the day of admission revealed severe respiratory distress as evidenced by nasal alar flaring, intercostal and subcostal recessions. The child was not cyanosed but had signs of superior vena caval syndrome as evidenced by distended neck veins, cough, facial swelling and hoarseness of voice. He had no signs of chronic lung or cardiac disease but had significant lymphadenopathy
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(>2 cm) involving axillary and inguinal regions. He had audible wheezes, with tracheal tug and a hoover sign. His heart sounds were normal, with no signs of pulmonary hypertension. Vital data monitoring while on oxygen by face mask showed saturations of 80%, a respiratory rate of 67 breaths per minute and heart rate of 182 beats per minute. The patient was intubated and ventilated in the paediatric intensive care unit (PICU) and commenced on oral prednisone and nebulised with fenoterol and ipratropium bromide hourly as a continuation of asthma therapy while looking for an alternative diagnosis. He was started on second-line antibiotics, tazocin and amikacin and required sedation with dormicum and morphine. The initial assessment was that of an intrathoracic airway obstruction secondary to a mediastinal mass and a respiratory tract infection. The following investigations were performed: • Chest X-ray, which confirmed that the endotracheal tube was in situ. It also showed widening of the mediastinum (Figs 1 and 2) • Urgent computerised axial tomography (CAT) scan of the chest (Figs 5 - 9) • Blood gas analysis, which documented respiratory acidosis. • Additional haematological tests (urea and electrolytes, full blood count, liver function, blood cultures and C-reactive protein) were mostly normal, with two exceptions – a high C-reactive protein of 172 ml/L and Klebsiella pneumoniae and Candida fumata were cultured from blood. The infections were treated appropriately. The differential diagnosis at this point included tuberculous adenitis, lymphoma, bronchogenic cyst and thoracic neuroblastoma. The mass was deemed to be exerting pressure on the oesophagus because, despite the insertion of a nasogastric tube, the child continued to vomit feeds. The cardiothoracic surgical team was consulted and a diagnosis of a bronchogenic cyst was made. A thoracotomy was performed on day four of admission. Intraoperative findings were consistent with an intrathoracic cyst. A cystectomy was performed and an infected
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Fig. 1. Pre-operative chest X-rays of the patient (not intubated). Widening of the superior mediastinum with early left lower-lobe patchy infiltrates and right lung upper-lobe early herniation to the left.
Fig. 3. Superior mediastinal mass, collapsed left lung showing mediastinal shift to left. Right lung hyperinflation and herniation to the left.
Figure 2. Patient intubated, rotated, left-lung early collapse consolidation and middle lobe consolidation. cyst was removed. Ventilation of the patient in PICU was continued postoperatively. The postoperative course was eventful â&#x20AC;&#x201C; a new fungal sepsis was identified and managed. A surgical relook procedure and bronchoscopy were performed and the intraoperative findings did not show any collections or puss in the excised cystic area. Bronchoscopy revealed a left bronchomalacia, with thick secretions (Fig. 12). The patient was successfully ventilated for thirty days in PICU. At the time of submission of this manuscript for publication, the patient
Fig. 4. Superior mediastinal mass, collapsed left lung showing mediastinal shift to left. Right lung hyperinflation and herniation to the left. was well and not receiving any treatment for asthma. Post discharge control chest X-rays and CAT scan of the chest were normal.
Histology of the cyst
Macroscopically, the specimen consisted of an opened cyst that measured 70 Ă&#x2014; 45 mm. Microscopic examination of the cyst showed a fibromuscular wall and a respiratory epithelium lining, with areas of squamous metaplasia. Cartilage was focally present in the wall. The cyst was multilocular with acute on chronic inflammation and destruction of epithelial lining and was identified as a mediastinal BC.
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Fig. 5. Middle mediastinal cystic mass lesion. Displacing and compressing the airway and the oesphagus.
Fig. 7. Prominent multiple veins noted on the anterior chest wall, neck and left shoulder with associated prominent right jugular vein from raised jugular venous pressure from compressed superior vena cava. Proximal oesophageal dilation with nasogastric tube in situ from proximal oesophageal mass effect.
Fig. 6. Atresia of left bronchus due to chronic mass effect.
