September 2016

Volume 114 u Number 09 u Kolkata u September 2016

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Dr S S Agarwal

Dr K K Aggarwal

Dr Debasish Mukherjee

Dr Santanu Sen

National President, IMA

Honorary Secretary General, IMA

Honorary Editor, JIMA

Honorary Secretary, JIMA

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Office bearers for the year 2015-16 IMA Headquarters Dr SS Agarwal (National President) Prof (Dr) A Marthanda Pillai (Imm Past National President) Dr Shailendra N Vora (National Vice President) Dr K Prameela Surender Rao (National Vice President) Dr Om Parkash Singh Kande (National Vice President) Dr Sharad Kumar Agarwal (National Vice President) Dr KK Aggarwal (Hony. Secretary General) Dr RN Tandon (Hony. Finance Secretary) Dr Rajeev Ardey (Hony. Jt. Secretary) Dr Ravi Malik (Hony. Jt. Secretary), Dr Ramesh Kumar Datta (Hony. Jt. Secretary) Dr Sanjoy Banerjee (Hony. Jt. Secretary, Calcutta) Dr Pravin Gogia (Hony. Jt. Secretary) Dr Hans Raj Satija (Hony. Asst. Secretary) Dr Manjul Mehta (Hony. Asst. Secretary) Dr Harish Gupta (Hony. Jt. Fin. Secretary) Dr Ujjwal Kr Sengupta (Hony. Jt. Fin. Secretary, Calcutta) IMA CGP DrVinod Kumar Monga (Dean of Studies) Dr A Raja Rajeshwar (Hony. Secretary)

IMA AMS Dr Kranshankar W Deoras (Chairman) Dr Pullarao Pasumarthy (Hony. Secretary)

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IMA AKN Sinha Institute Dr Shivkumar Utture (Hony. Director) Dr Arbind Kumar Sinha (Hony. Executive Secretary)

IMA NPP Scheme Dr Krishna M Parate (Chairman) Dr Jayairishnan A V (Hony. Secretary)

JIMA Dr Debasish Mukherjee (Hony. Editor) Dr Ranjan Kumar Chakraborty (Hony. Assoc. Editor) Dr Minakshi Gangopadhyay (Hony. Assoc. Editor) Dr Santanu Sen (Hony. Secretary) Dr Sukomol Das (Hony. Asst. Secretary)

IMA Hospital Board of India Dr RV Asokan (Chairman) Dr Ravi Wankhedkar (Hony. Secretary) Dr Anil S Pachnekar (HQs. Secretary)

Your Health Dr Amitabha Bhattacharya (Hony. Editor) Dr Rahul Dutta (Hony. Secretary) Apka Swasthya Dr Prabhat Kumar Tewari (Hony. Editor) Dr Arvind Singh (Hony. Secretary)

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IMA National Health Scheme Dr Ashok S Adhao (Chairman) Dr Alex Franklin (Hony. Secretary) IMA National Pension Scheme Dr Sudipto Roy (Chairman) Dr KV Devadas (Hony. Secretary)

CONTENTS Editorial : u Epistaxis — An Emergecy in day to day Practice — Debasish Mukherjee ................................4 Originals and Papers : u Experience of botulinum toxin therapy in cervical dystonia, blepharospasm and

hemifacial spasm — Satish Chandra, Ritu Agarwal, Jayantee Kalita, Usha K Misra ......................................................................................................7 u Role of nasal endoscopy in the management of intractable epistaxis : our experience — Dwaipayan Mukherjee, Chiranjib Das ......................................................11 Practitioners’ Series : u Incidence of urinary tract infection and urological symptoms in

depot-medroxyprogesterone users — B Nisha, Sunita Malik, Jagdev Kaur, Archana Aggarwal ...................................................................................................14 u A comparative study between skin sutures and skin staples in abdominal surgical wound closure — Chandrashekar N, Prabhakar GN, Vivek PO, Shivakumarappa GM, Fahad Tauheed .........................................................................................17 Preliminary Reports : u Rhinosporidiosis of different organs — a study of 57 cases with review of

literature — Palash Kumar Mandal, Nirmal Kumar Bhattacharyya, Sumedha Dey, Pranab kumar Biswas, Subrata Mukhopadhyay, Dibyendu Gautam ...............21 u Vascular tumours of the female genital tract : a clinicopathologic study of 11 cases — Sainath K Andola, Uma S Andola .............................................................................................25 GP Forum : u Dupatta injuries : an identifiable hazardous entity in a variety of work place

and social scenarios — Ashok Kumar, Pritish Singh, S K Babhulkar, Pramod Jain, Bhavya Sirohi, C M Badole ....................................................................................31 Case Note : u Recurrent cryptomenorrhoea — a successful outcome

— Pradip Kr Saha, Dipak Kr Giri, Haricharan Roy, Satabdi Majhi .........................................33 3

EDITORIAL 5

Editorial Epistaxis — An Emergecy in day to day Practice

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pistaxis, or bleeding from the nose, is a common complaint. It is rarely life threatening but may cause significant concern, especially among parents of small children. Most nosebleeds are benign, self-limiting, and spontaneous, but Dr Debasish Mukherjee some can be recurrent. Many uncommon causes are also noted.

MBBS, DLO, MS Anatomy Honorary Editor, JIMA The nose has a rich vascular supply, with substantial contributions from the internal carotid artery (ICA) and the external carotid artery (ECA).

The ECA system supplies blood to the nose via the facial and internal maxillary arteries. The superior labial artery is one of the terminal branches of the facial artery. This artery subsequently contributes to the blood supply of the anterior nasal floor and anterior septum through a septal branch.

The internal maxillary artery enters the pterygomaxillary fossa and divides into 6 branches: posterior superior alveolar, descending palatine, infraorbital, sphenopalatine, pterygoid canal, and pharyngeal.

The descending palatine artery descends through the greater palatine canal and supplies the lateral nasal wall. It then returns to the nose via a branch in the incisive foramen to provide blood to the anterior septum. The sphenopalatine artery enters the nose near the posterior attachment of the middle turbinate to supply the lateral nasal wall. It also gives off a branch to provide blood supply to the septum.

The ICA contributes to nasal vascularity through the ophthalmic artery. This artery enters the bony orbit via the superior orbital fissure and divides into several branches. The posterior ethmoid artery exits the orbit through the posterior ethmoid foramen, located 2-9 mm anterior to the optic canal. The larger anterior ethmoid artery leaves the orbit through the anterior ethmoid foramen.

The anterior and posterior ethmoid arteries cross the ethmoid roof to enter the anterior cranial fossa and then descend into the nasal cavity through the cribriform plate. Here, they divide into lateral and septal branches to supply the lateral nasal wall and the septum.

The Kiesselbach plexus, or Little’s area, is an anastomotic network of vessels located on the anterior cartilaginous septum. It receives blood supply from both the ICA and the ECA. Many of the arteries supplying the septum have anastomotic connections at this site.

Pathophysiology Bleeding typically occurs when the mucosa is eroded and vessels become exposed and subsequently break.

More than 90% of bleeds occur anteriorly and arise from Little’s area, where the Kiesselbach plexus forms on the septum.The Kiesselbach plexus is where vessels from both the ICA (anterior and posterior ethmoid arteries) and the ECA (sphenopalatine and branches of the internal maxillary arteries) converge. These capillary or venous bleeds provide a constant ooze, rather than the profuse pumping of blood observed from an arterial origin. Anterior bleeding may also originate anterior to the inferior turbinate.

Posterior bleeds arise further back in the nasal cavity, are usually more profuse, and are often of arterial origin (eg, from branches of the sphenopalatine artery in the posterior nasal cavity or nasopharynx). A posterior source presents a greater risk of airway compromise, aspiration of blood, and greater difficulty controlling bleeding.

Etiology Causes of epistaxis can be divided into local causes (eg, trauma, mucosal irritation, septal abnormality, inflammatory diseases, tumors), systemic causes (eg, blood dyscrasias, arteriosclerosis, hereditary hemorrhagic telangiectasia), and idiopathic causes. Local trauma is the most common cause, followed by facial trauma, foreign bodies, nasal or sinus infections, and prolonged inhalation of dry air. Children usually present with epistaxis due to local irritation or recentupper respiratory infection (URI).

In a retrospective cohort study of 2405 patients with epistaxis (3666 total episodes), Purkey et al used multivariate analysis to identify a series of risk factors for nosebleeds. The likelihood of epistaxis was found to increase in patients with allergic rhinitis, chronic sinusitis, hypertension, hematologic malignancy, coagulopathy, or, as mentioned, heredi4

tary hemorrhagic telangiectasia. The investigators also found increased nosebleeds in association with older age and colder weather.

Trauma Self-induced trauma from repeated nasal picking can cause anterior septal mucosal ulceration and bleeding.

This scenario is frequently observed in young children.

Nasal foreign bodies that cause local trauma (eg, nasogastric and nasotracheal tubes) can be responsible for rare cases of epistaxis.

Acute facial and nasal trauma commonly leads to epistaxis. If the bleeding is from minor mucosal laceration, it is usually limited. However, extensive facial traumacan result in severe bleeding requiring nasal packing. In these patients, delayed epistaxis may signal the presence of a traumatic aneurysm.

Patients undergoing nasal surgery should be warned of the potential for epistaxis. As with nasal trauma, bleeding can range from minor (due to mucosal laceration) to severe (due to transection of a major vessel).

Dry weather Low humidity may lead to mucosal irritation. Epistaxis is more prevalent in dry climates and during cold weather due to the dehumidification of the nasal mucosa by home heating systems.

Drugs Topical nasal drugs such as antihistamines and corticosteroids may cause mucosal irritation. Especially when applied directly to the nasal septum instead of the lateral walls, they may cause mild epistaxis. Medications such as nonsteroidal anti-inflammatory drugs (NSAIDs) are also frequently involved.