Discussion
Although BCs are relatively rare, they are the most common cysts in the mediastinum. [1] Serious and life-threatening events can occur due to compression of major airways, severe infections, haemorrhages, malignant transformation, fistula formation, superior vena caval syndrome and rupture.[2] Symptoms vary with age at presentation and up to a large extent depend upon size and location of the cyst. Most cysts are asymptomatic at an early age. Symptoms are related to the location and site of the cysts. In children, symptoms occur as a result of compression of the trachea, main bronchus and oesophagus.[3] Chest X-ray (CXR) findings are nonspecific, but do suggest a possible mediastinal mass;[4] however, CXR is the first line of investigations.[4] CAT scan of the chest is useful in localising these cysts while also helping with the diagnosis and decision of surgical treatment.[5] Treatment strategy of asymptomatic BCs is controversial, the consensus is that all BCs whether asymptomatic, symptomatic and those with complications should be treated surgically. [5] Some surgeons perform surgical intervention in all asymptomatic patents in order to establish diagnosis and to prevent complications.[4] Foregut cysts are classified as BCs, which can be mediastinal or intrapulmonary and represent many of bronchopulmonary malformations.[5] Foregut cysts arise from an abnormal budding
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Fig. 8. Middle mediastinal cystic mass with mass effect on the superior vena cava and trachea and with associated anterior displacement of the anterior mediastinum, left lung collapse and right lung compensatory hyperinflation.
Fig. 9. Right lung hyperinflation with multiple diffuse patchy infiltrates suggestive of infection and early consolidation.
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Fig. 12. Bronchoscopy showing narrowing of the left main bronchus.
Fig. 10. Postoperative chest X-rays. Endotracheal tube, nasogadtric tube, and intercostal drain in situ. Malaligned right upper ribs, right lung contusions. Superior mediastinum less dense and reduced. Left lung reinflated. Right lung normal volume.
Fig. 11. Endotracheal tube, nasogadtric tube, and intercostal drain in situ. Malaligned right upper ribs, right lung contusions. Superior mediastinum less dense and reduced. Left lung was reinflated, while the right lung had a normal volume. of primitive foregut before the 16th week of gestation. They can be classified into enteral or BCs.[3] The walls are made up of tissue similar to that of normal bronchial tree, including cartilages, elastic tissue, mucous glands and smooth muscle, usually solitary and lined by cuboidal or columnar ciliated epithelial cells.[3]
It is reported that the majority of BCs are located in the mediastinum, usually in close relationship to trachea and major conducting airways.[6] Although the majority of BCs are located inside the mediastinum, a series of 29 cases reported by Kosar et al.[5] found a majority of 78.4% in the intrapulmonary location.[5] This they describe as somewhat controversial to the literature. Intrapulmonary BCs are commonly located in the lower lobes of the lungs.[6] A study by Chang et al. [6] confirmed this location of intrapulmonary BCs. In their study the most common location was the subpleural region of the lower lobe (55%), followed by the midlung (30%) and perihilar region (15%).[6] Jiang et al. [7] confirmed in their case series that mediastinal BCs are observed more often in adults than children, 67% of their cases occurring in adults. A minority of BCs is asymptomatic.[2] This may lead to productive cough, dyspnoea, chest pain, haemoptysis, fever.[2] Wheezing can occur due to compression of the airways and the patient can have digestive tract symptoms. Haemorrhages, recurrent Infections occur later in life and pleural perforations are possible. Serious complications from BCs are rare, but can include superior vena caval syndrome, tracheal compression, pneumothorax, pleurisy and recurrent chest infections.[4] There is risk of malignant degeneration to rhabdomyosarcoma, adenocarcinoma, and malignant melanoma.[8] Bronchomalacia is one of the complications post removal of the cyst secondary to the compression of the mass to the bronchus. It can be associated with tracheobronchomalacia with the affected bronchus lacking rigidity because of insufficient cartilages or extrinsic compression.[9] Acquired or secondary bronchomalacia can result from extrinsic compression of the bronchus.[9] Flexible bronchoscopy performed by an experienced bronchoscopist is the gold standard in establishing the diagnosis of bronchomalacia. Differential diagnosis of BCs includes lung abscess, hydatid cyst, infected bullae, lung sequestration, lobar emphysema, lymphoma, Neuroblastoma and tuberculous adenitis. Clinical findings and plain radiographs may be sufficient to make diagnosis, but confirmation with computed tomography scanning may help to establish nature of the lesion and further management.[8] Barium swallows are sufficient for the diagnosis in 80% of cases at all ages. Computed axial tomography is accurate in determining the anatomy of mediastinal mass.[2]
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RESEARCH Definitive diagnosis can only be confirmed by histopathological examination following surgical operation.[5] For asymptomatic BCs treatment option remains controversial but general consensuses are that all BCs should be treated surgically even if asymptomatic, since the majority will ultimately become symptomatic or develop complications later on in life.[5] Complete surgical resections for all symptomatic patient is recommended.[10] This is because complete surgical resection is the only definite and radical management of mediasternal bronchogenic cyts, allows symptom suppression and achievement of a histological diagnosis.[11] Histological findings in BCs are mostly the presence of ciliated columnar epithelial cells, smooth muscles bronchial gland, cartilage and even lung alveoli along the wall of the cyst.[12] Other treatment options are transparietal, transbronchial or mediastinoscopic puncture and aspiration which are exceptionally preferred as temporal measures in case of acute compression in selected cases.[7] Thoracosopic resection has major advantages that include less pain, better cosmetic and decreased risk of rib fusion. Video-assisted thoracic surgery is reported to be safe, convenient and practical with minimal morbidity. It is especially better in those who do not prefer thoracotomy.[4] Lobectomy is the standard procedure as the cyst is often surrounded by areas of atelectasis and pneumonia for intrapulmonary cysts.[5]
Author contributions. All authors contributed equally to the preparation of the manuscript. Funding. None. Conflicts of interest. None.