Septal abnormality Septal deviations (deviated nasal septum) and spurs may disrupt the normal nasal airflow, leading to dryness and epistaxis. The bleeding sites are usually located anterior to the spurs in most patients. The edges of septal perforations frequently harbor crusting and are common sources of epistaxis.

Inflammation Bacterial, viral, and allergic rhinosinusitis causes mucosal inflammation and may lead to epistaxis.

Bleeding in these cases is usually minor and frequently manifests as blood-streaked nasal discharge.

Granulomatosis diseases such as sarcoidosis, Wegener granulomatosis, tuberculosis, syphilis, and rhinoscleroma often lead to crusting and friable mucosa and may be a cause of recurrent epistaxis.

Young infants with gastroesophageal reflux into the nose may have epistaxis secondary to inflammation.

Tumors Benign and malignant tumors can manifest as epistaxis. Affected patients may also present with signs

and symptoms of nasal obstruction and rhinosinusitis, often unilateral.

Intranasal rhabdomyosarcoma, although rare, often begins in the nasal, orbital, or sinus area in children.

Juvenile nasal angiofibroma in adolescent males may cause severe nasal bleeding as the initial symptom.

Blood dyscrasias Congenital coagulopathies should be suspected in individuals with a positive family history, easy bruising, or prolonged bleeding from minor trauma or surgery.

Examples of congenital bleeding disorders include hemophilia and von Willebrand disease.

Acquired coagulopathies can be primary (due to the diseases) or secondary (due to their treatments). Among the more common acquired coagulopathies are thrombocytopenia and liver disease with its consequential reduction in coagulation factors. Even in the absence of liver disease, alcoholism has also been associated with coagulopathy and epistaxis. Oral anticoagulants predispose to epistaxis.

Vascular abnormalities Arteriosclerotic vascular disease is considered a reason for the higher prevalence of epistaxis in elderly individuals.

Hereditary hemorrhagic telangiectasia (HHT; also known as Osler-Weber-Rendu syndrome) is an autosomal dominant disease associated with recurrent bleeding from vascular anomalies. The condition can affect vessels ranging from capillaries to arteries, leading to the formation of telangiectasias and arteriovenous malformations. Pathologic examination of these lesions reveals a lack of elastic or muscular tissue in the vessel wall. As a result, bleeding can occur easily from minor trauma and tends not to stop spontaneously.

Various organ systems such as the respiratory, gastrointestinal, and genitourinary systems may be involved. The epistaxis in these individuals is variable in severity but is almost universally recurrent.

Other vascular abnormalities that predispose to epistaxis include vascular neoplasms, aneurysms, and endometriosis.

Migraine Children with migraine headaches have a higher incidence of recurrent epistaxis than children without the disease.

The Kiesselbach plexus, which is part of the trigeminovascular system, has been implicated in the pathogenesis of migraine.

Hypertension The relationship between hypertension and epistaxis is often misunderstood. Patients with epistaxis commonly present with an elevated blood pressure. Epistaxis is more

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common in hypertensive patients, perhaps owing to vascular fragility from long-standing disease.

Hypertension, however, is rarely a direct cause of epistaxis. More commonly, epistaxis and the associated anxiety cause an acute elevation of blood pressure.

Therapy, therefore, should be focused on controlling hemorrhage and reducing anxiety as primary means of blood pressure reduction.

A study by Sarhan and Algamal, which included 40 patients with epistaxis and 40 controls, reported that the number of attacks of epistaxis was higher in patients with a history of hypertension, but the investigators were unable to determine whether a definite link existed between nosebleeds and high blood pressure. They did find, however, that control of epistaxis was more difficult in hypertensive patients; patients whose systolic blood pressure was higher at presentation tended to need management with packing, balloon devices, or cauterization.

Excessive coughing causing nasal venous hypertension may be observed in pertussis or cystic fibrosis.

Idiopathic causes The cause of epistaxis is not always readily identifiable.

Approximately 10% of patients with epistaxis have no identifiable causes even after a thorough evaluation.[14] Prognosis For most of the general population, epistaxis is merely a nuisance. However, the problem can occasionally be lifethreatening, especially in elderly patients and in those patients with underlying medical problems. Fortunately, mortality is rare and is usually due to complications from hypovolemia, with severe hemorrhage or underlying disease states.

Overall, the prognosis is good but variable; with proper treatment, it is excellent. When adequate supportive care is

provided and underlying medical problems are controlled, most patients are unlikely to experience any rebleeding.

Others may have minor recurrences that resolve spontaneously or with minimal self-treatment. A small percentage of patients may require repacking or more aggressive treatments.

Patients with epistaxis that occurs from dry membranes or minor trauma do well, with no long-term effects. Patients with HHT tend to have multiple recurrences regardless of the treatment modality. Patients with bleeding from a hematologic problem or cancer have a variable prognosis.

Patients who have undergone nasal packing are subject to increased morbidity. Posterior packing can potentially cause airway compromise and respiratory depression.

Packing in any location may lead to infection.

Patient Education For patient education resources, see the Breaks, Fractures, and Dislocations Center, as well as Broken Nose.

The following precautions should be imparted to the patient: · Use nasal saline spray. · Avoid hard nose blowing or sneezing. · Sneeze with the mouth open. · Do not use nasal digital manipulation. · Avoid hot and spicy foods. · Avoid taking hot showers. · Avoid aspirin and other NSAIDs. The following simple instructions for self-treatment for minor epistaxis should be provided: · Apply firm digital pressure for 5-10 minutes. · Use an ice pack. · Practice deep, relaxed breathing. · Use a topical vasoconstrictor.

Originals and Papers Experience of botulinum toxin therapy in cervical dystonia, blepharospasm and hemifacial spasm 1

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Satish Chandra , Ritu Agarwal , Jayantee Kalita , Usha K Misra

Botulinum toxin-A (BTx-A) has been recommended for cervical dystonia (CD) blepharospasm and hemifacial spasm (BHS). There are only few reports from developing countries. This study evaluates the efficacy and safety of BTx in 11 patients with CD and 22 patients with BHS from a teaching hospital. The severity of CD was assessed by Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS) and BHS by Jankovic Disability Rating Score (JDRS). Patient’s satisfaction was graded on 0-100 scale. BTx was injected as per standard protocol. The response was noted at 4 weeks. The side effects were recorded. The median age was 46 years and 13 were females. The median duration of illness was 24 (range 4-120) months. In CD group, only 2 patients had torticollis and remaining had various combinations of retrocollis, anterocollis and laterocollis. More than 50% improvement was noted in 87% BHS and 70% CD patients. The improvement was related to the dose of BTx in CD but not in BHS. The response was related to severity but not to the duration of dystonia. 50% patients had mild transient side effect. More than two third patients of CD and BHS responded to BTx which was related to severity of dystonia.

[J Indian Med Assoc 2016; 114: 7-10]

Key words : Cervical dystonia, blepharospasm, hemifacial spasm, botulinum toxin, response, side effect.

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dystonia and class II evidence in hemifacial spasm and blepharospasm. The disadvantage of BTx-A is transient paralysis, wearing off therapeutic response, high cost and need of repeated injections. From South East Asia large series have reported in CD and BHS especially from Thailand, Singapore and Taiwan and only small series from India. In this communication we report our experience of BTx-A injection in focal and segmental dystonia.

ystonia is a neurological disorder characterized by involuntary repetitive and sustained muscle contraction producing twitching, squeezing or other movements and abnormal postures. The dystonia may be due to underlying degenerative, vascular, toxic, metabolic or infective causes or may be idiopathic.

Topographically dystonia may be generalized or focal.

The prevalence of primary dystonia reported from India is 43.9/100,000 population. The crude prevalence of primary focal or segmental dystonia reported from Italy is 127.4 per 1,000,000 populations. Blepharospasm is the commonest (prevalence 68.2), followed by cervical dystonia (prevalence 44.8). The average prevalence of hemifacial spasm is 7.4 per 100,000 population in men and 14.5 per 100,000 in women.

Dystonia not only causes functional disability but also cosmetic and emotional disturbances. Very few patients with dystonia have a good response to medical treatment, therefore, the role of botulinum toxin-A (BTx-A) has been explored in various focal dystonia for two decades. There is class I evidence about the efficacy of BTx-A in cervical

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MATERIALS AND METHODS

The patients with disabling focal and segmental dystonia despite optimal pharmacological therapy for 3 months were included in the study. The patients with doparesponsive dystonia, generalized dystonia, pregnancy, lactation, peripheral neuropathy, neuromuscular disorders, bleeding or coagulation disorders and renal and hepatic failure were excluded.

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A detailed medical history and clinical examination were carried out. The patients were evaluated neurologically including mental status by Mini-Mental State Examination (MMSE). Cranial nerve palsy, muscle power, tone, reflex and sensations were examined.

Disclaimer The information and opinions presented in the Journal reflect the views of the authors and not of the Journal or its Editorial Board or the Publisher. Publication does not constitute endorsement by the journal.

JIMA assumes no responsibility for the authenticity or reliability of any product, equipment, gadget or any claim by medical establishments/institutions/manufacturers or any training programme in the form of advertisements appearing in JIMA and also does not endorse or give any guarantee to such products or training programme or promote any such thing or claims made so after.

— Hony Editor

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Department of Neurology, Sanjay Gandhi Post Graduate Institute of Medical Sciences, Lucknow 226014 1 DM, Post-Doctoral Fellow 2 DM, Post-Doctoral Fellow 3 DM, Professor 4 DM, Professor & Head

The topography, type and severity of dystonia were noted. All the patients were videotaped and the severity of dystonia was scored. For cervical dystonia, Toronto West7

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EXPERIENCE OF BOTULINUM TOXIN THERAPY — CHANDRA ET AL

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ern Spasmodic Torticollis Rating Scale (TWSTRS) and for hemifacial/blepharospasm Jankovic Disability Rating Score were used. Overall satisfaction of the patients was rated on a 0-100 scale. Two different preparations of botulinum toxin type A (Dysport, Speywood Pharmaceuticals Ltd., UK and Botox, Allergan, Inc, Irvine, CA) were used as per availability of the drug. In practice, experience has shown that one unit of Botox is equivalent to 4 units of Dysport ; therefore we have used 1:4 conversion in botox to disport.100 U botox was diluted in 2ml or 500U dysport in 2.5 ml of normal saline and administered within few minutes. Cervical dystonia was categorized into laterocollis, anterocollis, retrocollis, and torticollis or combinations.

BTx-A injection for the treatment of cervical dystonia was injected in splenius capitis, semispinalis capitis, trapezius, levator scapulae, sternocleidomastoid and scalenus medius as per a fixed protocol. For blepharospasm injection sites included the upper medial and lateral eyelid margins, lower middle and lateral lid margins, and separate injections above the eyebrow. The muscles which contract most was injected to treat HFS and included orbicularis oculi, corrugator, frontalis, zygomaticus major, buccinators, and depressor anguli oris.

The patients were followed up at 1, 4, 12, 16 and 20 weeks The onset to peak time and duration of response were noted. The disability rating scale was used to objectively document the dystonia. The outcome of the injection however was based 4 week response.

Adverse events : Pain, hematoma, weakness, speech and swallowing difficulty, watering from the eyes and any other symptoms following injection were noted along with its severity and duration.

Investigations : Blood count, hemoglobin, ESR, blood sugar, serum creatinine, bilirubin, transaminase, calcium, phosphorus, albumin and ceruloplasmin were estimated. In cervical dystonia, cranial and cervical MRI was carried out.

Statistical analysis : The patients were classified into 2 major groups- 1) cervical dystonia (CD) and 2) blepharospasm and hemifacial spasm (BHS). The responses of BTx-A in CD and BHS groups were compared from baseline to 4 weeks and 12 weeks using one way analysis of variance. The comparison of efficacy of BTx-A in BHS versus CD was done by Mann Whitney U test. The variables were considered significant if 2 tailed p-value was <0.05. All the statistical tests were done using SPSS version 12.0.

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nia, 10 hemifacial spasm and 12 blepharospasm. The median duration of symptoms was 24 (range 4 to 120) months. These patients were on various anti-dystonia drugs which included anticholinergic in 28, baclofen in 10 and tetrabenazine in 9 patients for a median duration of 3 months. Cranial and cervical MRI was carried out in 15 and did not reveal any abnormality. In these patients, 54 sessions of BTx-A therapy were undertaken. The mean dose of botulinum toxin-A for blepharospasm and hemifacial spasm was 44 units, for cervical dystonia 112.5 units of botox or equivalent dysport.

Blepharospasm/Hemifacial spasm (BHS) : In this group 22 patients received 38 sessions of BTx-A. Their median age was 50.5 (28-78) years. Hemifacial spasm was present in 10 and blepharospasm in 12 patients. One patient with hemifacial spasm also had trigeminal neuralgia. The median duration of onset of action of BTxA was 4 (range 3 to 8) days and peak effect was achieved in the 4 week. More than 50% improvement was present in 86.9% patients and lasted for median duration of 16 weeks. One patient each in hemifacial spasm and blepharospasm did not improve. The side effects were noted in 50% sessions and included ptosis in 6, facial weakness in 5, lid swelling in 3, ptosis with facial weakness in 3, lid swelling with blurred vision in 1, ptosis and lid swelling in 1. The change in Jankovic dystonia scale is shown in Fig 1.

Case report : A 55 years old lady had left sided hemifacial spasm and trigeminal neuralgia for last 10 years. Her MRI head with fiesta sequence was normal. She received botulinum injection twice (35U-botox first time and 120 U dysport in second time) and response in hemifacial spasm was 70% in each injection and the duration of response was 12 months following first and 5 months following second injection. There was mild short lasting left sided facial palsy in first and no side effect in the second injection. There was no response in neuralgic pain.

Cervical dystonia (CD) : Eleven patients had CD whose median age was 41 (18-58) years and only 1 was a female. The duration of illness was 16 (range 4 to 72) months. The pattern of CD included torticollis + laterocollis th

DISCUSSION

In the present study, the response of BTx-A in BHS was 87% and that of CD in 70% patients. Various studies of hemifacial/blepharospasm have shown improvement in 80.3%-100% and in cervical dystonia up to 85% of patients. The effects of BTx-A depend on the correct identification of the affected muscle and optimal dose.

Cervical dystonia is a complex movement disorder with a group of overactive muscles resulting in various combinations of neck dystonia. Identification of muscle for injection in torticollis, retrocollis, laterocollis and anterocollis is essential. The muscles are targeted after observing the pattern of shift, tilt and rotation of neck. We have not used EMG for targeting the muscles. The dose of BTx-A in our study ranged between 17.5 and 50 (median 27.5) units of botox or equivalent dysport for a single muscle, which is within the recommended dose of 20-60 units for different cervical muscles. Anterocollis is associated with spreading of dystonia to other parts. In our study, 4 patients had anterocollis with head protrusion and 2 of them improved more than 50%. Only 2 of our patients had cervical torticollis and the remaining 9 patients had different combinations of neck dystonia which may be responsible for relatively poorer response to BTx-A. The suboptimal response may be due to antibodymediated resistance, vial-to-vial variability in response, variability in injection location and technique, change in pattern of muscle involve6, 9, 22-24

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OBSERVATION

Thirty three patients aged 18 to 78 (median 48) years were included; 20 of whom were males. All these patients had primary dystonia. Eleven patients had cervical dysto-

+ retrocollis in 2, anterocollis + laterocollis in 2, torticollis + retrocollis in 2, torticollis + laterocollis in 1, torticollis+ anterocollis in 2 and only torticollis in 2 patients. These patients received 16 sessions of BTx-A. 11 cases of CD received 16 sessions, two patients recovered completely after single session. In the remaining 9 patients, two patients received 3 sessions, 1 received 2 and remaining only 1. Repeated injections in most patients were not possible because of high cost. The improvement started after a median duration of 7.5 (5-20) days and lasted for a median duration of 20 (2 to 24) weeks in all except 5 sessions in which the improvement was less than 50%.

Two patients after 2 injections remained asymptomatic for 2 years. The side effects were noted in 50% sessions and included dryness of mouth in 2, dysphagia in 2, pain and swelling at the injection site in 2 and dizziness and uneasiness in 1 patient each. These side effects improved in 2 weeks. The change in TWSTRS score at different time points is shown in Fig 2.

Comparison : 87% patients in BHS responded compared to 70% in CD (P=0.006). The improvement was related to the dose of BTx-A in cervical dystonia (r=0.54, P=0.037) but not in BHS (r=0.07, P=0.94). The response was related to severity of dystonia (r=0.55, P=0.03) but not to duration of illness (r=-0.47, P=0.34).

Fig 1 — The sequential improvement following botulinum toxin in patients with blepharospasm and hemifacial spasm in Jankovic Disability Rating Score (JDRS)

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Fig 2 — The sequential improvement following botulinum toxin in patients with cervical dystonia in Toronto Spasmodic Torticollis Rating Scale (TSTRS)

ment and inadequate dose. Many of our patients with severe dystonia may have suboptimal dose because of financial reason. The selection of muscles was carefully done by experienced neurologist and all the patients had some degree of improvement, therefore, it is unlikely that correct muscles were not targeted for BTx-A injection.

The BTx-A was obtained from the company maintaining the cold chain. The better response in BHS could be attributed to better dosing of target muscles. The subjective improvement was more with cervical dystonia compared to hemifacial spasm as BTx-A is highly effective in controlling pain associated with cervical dystonia. Though the objective measurements in our study revealed a lower improvement in cervical dystonia but patients felt better subjectively which highlight the greater functional improvement.

Repeated injections were given to 8 patients with BHS and 3 patients with cervical dystonia. On repeated injections decay of response was noted is 2 sessions.

Antibodies against BTx-A may have led to secondary therapeutic failure particularly in patients who had previously received high doses of toxin and were reinjected within a short period. The prevalence of immuno-resistance ranges in different studies, but it is usually less than 10%. Currently available BTx-A has 5 ng of neurotoxin complex per 100 U; therefore the formation of neutralizing antibody and subsequent wearing off effects has significantly reduced. We had mild to moderate side effects in 50% of sessions lasting for about 2 weeks and the reported side effects of BTx-A in hemifacial spasm and that in cervical dystonia is also similar. The higher frequency of muscle weakness in our study in BHS may be due to smaller muscle bulk and conversion of botox to disport in 1:4. The complications in the literature were encountered in up to 63.4% in HFS and up to 72.7% in BS. BTx-A therapy is regarded as a symptomatic treatment and repeated injections are needed. Two of our patients with cervical dystonia after 2 injections remained asymptomatic for 2 years. Both these patients however had short duration of illness. The patient who had both trigeminal neuralgia and BHS, BTxA 28

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injection relieved blepharospasm but not the neuralgic pain. The efficacy of BTxA in trigeminal neuralgia has been reported in 72.7 to 100 % patients.

In our study, more than two-third patients with BHS and CD respond to BTx-A therapy with mild transient side effects. A better response in BHS was related to the shorter duration of illness.

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Poungvarin N, Devahastin V, Chaisevikul R, Prayoonwiwat N, Viriyavejakul A — Botulinum A toxin treatment for blepharospasm and Meige syndrome: report of 100 patients. J Med Assoc Thai 1997; 80: 1-8.

Poungvarin N, Devahastin V, Viriyavejakul A — Treatment of various movement disorders with botulinum A toxin injection: an experience of 900 patients. J Med Assoc Thai 1995; 78: 281-8 Poungvarin N, Viriyavejakul A — Botulinum A toxin treatment in spasmodic torticollis: report of 56 patients. J Med Assoc Thai 1994; 77: 464-70.