The persistently wheezing child and infant present a diagnostic and therapeutic dilemma to paediatricians.[9] Patients who present with signs that mimic asthma and do not respond to the correct treatment for asthma need further investigations. In our case study the patient had more or recurrent chest infections and did not respond well to the asthma medications as prescribed by the general practioner. Simple chest X-rays and clinical assessment can provide more information to the clinician. Most of them are asymptomatic but can cause compressive symptoms or infections and complications.[13] Surgical excision is indicated to enable definitive pathological diagnosis and to prevent complications and potential risk of malignant transformation in these patients.[3]
1. Konstantinov IE, Saxena P, Vanden Driesen R, Newman MAJ. Large bronchogenic cyst: Diagnostic and surgical management. Heart Lung Circ 2008;17(2):146-148. https://doi.org/10.1016/j.hlc.2006.10.010 2. Hsu CG, Heller M, Johnston GS, Felberbaum M. An unusual cause of airway compromise in the emergency department: Mediastinal bronchogenic cyst. J Emerg Medic 2016;52(3):e91-e93. https://doi.org/10.1016/j.jemermed.2016.11.004 3. Goldsztein H, Miranda A, Pinczewski J, Pereira KD. Bronchogenic cyst: An unusual cause of respiratory distress in a neonate. Int J Pediatr Otorhinolaryngol 2011;6(4):297299. https://doi.org/10.1016/j.pedex.2011.02.001 4. Amore D, Francesco SC, Perrotta F, Cennamo A. Curcio C. Bilateral Simultaneous VATS for complete resection of bilateral posterior mediastinal bronchogenic cyst: A case report. Int J Surg Case Rep 2016;28:149-151. https://doi.org/10.1016/j. ijscr.2016.09.020 5. Kosar A, Tezel C, Orki A, Kiral H, Arman B. Bronchogenic cyst of the lung: Report of 29 cases. Heart Lung Circ 2008;18(3):214-218. https://doi.org/10.1016/j. hlc.2008.10.011 6. Chang YC, Chang YL, Chen SY, et al. Intrapulmonary bronchogenic cyst: Computed tomography, clinical and histopatholologic correlations. J Formos Med Assoc 2007;106(1):8-15. https://doi.org/10.1016/s0929-6646(09)60210-2 7. Kawaguchi Y, Hanaoka J, Asakura S and Fjiita T. Infected bronchogenic cyst treated with drainage followed by resection. Ann Thorac Surg 2014;98(1):332-334. https://doi. org/10.1016/j.athoracsur.2013.09.070 8. Rose SH, Elliott BA, Brown MJ, Long TR, Wass TC. Perioperative risk associated with an unrecognized bronchogenic cyst: Clinical significance and anesthetic management. Cardiothorac Vasc Anesth 2007;21(5):720-722. https://doi.org/10.1053/j. jvca.2006.07.003 9. Finder DJ. Primary Bronchomalacia in Infants and Children. J Pediatr 1997; 130:59-66 10. Rasihashemi SZ, Sokouti M, Bozorgi F. A pitfall in the diagnosis of giant bronchogenic cyst presented as loculated pleural effusion. Heart Lung Circ 2012;21(4):240-241. https://doi.org/10.1016/j.hlc.2011.07.016 11. Amore D, Cerqua FS, Perota F, Cernammo A, Curcio C. Bilateral simultaneous VATS for complete resection of bilateral posterior mediastinal bronchogenic cyst: A case report. Int J Surg Case Rep 2016;28:149-151. https://doi.org/10.1016/j. ijscr.2016.09.020 12. Ko SF, Hsieh M, Lin J, et al. Bronchogenic cyst of the oesophagus: clinical; and imaging features of seven cases. Clin Imag 2006;30(5):309-314. https://doi.org/10.1016/j. clinimag.2006.02.003 13. Jiang JH, Yen SL, Lee SY, Chuang JH. Differences in the distribution and presentation of bronchogenic cysts between adults and children. J Pediatr Surg 2015;50(3):399-401. https://doi.org/10.1016/j.jpedsurg.2014.06.008
Acknowledgements. Prof. Robin Green, who helped with corrections before the article was submitted.