Suputtitada A, Phanthumchinda K, Locharernkul C, Suwanwela NC — Hemifacial spasm: results of treatment with low dose botulinum toxin injection. J Med Assoc Thai 2004; 87: 1205-11.

Jamora RD, Tan AK, Tan LC — A 9-year review of dystonia from a movement disorders clinic in Singapore. Eur J Neurol 2006; 13: 77-81.

Behari M, Singh KK, Seshadri S, Prasad K,Ahuja GK — Botulinum toxin A in blepharospasm and hemifacial spasm.

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Bhaumik S, Behari M — Botulinum toxin A—injection for cervical dystonia. J Assoc Physicians India 1999; 47: 26770.

Thussu A, Barman CR, Prabhakar S — Botulinum toxin treatment of hemifacial spasm and blepharospasm: objective response evaluation. Neurol India 1999; 47: 2069.

Gupta M, Singh G, Khwaja G — Botulinum toxin in the treatment of dystonias—a hospital based study. J Assoc Physicians India 2003; 51: 447-53.

Crowner BE — Cervical dystonia: disease profile and clinical management. Phys Ther 2007; 87: 1511-26.

Brin MF, Jankovic J, Comella C — Treatment of dystonia using botulinum toxin. In: Treatment of movement disorders.

Kurlan R, J.B. (Ed.) Lippincott Company 1995; 183-230.

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19 Camargo CH, Teive HA, Becker N, Baran MH, Scola RH, Werneck LC — Cervical dystonia: clinical and therapeutic features in 85 patients. Arq Neuropsiquiatr 2008; 66: 15-21 20 Pang AL, O’Day J — Use of high-dose botulinum A toxin in benign essential blepharospasm: is too high too much? Clin Experiment Ophthalmol 2006; 34: 441-4.

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34 Park YC, Lim JK, Lee DK — Botulinum a toxin treatment of hemifacial spasm and blepharospasm. J Korean Med Sci 1993; 8: 334-40.

35 Borodic GE, Acquadro MA — The use of botulinum toxin for the treatment of chronic facial pain. J Pain 2002; 3: 21-7.

36 Piovesan EJ, Teive HG, Kowacs PA, Della Coletta MV, Werneck LC, Silberstein SD — An open study of botulinumA toxin treatment of trigeminal neuralgia. Neurology 2005; 65: 1306-8.

37 Zúñiga C, Díaz S, Piedimonte F, Micheli F — Beneficial effects of botulinum toxin type A in trigeminal neuralgia. Arq Neuropsiquiatr 2008; 66: 500-3.

Originals and Papers Role of nasal endoscopy in the management of intractable epistaxis : our experience Dwaipayan Mukherjee1, Chiranjib Das2 Epistaxis is the most frequent emergency in Otorhinolaryngology. Intractable epistaxis is traditionally managed with multiple nasal packing. This is due to the covert areas of the nose. The availability of the nasal endoscope has been a boon to the otolaryngologist, since it not only helps in proper visualization of bleeding points, but also offers a direct mode of treatment to the area. The present work was undertaken to elicit the role of nasal endoscopy in detecting the site and the possible hidden causes of the intractable epistaxis and their treatment with comparison to conventional management. We conducted this prospective study in the Otorhinolaryngology department of a tertiary care hospital of West Bengal from March 2013 to February 2015. Cases where no apparent local or systemic cause of epistaxis could be detected, were included in this study. Patients who were not willing to give consent, patients with cardiovascular disease, bleeding disorder or receiving anticoagulant drugs were excluded. When no bleeding points were seen on anterior rhinoscopy and no systemic cause was found out, nasal endoscopy was performed with rigid nasal endoscopes. Patients were treated with endoscopic nasal cautery, selective nasal packing, polypectomy, excision of angioma, sphenopalatine artery cauterization, spurectomy or excision of angiofibroma. Among total 56 patients there were 15 female patients and 41 male patients. Most patients were in the age group of 21-30 years. The most common cause of epistaxis was bleeding point in the crevices of the lateral nasal wall (32%). All patients had successful control of epistaxis. Seven patients had anterior epistaxis in follow up period, which were managed with conventional treatment. Endoscopic examination of the nasal cavity has the advantage of providing better view of the nasal cavity and also aids in appropriate management of epistaxis based on their merit. It is a cost-effective and less invasive procedure. It has minimal morbidity and failure rates.

[J Indian Med Assoc 2016; 114: 11-3 & 20]

Key words : Intractable Epistaxis, Endoscopy, Selective nasal cautery, Selective nasal packing.

E

pistaxis is the most frequent emergency in Otorhino-laryngology, presenting with a prevalence of about 10% to 12% . Intractable epistaxis is a challenging problem due to the covert areas situated in the posterior and lateral part of the nose, which are difficult to access by anterior rhinoscopy . It is traditionally managed with multiple nasal packing and prolonged hospital stay and associated with significant patient morbidity and high health care costs . The availability of the nasal endoscope has been a boon to the otolaryngologist, since it not only helps in proper visualization of bleeding points, but also offers a direct mode of treatment to the area (Kennedy et al, 1985) .Nasal endoscopy enables targeted haemostasis of the bleeding points using electrocautery, direct pressure with miniature targeted packs, endoscopic ligation or cauterization of the

sphenopalatine artery, endoscopic ligation of ethmoidal arteries, cryotherapy and lasers .

Aims and objectives: The present work was undertaken to elicit the role of nasal endoscopy in 1. Detecting the site and the possible hidden causes of intractable epistaxis.

2. Treatment of epistaxis with comparison to conventional management.

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MATERIALS AND METHODS

We conducted this prospective study in the Otorhinolaryngology Department of a tertiary care hospital of West Bengal from March 2013 to February 2015.

Inclusion Criteria : (1) Cases where no apparent local or systemic cause of epistaxis was found.

Exclusion Criteria : (1) Patients who were not willing to give consent.

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MS (ENT) Associate Professor, KPC Medical College & Hospital, Kolkata 700032 2 MS (ENT) RMO cum Clinical tutor, Bankura Sammilani Medical College & Hospital, Bankura 722102 11

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J INDIAN MED ASSOC, VOL 114, NO 9, SEPTEMBER 2016

bromas were subjected to excision of the angiofibromas (2) Patients with cardiovascular disease.

(3) Patients with bleeding disorder or receiving and the specimens were sent for histopathological examination. anticoagulant drugs.

The patients were followed up in the post-operative period Based on the above criteria, 56 patients were selected at 1 week, 2 weeks, 1 month, 3 months, 6 months and 1 in our study. First of all vitals were checked. In severe year with endoscopy. epistaxis, first of all bleeding was controlled by nasal Results : packing and patient was made haemodynamically stable.

Most patients in our srudy were in the age group of 21When the bleeding was controlled, detailed history of the patient was taken followed by general and 30 years (Fig 1). There were 15 female patients and 41 otorhinolaryngology examination including thorough male patients. The most common cause of epistaxis was anterior rhinoscopy. Laboratory investigations were done bleeding point in the crevices of the lateral nasal wall to rule out any systemic causes for epistaxis. (32%), followed by bleeding ulcer in the crevices of lateral Investigations like haemoglobin estimation, total and nasal wall (16%), bleeding ulcer posterior to deviation of differential leucocyte count, platelet count, ESR, bleeding septum (14%), angioma in lateral nasal wall (13%), septal time, clotting time, prothrombin time, a PTT, renal spur (11%), congested polyp in middle meatus (9%), function tests, liver function tests, blood grouping were angiofifbroma just posterior to middle turbinate (5%) done routinely. When no bleeding points were seen on (Table 1). All of them were treated with endoscopic nasal anterior rhinoscopy and no systemic cause was found out; cautery, selective nasal packing, polypectomy, excision of nasal endoscopy was performed with nasal endoscopes. angioma, sphenopalatine artery cauterization, spurectomy 0 and 30 rigid nasal endoscopes of 2.7 mm and 4 mm or excision of angiofibroma (Table 2). All patients had diameter were used. Light cotton pledgets soaked with 4% successful control of epistaxis. No significant lidocaine was used. No adrenaline or prior nasal drops was complication or morbidity has been noted in the used and no extra pressure was exerted to avoid missing of postoperative follow-up period of 1 year. Seven patients the bleeding points. Then diagnostic nasal endoscopy was had anterior epistaxis in follow up period either due to nose undertaken in three steps. The first step consisted of an picking or nose blowing. All of them were managed with inspection of nasal vestibule, nasopharynx and inferior conservative treatment (Fig 2 & 3). nasal meatus. This was followed by an examination of sphenoethmoidal recess and superior meatus. Finally, an Table 1— Distribution of patients according to Endoscopic findings examination of middle meatus was done. If nasal mass was found, CT Endoscopic diagnosis Number of Percentage scan was done afterwards. patients When the bleeding point was Bleeding point in the crevices of lateral nasal wall 18 32% identified, endoscopic nasal cautery Bleeding ulcer in the crevices of lateral nasal wall 9 16% was done with insulated sucker cum Congested polyp in middle meatus 5 9% cautery or bipolar cautery. When the Angioma in lateral nasal wall 7 13% bleeding point was located in the Angiofibroma just posterior to middle turbinate 3 5% posterior part of nasal cavity, Bleeding ulcer posterior to deviation of septum 8 14% endoscopic sphenopalatine artery Septal spur 6 11% cauterization was done. If the bleeding point was not reachable for cauterization then selective nasal Table 2 — Distribution of patients according to treatment packing was done with gelfoam. If Endoscopic treatment Number of Percentage bleeding was coming from an ulcer, patients gelfoam was tightly packed between the ulcer and the nasal septum or the Endoscopic nasal cautery 10 18% turbinates. Patients having septal spur Endoscopic selective nasal packing 17 30% were undergone endoscopic spurectomy.

Endoscopic polypectomy 5 9% When congested polyp was found in the Endoscopic excision of angioma 7 13% middle meatus endoscopic polypectomy Endoscopic sphenopalatine artery cauterization 8 14% was done and sent for histopathological Endoscopic spurectomy 6 11% examination. The patients who Excision of angiofibroma 3 5% were diagnosed to have angiofi0

ROLE OF NASAL ENDOSCOPY IN THE MANAGEMENT OF INTRACTABLE EPISTAXIS — MUKHERJEE AND DAS 13

Fig 1: Distribution of patients according to age

Fig 2 — Clinical photograph of angioma over inferior turbinate

0

DISCUSSION conventional packing . Other less invasive procedures, The anterior and posterior rhinoscopies give a very such as selective nasal packing and endoscopic restricted view of the nasal cavity, resulting in poor cauterization of sphenopalatine artery could be done with visualization of certain areas. Due to this reason, the cause high efficacy rates . When selective cauterization of of the epistaxis many a times remains an enigma . Even if bleeding point is not feasible as in inferior meatus, high in the area is seen, it is difficult sometimes to apply direct the lateral nasal crevices and bleeding ulcer; a selective pressure to ensure stoppage of bleeding . Traditionally the nasal packing is done with gelfoam. It is a dissolvable treatment options were one or more of the following: nasal synthetic matrix that has a procoagulant effect. The packing, septoplasty, and ligation of external carotid gelfoam becomes nonadherent and it begins to dissolve in artery or internal maxillary artery. Apart from high failure a matter of weeks . The detection of angiofibroma on rates ranging from 26-52%, these procedures have endoscopy was a great source of relief as it would have significant morbidity . Conventional nasal packing is normally been missed. The small mass which was later associated with considerable discomfort, mucosal trauma confirmed on a CT scan, was situated in a site which could and morbidity due to hypoxia. The large size of the have not been possible to detect with anterior rhinoscopy. packing material exerts pressure not only on the point of We feel it is important to do a regular endoscopy in the bleeding but also on the normal mucosa. Sometimes haste vulnerable age group with epistaxis to see if the bleeding is instrumentation may cause a fresh bleed . External carotid artery ligation is associated with risk of damage to not due to an angiofibroma. There are many other methods hypoglossal and vagus nerve . The transantral approach to that can be applied for the treatment of epistaxis; internal maxillary artery may cause damage to like lasers, cryotherapy, endoscopic ligation of nasolacrimal duct or infraorbital nerve . Recently t h e s p h e n o p a l a t i n e a r t e r y a n d e t h m o i d a l angiography and embolization of bleeding vessels have arteries. Due to lack of facilities, these procedures been added to treatment option. But it requires expertise of an experienced interventional radiologist, which is not uniformly available.

Moreover, it is also associated with serious neurological complications .Regular use of nasal endoscopy during the last decade amplified the knowledge on the aetiology and treatment of epistaxis. The bleeding source inside the nasal cavity could be more easily and accurately identified. Cauterization of the bleeding point, which was previously limited to anterior portions of the nasal cavity, could be applied to posterior regions with the advent of endoscope . Moreover, nasal endoscopy is the only way for Fig 3 — Clinical photograph of bleeding point over middle turbinate preventing trauma to the normal mucosa due to 2

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Practitioners' Series

INCIDENCE OF URINARY TRACT INFECTION AND UROLOGICAL SYMPTOMS — NISHA ET AL 15

Incidence of urinary tract infection and urological symptoms in depot-medroxyprogesterone users B Nisha1, Sunita Malik2, Jagdev Kaur3, Archana Aggarwal4

Progesterone due to its facilitatory effect on the b-receptors present in the urinary system may decrease the tone and peristalsis of urethra and ureter, causing dilatation of the urinary collecting system, decreased flow, relative stasis and defective clearance of the bacteria which can lead to urinary tract infection in women using Depot-medroxyprogesterone acetate. The other contributory factor for urinary tract infection seen recently is that use of progesterone may decrease the production of human beta defensin-2 receptor in the vaginal epithelium and may increase the susceptibility to urinary tract infection.

In this study, 50 cases who opted for depot-medroxyprogesterone acetate for contraception after medical termination of pregnancy were compared with 50 controls who underwent concomitant tubal ligation along with medical termination of pregnancy after 3 months for urinary symptoms and urinary tract infection.

None were given antibiotic after the procedure. Women found to have urinary tract infection were treated according to antibiotic sensitivity report. The rate of urinary infection (p -0.031) and urological symptoms in the study group were higher than in the control group. Escherichia coli was the most common microorganism that caused urinary tract infection and second was Staphylococcus aureus. We recommend routine screening for presence of urological symptoms, urinary tract infection and asymptomatic bacteriuria in women using depot-medroxyprogesterone acetate in order to avoid complications of untreated infection.

[J Indian Med Assoc 2016; 114: 14-6]

Key words : Urinary tract infection, depot-medroxyprogesterone acetate, urological symptoms.

D

epot-medroxyprogesterone acetate (DMPA) has been the most widely studied injectable contraceptive. Since its introduction many studies have been done to evaluate its benefits and adverse effects. Among them urinary tract infection (UTI) is a recently observed short term effect.

It has been seen that progesterone has a facilitatory effect on the b-receptors present in the urinary system thereby, decreasing the tone and peristalsis of urethra and ureter due to its relaxant effect on the smooth muscles.

These features contribute to UTI due to the dilation of the urinary collecting system, with decreased flow, relative stasis and defective clearance of the bacteria.

Progesterone also decreases the vascularity of the urinary system by counter acting the effect of estrogen, which helps in preparation of the tissue to combat infection as seen in animal experiment by Batra S et al .

Human beta defensin-2 receptor (HBD-2) plays an

important role at the innate defense on genitourinary tract.

This receptor remains unaffected during the normal state.

It is seen that during infection estrogen increases the production of this receptor whereas progesterone decreases it . Hence, lack of estrogen during menopause or use of progesterone based oral contraceptive in sexually active women may influence the production of HBD-2 receptor in vaginal epithelium and may increase susceptibility to bacterial vaginitis or recurrent UTI. In addition to the above feature it has also been seen that progesterone dominance to some degree may also be responsible for increased incidence of genuine stress incontinence as progesterone counteracts the effect of estrogen .

Based on the above observations, due to the possible effect of progesterone on urinary tract, the present study was undertaken to determine whether DMPA increases the rate of urinary tract infection in those who received this drug for contraception after medical termination of pregnancy (MTP).

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Department of Obstetrics and Gynaecology, Vardhman Mahavir Medical College and Safdarjang Hospital, New Delhi 110029 1 MBBS, MD, Senior Resident 2 MBBS, MD, FICOG, Professor and Consultant 3 MBBS, MD, Associate Professor of Microbiology 4 MBBS, DMRD, Consultant Radiodiagnosis

MATERIALS AND METHODS

A pilot study was conducted in the family welfare 14

wing, in the Department of Obstetrics and Gynecology, Safdarjang Hospital, New Delhi, India, to observe the effect of DMPA on the urinary tract, which was compared with matched (based on age, gravidity, socioeconomic and educational status) control. However, expecting the higher dropout, more than 100 women were recruited and 2 groups were formed.

Study Group — 78 postabortal women who had undergone MTP with suction and evacuation method, received injection Depot-medroxyprogesterone acetate 150mg intramuscularly before discharge from the hospital. These women formed the study group. Out of 78 subjects, 23 were lost to follow up, 5 were excluded (4 subject had bacterial growth in urine culture at 0 month and 1 took antibiotic) and the rest 50 subjects were compared with matched control.

Control Group — 69 postabortal women who had undergone MTP with concomitant tubal ligation formed the control group. Out of 69 subjects, 15 were lost to follow up, 4 were excluded (3 took antibiotic 1 had bacterial growth in urine at 0 month), and the rest 50 subjects were compared with the study group.

The inclusion criteria were: (1) Apparently healthy women aged between 20-40 years, (2) no medical or surgical illness, and (3) intended to come for follow up at 3 months.

The exclusion criteria were: Any history of (1) UTI more than twice per year, (2) presence of UTI, urinary stone, urinary tract anomaly, or asymptomatic bacteriuria, urological symptoms (3) diabetes mellitus, hypertension, (4) use of any hormonal contraceptive or intrauterine device in the past 3 months, (5) presence of vaginitis or abnormal vaginal discharge, and (6) consumption of any antibiotic in the past 3 months.

Preliminary contact was made on the day of MTP and informed consent was taken. Questionnaires were used to evaluate urological symptoms and relevant history. Patient then immediately received injection DMPA, 150 mg intramuscular after MTP by suction and evacuation method. No antibiotic was given in both groups.

Mid-stream urine sample was collected on the day of procedure and sent for routine microscopy examination and bacteriological culture and sensitivity test using MacConkey Agar media.

However, women showing UTI and asymptomatic bacteriuria were appropriately treated as per the antibiotic sensitivity test and were excluded from the study.

At 3 months, women in the two groups were followed and evaluated for urological symptoms such as frequency (ie, daily void >8 times), burning micturition, urinary incontinence (urge/stress) and UTI. Bacterial count > 105 org. / ml. was considered as significant.

Pearson Chi-Square and Fisher-exact test were used to compare parameters between the two groups. Value of p < 0.05 was considered significant.

RESULT

At the beginning of study there were no significant differences in age, educational status, occupation and gestational age.

At 3 months, the effect of DMPA on the rate of UTI and urological symptoms are shown in Table 1.

The results showed that rate of UTI in the study group (p - 0.031) were significantly higher than in the control group. Frequency of urological symptom though not statistically significant but were found be higher in the DMPA users. In the control none had increased frequency of micturition. Urinary incontinence was not reported by any one.

The most common organism responsible for causing UTI was Escherichia coli (5 cases out of 8). The other micro-organisms were Staph aureus (2 cases out of 8) and Klebsiella (1 cases out of 8).