Accepted 18 January 2018.
Conclusion
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Complete lung collapse in a young adolescent T H Ahmed, MB ChB, MD ; A Esmail, MD, FCP, Cert Pulm; G Calligaro, BSc Hons (Phys) MB ChB, FCP, MMed, Cert Pulm; K Dheda, MB ChB, FCP, FRCP, PhD Division of Pulmonology, Department of Medicine, University of Cape Town Lung Institute and Groote Schuur Hospital, Cape Town, South Africa Corresponding author: T H Ahmed (dr.tarig@hotmail.com)
Bronchial carcinoid tumours (BCTs) arise from the neuroendocrine cells of the bronchial epithelium known as Kulchitsky cells. They represent ~25% of all carcinoid tumours, usually have a central distribution, and present with features of bronchial obstruction. They are the most common lung malignancy in children. Here we report the case of a 14-year-old girlwith chronic respiratory symptoms and left lung collapse due to bronchial carcinoid. The differential diagnosis of segmental, lobar or total lung collapse in a young person also includes mucus plugging or foreign body aspiration. Afr J Thoracic Crit Care Med 2018;24(2):97-98. DOI:10.7196/SARJ.2018.v24i2.209
Bronchial carcinoid tumours (BCTs) were first described in 1888 by Lubarsch, who found multiple tumours in the distal ileum of two patients on autopsy. In 1907, Oberndorfer coined the term ‘karzinoid tumoren’ to describe ileal tumours that appeared to behave less aggressively than typical adenocarcinomas.[1] BCTs represent ~25% of all carcinoid tumours – the remainder can be seen primarily in the gastrointestinal tract, but also the mediastinum, thymus, liver, pancreas, ovaries, prostate and kidneys. BCTs are divided into typical and atypical tumours based on their pathological tumour grade. Typical BCTs are slow-growing tumours that rarely metastasise, whereas atypical tumours metastasise early to the hilar or mediastinal nodes and are associated with a higher recurrence rate.[2] Surgery is the gold standard for patients with resectable lung carcinoids, but treatment options are very limited for patients with metastatic or unresectable disease. The incidence of BCTs has increased over the past 30 years, which may be due to a greater effort to better characterise these neoplasms, with the help of a multidisciplinary approach.
Case
A 14-year-old girl was referred to our unit from the emergency department at our hospital with a short history of dyspnoea and leftsided chest pain. Her symptoms worsened acutely following a suspected lower respiratory tract infection. She had a three-month history of progressive exercise limitation and wheeze. She was presumptively diagnosed with asthma and started on bronchodilators. Clinical examination revealed tachypnoea and saturations of 92% on room air, reduced expansion of the left chest, tracheal deviation to the left, and absent breath sounds over the left hemithorax. A chest X-ray showed complete collapse of the left lung with cut-off in the proximal left main bronchus (Figs 1A and 1B). There was no history of foreign body aspiration. A flexible bronchoscopy was done which showed a smooth, reddish-yellow polypoidal mass occluding the entire lumen of the left main bronchus (Fig. 1C). Due to the proximity of the mass to the carina, and its vascularity, endobronchial biopsies were not performed. Computed tomography (CT) of the chest showed the endobronchial mass to be part of a large inhomogeneous lesion occupying the entire left upper lobe (Fig. 1D), and the patient was
referred for pneumonectomy. The surgical specimen is shown in Figs 1E and 1F. Histological evaluation showed the presence of uniform polygonal cells with finely granular chromatin in round nuclei and a moderate amount of eosinophilic cytoplasm without any nuclear atypia, mitosis or necrosis (Fig. 1G). Immunohistochemistry showed neuroendocrine differentiation of tumour cells with cytoplasmic positivity of cytokeratin, chromogranin A and synaptophysin (Fig. 1H). A diagnosis of typical carcinoid tumour was confirmed.
Discussion
BCTs are rare neuroendocrine neoplasms of the lung, which is the second most common site of carcinoid tumours after the abdomen. BCTs may be central or peripheral, and have been reported in all age groups, but the peak incidence is around the fourth to fifth decade of life.[3] About 80% of bronchial carcinoids arise centrally in the main, lobar and segmental bronchi with no specific lobar distribution.[4] Central tumours are usually symptomatic with features of bronchial obstruction, whereas peripheral tumours are usually asymptomatic.[4] The most common symptoms are haemoptysis, cough, recurrent chest infections, fever, unilateral wheezing and dyspnoea.[5] Due to the high tumour vascularity, haemoptysis occurs in at least 50% of patients,[6] whereas 25% of patients are diagnosed incidentally.[7] Patients are often misdiagnosed with airways disease, as occurred with our patient. Diagnosis in our case was further delayed as this patient had no haemoptysis despite having a centrally located tumour. Aetiologically, no association has been found between BCTs and cigarette smoking, ambient radiation or exposure to other known carcinogens;[8] however, a recent study described a possible association between atypical carcinoids and smoking.[9] Radiologically, typical and atypical BCTs have similar features, depending on tumour location. Most BCTs appear on chest radiographs or chest CT as circumscribed, centrally located lesions with a diameter of 2 - 5 cm.[10] They are therefore usually easily accessible via flexible bronchoscopy. Due to their high vascularity, there has been a historical concern about the safety of endobronchial biopsy in BCTs; however, recent studies, as well as the British Thoracic Society, refer to the incidence of serious
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RESEARCH as possible. Our patient presented with complete obstruction of the left lung with a mass occupying a significant proportion of the lung parenchyma, and, as a result, underwent pneumonectomy. The survival following surgery for BCTs is excellent: a 5-year survival rate of 94% was observed in 1 109 patients with typical BCTs by the European Society of Thoracic Surgeons Neuroendocrine Tumours Working Group, and a 3-year survival rate of 67% was observed in the USA in a database analysis of 441 patients[13,14] following resection of the tumour. Chemotherapy and radiation therapy is largely unhelpful if unresectable or metastatic disease is present. Typical bronchial carcinoids generally have an excellent prognosis, whereas atypical bronchial carcinoids have a poorer prognosis. Delay in diagnosis can result in potential complications from increased tumour size causing obstruction of the bronchus and destructive changes in the lung. Our patient had an uneventful recovery, with no residual respiratory symptoms or functional limitation. Acknowledgements. THA thanks the patient for consenting to publication of her case, and the co-authors (KD, GC and AE) for their support and comments during the preparation of this manuscript. Author contributions. GC and TAH conceptualised the paper. TAH prepared the manuscript. KD and AE assisted with manuscript preparation and final review. Funding. None. Conflicts of interest. None.
Fig. 1. (A and B) Chest radiograph PA and lateral showing left lung collapse (left hemithorax homogenous opacity with volume loss). (C) Mediastinal window shows a well-defined intraluminal growth in the the left main bronchus occluding the airway (arrow). (D) Fibre optic bronchoscopy, reddish-yellow polypoidal mass occluding whole lumen of the left main bronchus. (E and F) Post pneumonectomy view. Huge bulky tumour removed from left main bronchus. (G) Round nuclei and ample amounts of granular cytoplasm seen at high power. (H) Stain positive for chromogranin and synaptophysin. bleeding complications during bronchoscopic biopsy as being very low (<1%).[11] Final diagnosis is based on histology and confirmed with immunohistochemical staining for neuroendocrine markers. Resection is the treatment of choice,[12] and surgical procedures include pneumonectomy, lobectomy, segmentectomy, sleeve resection and wedge resection. The aim is to remove the primary tumour and affected lymph nodes radically, saving as much of the lung parenchyma