Asymptomatic bacteriuria (ASB) — At the beginning of the study, among 117 subjects significant bacteriuria was found in 4 who were excluded. However, these 4 subjects did not report any urological symptoms, showing prevalence of ASB as 3.42% which is close to the value seen in another study done on pregnant population belonging to same geographical area 4.34% . At 3 months out of 50 subjects of study group significant bacteriuria without any urological symptoms were found in 2 subjects, showing incidence of ASB with DMPA as 4%.

10

DISCUSSION

We have seen in the present study that women using DMPA are predisposed to UTI, possibly due to the effect of progesterone on muscle tone, peristalsis of the ureter and also urinary vasculature. ASB, UTI, increased bladder capacity, hydroureter, increased bladder capacity, and urinary incontinence have been seen during pregnancy, due to the possible effect of progesterone on the smooth muscle of the urinary system .

With this background that the progesterone causes dilatation of ureter, high dose progesterone have shown beneficial effect in patients with ureteral stone and 11-13

14,15

Table 1 — Comparison of the parameters in the two group after 3 months Study Group Control group n (%) n (%) Urinary symptoms Increased frequency Burning Micturition Urinary Infection

6 (12) 4 (8) 2 (4) 8 (16)

1 (2) 0 1 (2) 1 (2)

pvalue 0.112 0.117 1.000 0.031

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J INDIAN MED ASSOC, VOL 114, NO 9, SEPTEMBER 2016

benign prostatic hypertrophy in men. Raz and colleagues have reported that oral medroxyprogesterone acetate 20 mg daily exacerbated stress incontinence in 60% of women treated with corresponding changes in urethral pressure.

Increased incidence of bacteriuria has also been reported in women taking oral contraceptives, especially those with high dose progesterone content .

S Ziaei et al has reported significantly higher rate of UTI and urological symptoms in the DMPA users than the control. As so far only one study of this kind has been published, its comparison with present study is shown in Table 2.

The comparison between two studies after 3 months of DMPA use shows that (i) DMPA may be responsible for urological symptoms and UTI. (ii) In our study urinary incontinence was not seen in any subject. (iii) The most common organism of urinary infection was Escherichia coli in both the studies.

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medroxyprogesterone acetate in order to avoid complications of untreated infection 1

2 3

20

CONCLUSION

DMPA, although quite safe and effective method of contraception, may be responsible for increase in the frequency of urinary infection. Hence, women who have any history of current or recurrent UTI, UTI during pregnancy, presence of urological symptoms (such as frequency and burning micturition), urinary stone or urinary tract anomaly or diabetes mellitus should be offered an alternative method of contraception.

We recommend routine screening for presence of urological symptoms, urinary tract infection and asymptomatic bacteriuria in women using depot-

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Table 2 — Comparison of the parameters in two studies after 3 months of DMPA use Parameters

S Ziaei et al (2003)

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Present Study 17

%

P

%

P 18

Urological symptoms Frequency Burning Incontinence

<0.000 22 0 7.5

8 4 0

0.112 0.117 1.000

19

20 Urinary Infection

5

0.018

16

0.031

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Batra S and Iosif S — Progesterone Receptors in the female lower urinary tract. J Urol 1987; 138: 1303-4.

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Mikkilsen AL, Meyhoff HH, Lindahl F, Christensen J — The effect of hydroxyprogesterone on ureteral stones. Int Urol Nephrol 1988; 20: 257-260.

Onu PE — DMPA in the management of BPH. Eur Urol 1995; 28: 229-35.

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Ziaei S, Ninavaei M, Faghihzadeh S — Urinary Tract Infection in the Users of Depot-Medroxyprogesterone Accetate. Acta Obstet Gynecol Scand 2004; 83: 909-911.

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Dr. Santanu Sen Hony. Secretary, JIMA Hony. Secretary, IMA Bengal

A comparative study between skin sutures and skin staples in abdominal surgical wound closure 1

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Chandrashekar N , Prabhakar GN , Vivek PO , Shivakumarappa GM , Fahad Tauheed

The skin stapling devices have revolutionized surgery for the purpose of rapid closure of abdominal wounds. However, staples have their own drawbacks. In view of this, this prospective study has been undertaken to highlight the outcomes of closure by staples and sutures with respect to speed of closure, cost effectiveness and post operative wound dehiscence, acceptance of scar and post operative pain. This is a prospective hospital based study conducted in our hospital from October 2009 to September 2011 involving a total of 200 patients who underwent abdominal surgery both on an emergency and elective basis.

Results were analyzed and compared with previous studies. It has been found that the use of staples in abdominal surgical wound closure gives faster speed of closure, less postoperative pain, and better cosmetic results. Staples, however, are costlier, and when used in emergency cases, associated with higher rates of wound dehiscence and a less acceptable scar.

[J Indian Med Assoc 2016; 114: 17-20]

Key words : Skin staples, nylon suture material, post operative wound infection, operative scar.

A

ny surgical intervention will result in a wound in order to get access to and deal with the underlying pathology. In this situation, the surgeon’s task is to minimize the adverse effects of wounds, remove or repair the damaged structures and harness the process of wound healing to restore function.

The principle aims of tissue repair of surgical skin incisions are rapid acquisition of strength and minimum tissue damage, with minimum inflammation and a good scar. Many factors including the choice of suture materials and its placements influence these aims; of particular relevance is the accurate co-optation of dermal edges; eversion or inversion leads to sub optimal healing.

For many years sutures have been used to approximate the skin edges, and also to hold the cut tissues together until the wound has healed sufficient enough as to be self supportive. Throughout antiquity many materials have been used to approximate the skin edges. Suture technology and suture sterilization have kept pace with advancement in surgical techniques and provided the surgical fraternity a wide range of sutures in different size as fine as 30 microns. Now the surgeon has at his disposal a wide variety of suture materials like natural and synthetic, non absorbable and absorbable, monofilament to poly filament. However, sutures have the disadvantage

of consuming more time in applying and with a cosmetically inferior scar. The use of other methods to approximate the wound edges like stapling devices, glue or adhesive tapes have becoming more popular of late to overcome these disadvantages. At present, cost effectiveness is debatable.

MATERIAL AND METHODS

This proposed study was conducted at Sree Siddhartha Medical College, Hospital and Research Centre, Tumkur, Karnataka, over a period of 2 years from October 2009 to September 2011.

The study included 200 patients who underwent abdominal surgeries, including both emergency and elective surgical procedures. All patients with abdominal surgical wounds were included, but excluded from the study were patients with skin infection, patients with post burst abdomen, wounds secondary to burns, all patients with anemia and diabetes. The patients were allotted alternately into two groups of 100 each. In group 1, abdominal wound closure was done (using 2-0/3-0 monofilament nylon) with mattress sutures. In group 2, abdominal wounds were closed with surgical skin stapling device.

The outcome was measured in terms of: (1) Speed of closure.

(2) Cost effectiveness.

(3) Post operative pain.

(4) Post operative wound dehiscence.

(5) Acceptance of the scar.

Results were analyzed and compared with the previous studies.

Department of General Surgery, Sree Siddhartha Medical College, Hospital & Research Centre, Tumkur 572107 1 MS, FIAGES, FAMS, Associate Professor 2 MS, FIAGES, Professor 3 MS, Assistant Professor 4 MS, Professor and Head 5 MBBS, Postgraduate (MS) student 17

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A COMPARATIVE STUDY BETWEEN SKIN SUTURES AND SKIN STAPLES — CHANDRASHEKAR ET AL

J INDIAN MED ASSOC, VOL 114, NO 9, SEPTEMBER 2016 RESULTS

Speed of closure — Speed of closure was faster with staples. The average duration of closure was 8minutes in suture group and 60 to 70 sec in staples group. The abdominal wound closure in case of staples group was hence 5 to 7 times faster in comparison to suture group (on an average the length of abdominal surgical wound was 12 to 15 cms) (Fig 1).

Postoperative pain — Post operative pain assessment was done using visual analog score. Immediate post operative pain scores were higher with the use of sutures as compared to staples in both elective and emergency cases.

56% of elective cases in whom suture closure was used had a pain score of 3 or higher, compared to only 17.2% in staple closure group. For emergency cases, the figures were 84% for the sutures group and 14.3% for the staples group (Fig 2).

Post operative wound infection and dehiscence — Post-operative wound infections were marginally higher (almost comparable) in staples group (13.7%) compared to sutures group (12%) in elective cases; but significantly higher in staples group (38.09%) compared to sutures group (16%) in emergency cases (Fig 3).

Cosmetic results — Cosmetic results were better with staples in elective cases. However when staple closure was used in emergency cases, post operative wound infection and dehiscence were higher and hence healing was with secondary intention; the resultant scar was thick and cosmetically less acceptable. (scars were compared after 3 months of surgery) (Figs 4-7).

Cost — The average median cost in suture group was Rs 100/- and in staple group Rs 310/-. Staple closure was hence thrice as costlier as sutures.

DISCUSSION

Wound closure is a vital step for producing a healthy and strong scar and also for ensuring aesthetically pleasing appearance.

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study of 20 pa tients concluded that the use of skin staplers speed up closure by 80%, with better cosmetic results .

John T Kanagaye, Cheryl W Vance, Linda Chan, and Nancy Schonfeld (1997) at the Children hospital, Los Angeles, USA, reported that staple closure was safe, rapid and cost effective and resulted in a cosmetically acceptable scar .

Iavazzo et al (2011) from a meta analysis of randomized controlled trials comparing sutures with Fig 5 — Abdominal surgical wound closure by staples for the management Fig 4 — Abdominal surgical wound closure by using disposable staplewounds gun using non-absorbable suture material of surgical reported that staples were faster, with (2-0 monofilament nylon) fewer wound infections but associated with more pain additional expenses, as the cost was forty seven times compared with sutures. Cosmetic results were comparable . higher than that of the suture with Dermalon .