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1. Oberndorfer S. Karzinoide tumoren des dunndarms. Frankfurt Z Pathol 1907(1):426432. 2. Gonzalez JM, Garcia-Yuste M, Moreno-Mata N, et al. Typical and atypical carcinoid tumors (NEC grades 1 and 2): Prognostic factors in metastases and local recurrence. Lung Cancer 2005;49:S60. https://doi.org/10.1016/s0169-5002(05)80313-7 3. Tsilimigras DI, Moris D, Ntanasis-Stathopoulos I, Patrini D, Panagiotopoulos N. Endobronchial carcinoid tumor totally occluding the left main bronchus without producing symptoms of bronchial obstruction. In Vivo 2017;31(5):1023-1025. https:// doi.org/10.21873/invivo.11164 4. Nessi R, Basso PR, Basso SR, Bosco M, Blanc M, Uslenghi C. Bronchial carcinoid tumors: Radiologic observations in 49 cases. J Thorac Imaging 1991;6(2):47-53. 5. Zuetenhorst JM, Taal BG. Metastatic carcinoid tumors: A clinical review. Oncologist 2005;10(2):123-131. https://doi.org/10.1634/theoncologist.10-2-123 6. Dusmet ME, McKneally MF. Pulmonary and thymic carcinoid tumors. World J Surg 1996;20(2):189-195. https://doi.org/10.1007/s002689900029 7. Ducrocq X, Thomas P, Massard G, et al. Operative risk and prognostic factors of typical bronchial carcinoid tumors. The Annals of thoracic surgery, 1998. 65(5):1410-1414. https://doi.org/10.1016/S0003-4975(98)00083-6 8. Davila DG, Dunn WF, Tazelaar HD, Pairolero PC. Bronchial carcinoid tumors. Mayo Clin Proc 1993;68(8):795-803. https://doi.org/10.1016/S0025-6196(12)60641-7 9. Fink G, Krelbaum T, Yellin A, et al. Pulmonary carcinoid: Presentation, diagnosis, and outcome in 142 cases in Israel and review of 640 cases from the literature. Chest 2001;119(6):1647-1651. https://doi.org/10.1378/chest.119.6.1647 10. Hage R, de la Rivière AB, Seldenrijk CA, Van den Bosch JM. Update in pulmonary carcinoid tumors: A review article. Ann Surg Oncol 2003;10(6):697-704. https://doi. org/10.1245/ASO.2003.09.019 11. Kaifi JT, Kayser G, Ruf J, Passlick B. The diagnosis and treatment of bronchopulmonary carcinoid. Deutsches Ă&#x201E;rzteblatt International 2015;112(27-28):479. https://doi. org/10.3238/arztebl.2015.0479 12. Caplin ME, Baudin E, Ferolla P, et al. Pulmonary neuroendocrine (carcinoid) tumors: European Neuroendocrine Tumor Society expert consensus and recommendations for best practice for typical and atypical pulmonary carcinoids. Ann Oncol 2015;26(8):16041620. https://doi.org/10.1093/annonc/mdv041 13. Filosso PL, Guerrera F, Evangelista A, et al. Prognostic model of survival for typical bronchial carcinoid tumours: analysis of 1109 patients on behalf of the European Association of Thoracic Surgeons (ESTS) Neuroendocrine Tumours Working Group. Eur J Cardio-Thorac Surg 2015;48(3):441-447. https://doi.org/10.1093/ejcts/ezu495 14. Steuer CE, Behera M, Kim S, et al. Atypical carcinoid tumor of the lung: A surveillance, epidemiology, and end results database analysis. Thorac Oncol 2015;10(3):479-485. https://doi.org/10.1097/JTO.0000000000000419
Accepted 12 April 2018.
BREATH-TAKING NEWS
Triple therapy has an impact in COPD! The debate on the optimal therapy for severe chronic obstructive pulmonary disease (COPD) has long been characterised by divided opinions, with the central point of dispute being whether or not an inhaled corticosteroid (ICS) provides additional benefit over a long-acting beta agonist/long-acting muscarinic antagonist (LABA/LAMA) combination, and in which patients their uses are appropriate. One camp cites the FLAME (effect of indacaterol/ glycopyrronium v. fluticasone/salmeterol on COPD exacerbations) study and its supporting literature as evidence that exacerbation reduction from the LABA/LAMA combination is superior to that provided by a LABA/ICS combination.[1,2] Additionally, concerns have been raised about the increased rate of pneumonia in COPD patients treated with ICS, which was first identified by the TORCH (towards a revolution in COPD health) study and later confirmed by post-hoc analyses of other large cohorts. [3,4] Other clinicians will reference the WISDOM (withdrawal of inhaled steroids during optimised bronchodilator management) trial and similar studies, which identified an increase in exacerbations and potentially accelerated decline in lung function when ICS therapy was withdrawn. [5,6] The discussion becomes further nuanced when considering the eosinophilic phenotype of COPD, with the literature (mostly post hoc analyses) suggesting that this subset of patients may have not only experienced symptomatic benefits and exacerbation reduction, but also reduction in lung function decline when treated with an ICS. [7] The current international guidelines recommend only initiating so-called ‘triple therapy’ with LABA/LAMA/ICS in patients with severe symptoms as well as exacerbations (GOLD (Global Initiative for Obstructive Lung Disease) D classification).[8] A large, randomised and controlled trial directly comparing triple therapy with dual bronchodilator therapy (LABA/LAMA) has long been awaited,[9] and the recently published IMPACT (informing the pathway of COPD treatment) study has met all these criteria.[10] IMPACT was a three-group randomised trial involving over 10 000 patients, most of whom fell into the GOLD D category and were followed over 1 year. The study directly compared triple therapy (vilanterol/umeclidinium/fluticasone fuorate) with LABA/ICS (vilanterol/fluticasone fuorate) and LAMA/ LABA (umeclidinium/vilanterol), all administered once daily via a single dry-powder inhaler. The data showed a 25% reduction in exacerbations in patients who were on triple therapy, compared with those who received LABA/LAMA. This difference was even more pronounced in the eosinophilic group of patients, where the reduction in the annual rate of moderate or severe exacerbations was 44%. As expected, there was a small increase in the incidence of pneumonia in the groups treated with ICS, with an absolute increase in incidence of 3% in the triple-therapy group, compared with those on bronchodilators alone. The change in trough FEV1 from baseline was significantly larger in the triple-therapy group
than both other groups, and the data suggest that both the LAMA and ICS components contributed to this difference, with the latter augmenting the effect of the bronchodilators. Although none of the lung function differences reached the 100 mL value of the minimal clinically important difference, the change in St George’s Respiratory Questionnaire (SGRQ) score reached clinical significance, in the triple-therapy group only. Lastly, and perhaps most importantly, all-cause mortality was significantly lower in the ICS-containing groups than the LABA/LAMA group, with a hazard ratio of 0.58 for triple therapy and 0.61 for LABA/ICS. IMPACT provides good evidence for the step-up to triple therapy in this patient population, and particularly in patients with blood eosinophilia – unless there is a high risk or previous history of pneumonia. It will be interesting to see how the debate evolves in this important disease arena. J A Shaw Division of Pulmonology, Department of Medicine, Tygerberg Academic Hospital and Stellenbosch University, Cape Town, South Africa janeshaw@sun.ac.za
Afr J Thoracic Crit Care Med 2018;24(2):99. DOI:10.7196/ SARJ.2018.v24i2.219 1. Wedzicha JA, Banerji D, Chapman KR, et al. Indacaterol-Glycopyrronium versus Salmeterol-Fluticasone for COPD. N Engl J Med 2016;374(23):2222-2234. https:// doi.org/10.1056/nejmoa1516385 2. Rodrigo GJ, Price D, Anzueto A, et al. LABA/LAMA combinations versus LAMA monotherapy or LABA / ICS in COPD: A systematic review and meta-analysis. Int J COPD 2017;12:907-922. https://doi.org/10.2147/copd.s130482 3. Calverley PM, Anderson JA, Celli B, et al. Salmeterol and fluticasone propionate and survival in chronic obstructive pulmonary disease. N Engl J Med 2007;356(8):775789. https://doi.org/10.1056/nejmoa063070 4. Kew KM, Seniukovich A. Inhaled steroids and risk of pneumonia for chronic obstructive pulmonary disease. Cochrane Database Syst Rev 2014;3:CD010115. https://doi.org/10.1002/14651858.cd010115.pub2 5. Magnussen H, Disse B, Rodriguez-Roisin R, et al. Withdrawal of inhaled glucocorticoids and exacerbations of COPD. N Engl J Med 2014;371(14):1285-1294. https://doi. org/10.1056/nejmoa1407154 6. Calzetta L, Matera MG, Braido F, et al. Withdrawal of inhaled corticosteroids in COPD: A meta-analysis. Pulm Pharmacol Ther 2017;45(August):148-158. https:// doi.org/10.1016/j.pupt.2017.06.002 7. Bafadhel M, Peterson S, De Blas MA, et al. Predictors of exacerbation risk and response to budesonide in patients with chronic obstructive pulmonary disease: A post-hoc analysis of three randomised trials. Lancet Respir Med 2018;6(2):117-126. https://doi.org/10.1016/s2213-2600(18)30006-7 8. Global Initiative for Chronic Obstructive Lung Disease (GOLD). Global Strategy for the Diagnosis, Management and Prevention of COPD. Fontana: GOLD, 2017. www.goldcopd.org%257B%2525%257D0A (accessed 1 May 2018). 9. Calverley PMA, Magnussen H, Miravitlles M, et al. Triple therapy in COPD: What we know and what we don’t. COPD J Chronic Obstr Pulm Dis 2017;14(6):648-662. https://doi.org/10.1080/15412555.2017.1389875 10. Lipson DA, Barnhart F, Brealey N, et al. Once-daily single-inhaler triple versus dual therapy in patients with COPD. N Engl J Med 2018;378(18):1671-1680. https://doi. org/10.1056/nejmoa1713901
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BREATH-TAKING NEWS