In our present prospective study comparing skin Meiring et al (1982) reported slightly better cosmetic sutures and skin staples for abdominal surgical wound results in a group of 40 patients undergoing laparotomy closure, it has been noted that though the method of with an 80% in time saving. They also concluded that the closure by the staples was significantly faster in final cost of the stapler was crucial for selecting the comparison to sutures, it is met with certain drawbacks such as post operative infection with wound gaping , and method . subsequent prolonged duration of hospital stay posing Gatt et al (1985) concluded from a controlled trial of economic burden on patients and doubling the cost factor staples for wound closure that the speed and convenience in emergency infective cases as compared to clean elective of the skin staples outweigh the extra cost . cases.

In elective cases, the scar was found to be cosmetically Lubowski and Hunt (1985) consider proximate staple superior with better patient acceptance in staples group in closure a suitable and faster method for vertical abdominal comparison to sutures group. But in emergency wound compared to sutures .

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Fig 2 — Duration of closure by using staples (in seconds)

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Surgical stapling was developed in 1908 by a Hungarian surgeon, Humer Hultl . The original instrument was massive by today’s standards weighing 7.5 pounds.

Modifications performed by Von Petz provided a lighter and simpler device, and in 1934 Fredrick of Ulm designed an instrument that resembled the modern linear stapler. In 1958, Ravich, refined the instruments to their current state and wide spread use today .

Staplers are made up of stainless steel. They are virtually inert. They have uniform shape and constant staple depth providing even wound tension. Rectangular shape design minimizes the trauma and minimizes the tissue compression thereby causing minimal tissue reaction and trauma and leads to wound healing with minimum scar.

The development of disposable skin staplers has made this method of wound closure an increasingly popular technique. Skin staplers are quick and easy to use and numerous studies have confirmed the speed and efficacy of staping compared with suture repair.

Eldrup et al (1981) analysed 137 patients undergoing abdominal or thoracic surgeries, and concluded that the main advantage of using staples was the time saved, as closure with mechanical suture took one third of the time required for the conventional method. On the other hand closure with staples resulted in the major disadvantage of 1

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Stockey and Elson (1987) compared the results of closure with staple and nylon sutures found a higher incidence of inflammation, discomfort on removal and spreading of the healing scar with staples. The only advantage of staples was speed of wound closure .

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Ranabaldo and Rowe-Jones (1992) compared staple with subcuticular sutures in 48 patients undergoing laparotomy and concluded that the difference in time was significant, nevertheless, the cost was five times greater with staples .

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Fig 1 — Duration of closure by using sutures (in minutes)

Fig 3 — Postoperative wound infections for elective and emergency cases in sutures and staples groups

Luiz R Medina dos Santos et al (1995) in their

Fig 6 — Abdominal surgical wound scar after 6 week follow-up in staple closure

Fig 7 — Abdominal surgical wound scar after 6 week follow-up in suture closure

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J INDIAN MED ASSOC, VOL 114, NO 9, SEPTEMBER 2016

cases, due to post operative wound dehiscence the patient acceptance of the scar was poorer in staples group as compared to sutures group.

The sutures were more cost effective compared to staples; the immediate post operative pain was comparatively higher in the sutures group.

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CONCLUSION

In our present study, we conclude that though the staples cost higher in comparison to sutures, the wound closure time was much faster which was statistically significant and in agreement with the literature reviewed.

This has a great impact on post operative recovery as the patient can be weaned off from the anaesthesia faster and thereby reducing overall operating time and hence decreasing post operative morbidity and mortality.

In terms of patient acceptance of scar, we conclude that staples have good acceptance in clean elective cases and they are met with significant post operative wound infection in contaminated and emergency cases. Since staples are easier and faster to apply compared to sutures, the study showed that staples form an important surgical armamentarium for wound closure for elective and clean cases.

REFERENCES 1 Baker RS, Foote J, Kemmeter P, Brady R, Vroegop T, Serveld M — The science of stapling and leaks. Obes Surg 2004; 14: 1290-8.

2 Ravitch MM, Lane R, Cornell WP, Rivarola A, McEnany T —

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Closure of duodenal, gastric and intestinal stumps with wire staples: experimental and clinical studies. Ann Surg 1966; 163: 573-9.

Eldrup J, Wied U, Andersen B — Randomised trial comparing Proximate stapler with conventional skin closure.

Acta Chir Scand 1981; 147: 501-2.

Meiring L, Cilliers K, Barry R, Nel CJ — A comparison of a disposable skin stapler and nylon sutures for wound closure.

S Afr Med J 1982; 62: 371-2.

Gatt D, Quick CR, Owen-Smith MS — Staples for wound closure: a controlled trial. Ann R Coll Surg Engl 1985; 67: 318-20.

Lubowski D, Hunt D — Abdominal wound closure comparing the proximate stapler with sutures. Aust N Z J Surg 1985; 55: 405-6.

Stockley I, Elson RA — Skin closure using staples and nylon sutures: a comparison of results. Ann R Coll Surg Engl 1987; 69: 76-8.

Ranaboldo CJ, Rowe-Jones DC — Closure of Laparotomy wounds: skin staples versus sutures. Br J Surg 1992; 79: 1172-3.

Luiz R Medina dos Santos, Carlos AF Freitas, Flavio C Hojaij, Vergilius JF Araújo Filho, Claudio R Cernea, Lenine G Branda, Alberto R Ferraz — Prospective study using skin staplers in head and neck surgery. Am J Surg 1995; 170: 451-2.

John T Kanagaye, Cheryl W Vance, Linda Chan, Nancy Schonfeld — Comparison of skin stapling devices and standard sutures for pediatric scalp lacerations: A randomized study of cost and time benefits. J Paediatrics 1997; 130: 808-13.

Iavazzo C, Gkegkes ID, Vouloumanou EK, Mamais I, Peppas G, Falagas ME — Sutures versus staples for the management of surgical wounds: a meta-analysis of randomized controlled trials. Am Surg 2011; 77: 1206-21.

Preliminary Report Rhinosporidiosis of different organs — a study of 57 cases with review of literature Palash Kumar Mandal1, Nirmal Kumar Bhattacharyya2, Sumedha Dey3, 4 5 6 Pranab Kumar Biswas , Subrata Mukhopadhyay , Dibyendu Gautam Rhinosporidiosis is a chronic granulomatous disease caused by Rhinosporidium seeberi, an aquatic parasite of class mesomycetozoa. It affects most commonly nasal cavity and rarely aerodigestive tract, tracheobronchial tree, conjunctiva, skin, penis, parotid duct and bone and mostly present as polypoid, reddish,friable mass mimicking neoplastic mass. As the lesion is increasing in our hot climate, our aim of study is to show how different organs including nasal cavity were affected by the infection. Total 57 cases were studied in last five years in which 45 cases were seen as nasal mass, 4 cases were in conjuctiva, 4 cases were in oropharynx and one case each in larynx, skin, anus and penis. The formalin-preserved specimens appeared as friable, polypoid mass and histopathology of each case revealed classical microscopic appearance of rhinospoidiosis. To exclude from other fungal infections like coccidioidomycosis we performed periodic acid Schiff (PAS) stain and Gomori’s methenamine silver stain besides routine stain. We collected occupational history of each patient and most of them were farmers or cattle shed workers. Similar reports are available in international literature which showed that most cases of rhinosporidiosis were seen in nasal cavity and rarely other sites like conjunctiva, skin, bone, anus and penis were also involved. Surgical removal followed by dapsone therapy is the mainstay of treatment and recurrence is rare. Increasing incidence of this rare infection indicates that hot ,humid weather of our country and poor hygiene of cattle shed workers are the main culprit. So, health administration should take necessary steps to minimize this infection.

[J Indian Med Assoc 2016; 114: 21-4]

Key words : Rhinosporidiosis, Nose, Extranasal sites.

(Continued from page 13)

were not done in our hospital.

Conclusion : Though anterior and posterior rhinoscopy is done routinely as a part of clinical examination of epistaxis, they have their own limitations. Endoscopic examination of the nasal cavity has the advantage of providing better view of the nasal cavity and also aids in appropriate management of epistaxis based on the merit. It is an effective and less invasive procedure. It has minimal morbidity and failure rates. It also has an added advantage of preventing damage to nasal mucosa by blindly packing and instrumentation. It reduces need for prolonged hospitalization.We conclude that if the bleeding source is not identified by anterior rhinoscopy, nasal endoscopy is mandatory in management of epistaxis.

REFERENCES 1 Rodrigo P Santos, Fernando D Leonhard, Ricardo G Ferri, Luiz C Gregorio — Endoscopic endonasal ligation of the sphenopalatine artery for severe epistaxis, Brazilian Journal of Otorhinolaryngology, 2002; 68: edition 4. 511-4.

2 Safaya A, Venkatachalam VP, Chaudhary N — Nasal Endoscopy-Evaluation in Epistaxis, Indian Journal of Otolaryngology and Head and Neck Surgery. 2000; 52: 1336.

3 Rudmik L and Smith T — Management of intractable sponta

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neous epistaxis. American Journal of Rhinology & Allergy.

January–February 2012, 26: 55-60.

Babu M, Gowda B, Satish HS — Role of Rigid Nasal Endoscopy in the Diagnosis and Management of Epistaxis.

IOSR Journal of Dental and Medical Sciences. 2014, 13: 405.

Schaitkin B, Strauss M, Houck JR. Epistaxis — Medical versus Surgical therapy, Comparison of efficacy, complications and economic considerations. Laryngoscope 1987; 97: 1392-5.

Shaw CB, Wax MK, Wetmore SJ — Epistaxis: a comparison of treatment. Otolaryngol Head Neck Surg 1993; 109: 60-5.

Thakar A, C J Sharan — Endoscopic sphenopalatine artery ligation for refractory posterior epistaxis. Indian Journal of Otolaryngology and Head and Neck Surgery, 57: 2005, 2158.