A four-gene pan-African blood signature predicts progression to tuberculosis With tuberculosis (TB) now known to be the leading infectious cause of death worldwide, the battle to find an effective preventive strategy is becoming ever more urgent. One approach to the problem is to identify individuals with latent TB infection (LTBI) who are at high risk of progressing to active disease, with a view to intervention. The currently available methods of identifying LTBI, the tuberculin skin test (TST) and interferon-γ release assays (IGRAs), unfortunately have very low positive predictive values of 1.5% and 2.7%, respectively, for progression to active TB. Suliman et al.[1] reported on their efforts to identify a genetic signature in individuals among household contacts (HHCs) of known TB cases who are more likely to progress to active TB. This was a nested case-control study from within the Grand Challenges 6-74 (GC6-74, GC6) HHC study cohort conducted in South Africa (SA), Ethiopia, Uganda and The Gambia, with external validation on the subjects of the Adolescent Cohort Study (also a local cohort). The investigators enrolled 4 466 HIV-negative healthy HHCs of 1 098 index TB cases between 2006 and 2010 into the GC6-74 cohorts, and generated site-specific signatures of TB risk using complex RNA sequencing techniques and transcriptomics. A pooled multisite signature was then identified. The RISK4 signature, which comprises four unique genes – GAS6 (growth arrest-specific 6) and SEPT4 (septin 4), which were upregulated, and CD1C (cluster of differentiation 1C) and BLK (B lymphocyte kinase), which were downregulated in progressors (defined as individuals who developed active TB 3 - 24 months after household contact) – was compared with matched control subjects. On external validation, the RISK4 signature significantly predicted progression in the entire combined test set (area under the curve (AUC) 0.67; 95% CI 0.57 - 0.77; p=2.6 × 10–4) and in each individual site (SA, The Gambia and Ethiopia, with AUCs of 0.66 - 0.72; p<0.03). RISK4 performed similarly well in the samples collected within 2 months of the index case being diagnosed. The researchers then compared the RISK4 signature with three other previously reported prediction scores, finding that it had equal efficacy but was the only one was which validated across all four of the sites. Lastly, the investigators found that when the components of the RISK4 score were individually validated, the ratio between the SEPT4 and BLK primers reproduced the performance of the RISK4 signature
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on all test set cohorts, which suggests that a highly simplified version of RISK4 may be feasible. This highly complex article includes a post hoc meta-analysis of the combined datasets to determine whether the accuracy could be further improved for a signature performing well at all sites. They identified an optimal pair of upregulated and downregulated transcripts, consisting of C1QC (complement C1q C-chain; upregulated) and TRAV27 (T-cell receptor alpha variable gene 27; downregulated), which achieved an AUC >0.76 on all sites. The AUC was further increased to 0.79 when the C1QC/TRAV27 ratio was combined with the ratio between ANKRD22 (ankyrin repeat domain 22; upregulated with TB progression) and OSBPL10 (oxysterol-binding protein-related protein 10; downregulated with progression); however, on external validation with a cohort of adolescents with LTBI, the ANKRD22/OBSPL10 ratio strongly predicted TB progression, but the C1QC/TRAV27 ratio and the combination performed poorly. Overall, this vast and complex body of work must be seen as a signal that the potential for a simple polymerase chain reaction-based blood test to identify individuals likely to progress from LTBI to active TB exists, and may even be feasible in the near future. The authors identify the next steps in investigating this potential as: (i) the assessment of the performance of RISK4 and the two-transcript C1QC/TRAV27 signature in other settings, including non-African populations; and (ii) a determination of the feasibility of developing a near-patient test for targeted intervention. Watch this space. Sami M Bennji Division of Pulmonology, Department of Medicine, Tygerberg Academic Hospital and Stellenbosch University, Cape Town, South Africa saminj12@gmail.com
Afr J Thoracic Crit Care Med 2018;24(2):100. DOI:10.7196/ SARJ.2018.v24i2.220 1. Suliman S, Thompson EG, Sutherland J, et al. Four-gene pan-African blood signature predicts progression to tuberculosis. Am J Respir Crit Care Med 2018;197(9):11981208. https//doi.org/10.1164/rccm.201711-2340OC