Spafford P, Durham JS — Epistaxis: efficacy of arterial ligation and long term outcome. J Otolaryngol 1992; 21: 2526.

Siniluoto TM, Leinonen AS, Kartunnen AK — Embolization for management of posterior epistaxis. Arch Otolaryngol 1993; 119: 837-41.

Metson R, Hanson DG — Bilateral facial nerve paralysis following arterial embolization for epistaxis. Otolaryngol Head and Neck 1983; 91: 299-301.

O’dnnell M, Robertson G, Mcgarry GW — A new bipolar diathermy probe for the outpatient management of adult acute epistaxis. Clin Otolaryngol 1999; 24: 537-41.

Bhatnagar RK, Berry Sandeep — Selective Surgicel Packing for the Treatment of Posterior Epistaxis Ear, Nose and Throat Journal 2004; 83: 633-4.

R

hinosporidiosis is a chronic granulomatous disease caused by fungus –like organism known as Rhinosporidium seeberi, an aquatic parasite of class mesomycetozoa . The lesion is grossly characterized by reddish, polypoidal or sessile and friable mass. It affects most commonly different parts of nasal cavity (70%) such as anterior part of nasal septum, vestibule and nasopharynx because of affinity of the organism for mucous membrane of nasal cavity and nasopharynx.

Besides nasal cavity, it may rarely affect lower aerodigestive tract, tracheobronchial tree, lips, uvula, conjunctiva, penis, parotid duct and bone .

The infection is more commonly observed in

hot tropical climates of the endemic zones such as India and SriLanka . It usually affects males of age group of 15 years to 40 years. The diagnosis in nasal cavity is easier, but delayed in extranasal sites which often clinically mimick neoplasm. Besides classical histopathological appearances, occupational and personal history are also needed for diagnostic corroboration. Aim of this study is to show how different organs may be affected by Rhinosporidium seeberi and increasing incidence of it in Kolkata.

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MATERIALS AND METHODS

The study is a retrospective one and done in department of Pathology, Medical College, Kolkata from July 2007 to June 2012. The formalin preserved specimens appeared as friable, lobulated mass (Fig 1) which were routinely processed and stained by Haematoxylin and Eosin (H&E), Periodic acid Schiff (PAS) and Gomori’s methenamine silver. Total of 57 cases were retrieved within the study period which included mostly nasal masses and fewer of other different organs (Table 1).

Microscopically each case showed thick-walled, rounded cysts called sporangia containing endospores.

Sporangium measures 0.5mm in diameter while each endospore measures 6-7 micron. (Fig 2).

2,3

Department of Pathology, North Bengal Medical College, Darjeeling 734012 1 MD (Pathol), Associate Professor 2 MD (Pathol), Associate Professor, Department of Pathology, Medical College & Hospital, Kolkata 700073 3 MD (PGT), Department of Pathology, Medical College & Hospital, Kolkata 700073 4 MD (Pathol), Professor, Department of Pathology, Medical College & Hospital, Kolkata 700073 5 MS (ENT), Professor and Head, Department of ENT, Medical College & Hospital, Kolkata 700073 6 DMRT, MS (Gen Surg), Professor, Department of General surgery, Medical College & Hospital, Kolkata 700073 21

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J INDIAN MED ASSOC, VOL 114, NO 9, SEPTEMBER 2016

RHINOSPORIDIOSIS OF DIFFERENT ORGANS — MANDAL ET AL

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DISCUSSION

RESULTS

Out of 57 total cases, 45 cases are seen in nasal cavity, 4 cases were in conjunctiva, 4 cases in oropharynx, one case each in larynx, skin, penis and anal region. All cases except one were observed in males. The only female patient had it in nasal cavity. The clinical features of nasal rhinosporidiosis commonly were nasal obstruction, epistaxis, polyp. Occupational history revealed that most patients were farmers, 6 had domestic cattles in their houses all used to bath in community ponds. In extranasal sites, most patients were seen to be affected by autoinoculation from nasal mass. The rhinosporidiosis in extranasal sites presented as polypoidal, friable mass which bled on touch and so excised as suspicious neoplastic lesions but histopathology revealed the nature of lesion.

Rhinosporidiosis reviewed in 1998, has been known for more than a century since its first description in Argentina and was described as a chronic granulomatous disease with frequent recurrence after surgery and occasional dissemination from the initial focus which is most commonly seen in upper respiratory sites . It occurs universally, although it is endemic in south Asia, notably southern India and Sri Lanka.

The literature on rhinosporidiosis and on the different morphological stages of R.seeberi contains many synonymous, overlapping terms. For example, the morphological element for which the proposed term is “ Electron- dense body” were earlier termed variously as spherule, electron-dense circular structure, protrusion of 5

Table 1 — Showing Nasal Masses and Fewer of Other Different Organs Site

No

Sex

Clinical Features

Spread

Diagnosis

Treatment

Follow-up (3yrs) Recurrence

Cause of

Nasal Cavity

45

44 M,1F

Nasal obstruction, epistaxis, polyp

38 farmers, 6 had cattles in house, all used to bathe in community pond

Histopathology of the excised mass

Excision of the mass f/b electrocauterisation of the base

10 lost follow-up, 32 had no recurrence, 3 presented with similar symtoms

Seeding of endospores during surgery

Conjunctiva 4

M

Reddish polyp over palpebral conjunctiva, epiphora, Photophobia

History of trauma from dust & vegetable particles

Clinically suspicious. confirmed by HPE of the excised mass

do

none

—

Oropharynx 4

M

Foreign body sensation in throat, dysphagia

All had associated polyp in their nose histology of excised mass

Clinical features & history was suggestive, confirmation by

do

none

—

Hoarseness of voice, h/o nasal polypectomy 3yrs ago under GA

Orotracheal intubation

confirmation by histology of excised mass

Polypoid leison on back recurrence at the same site after removal.

abrasion, FNAC needle injury in c/o recurrence.

Difficulty in micturation, bleeding from meatus, growth in the distal urethra

Autoinoculation from nasal mass

Growth in the anal region which bleeds on touch.

B/L nasal polyps

Autoinoculation from nasal mass

Larynx

Skin

Penis

1

1

1

Anal Region 1

M

M

M

M

Excision of the mass f/b electrocauterisation+ Dapsone

none

—

FNAC of the nodule, histology was confirmatory.

do

Satellite leisons developed at the same site

May be because of FNAC needle injury

Confirmed by histology of the excised mass

do

none

—

Fig 1 — Clinical presentation of Rhinosporiodosis of different organs (a) Laryngeal Nodule, (b) Penile growth, (c) Anal Growth, (d) Skin nodule on back

cell-wall, electron-dense inclusion, germinative body, sporozoite spore, sporule and spherical body .

In addition to numerous cases in human, it has been documented to occur in several species of domestic and wild animals also such as cattle, buffaloes, dogs, cats and goats. Majority cases are sporadic with only one history of epidemic in 1990 in humans and swans. Another short outbreak was reported in people of Serbia where nasal and ocular rhinosporidiosis were observed and a lake was incriminated as source of infection where all the patients used to bath .

The presumed mode of infection from the aquatic habitat of R.seeberi is through the traumatized epithelium most commonly in nasal sites .

The occurrence of Rhinosporidiosis in river-sand workers in India and Sri Lanka is particularly relevant to such a mode of infection through abrasions caused by sand particles followed by infection by the pathogen in the putative habitat ground water. Trauma from R.seeberi contaminated stones used for mopping up residual drop of urine is claimed to be responsible for anterior urethral rhinosporidiosis in the males. Autoinoculation was considered by Karmaratne in his monogram on rhinosporidiosis where he explained the occurrence of satellite lesions adjacent to granulomatous lesion especially by spillage of endospores from nasal polypoidal lesion to upper aerodigestive tract after trauma 6

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Confirmed by histology of the excised mass

do

none

—

Fig 2 — Microscopic features (a) Haematoxylyn and eosin stained section of sporangium of rhinosporidiosisx 400x, (b) Periodic acid Schiff stained section of sporangium of rhinosporidiosis x 400x

or surgery. Haematogenous spread is another way of spread of disseminated granuloma in subcutaneous tissue as reported by Kumari R et al , Shenoy et al and Tolat et al .

Clinically, the majority of cases occur in upper respiratory tract, less commonly in upper aerodigestive tract and conjunctiva and rarely in skin, penis, anus and bones. Other than bony lesion, everywhere the infection presents as polypoidal, friable mass grossly resembling a neoplastic lesion. Though the organism has predilection for moist mucous membrane of different parts, one case has been reported in parotid duct .

Diagnosis is best done by its histopathology by routine haematoxylin and eosin stain, periodic acid Schiff stain and Gomori’s methenamine silver stain. Differential diagnosis includes spherulocystic disease caused by endosporulating fungus that cannot be stained by silver stain. Another differential diagnosis includes coccidiodes immitis which cause formation of endospores of much smaller size.

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Preliminary Report

J INDIAN MED ASSOC, VOL 114, NO 9, SEPTEMBER 2016

Cytodiagnosis of rhinosporidiosis are not of much help as the endospores in smear resemble epithelium of nasopharynx .

Regarding treatment of polypoidal rhinosporidiosis –surgery with electrocautery is recommended by which recurrence is rare. In other cases- dapsone, primarily an antileprosy drug , has also effect in gradual elimination of R.seeberi by arresting maturation of organism and cause fibrosis . Most of our cases have been treated by surgery followed by dapsone therapy for six months and however none of our cases have history of recurrence.

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CONCLUSION

Rhinosporidiosis, though most commonly seen in nasopharynx, rarely may affect other mucosal surfaces with presentation resembling neoplasm. It is not uncommon in our hot climate, so elimination is needed to be oriented particularly for those patients who work in cattle-sheds or farmhouse. Surgery followed by dapsone therapy is sufficient to cure the lesion.

REFERENCES 1 Emmons CD, Binford CH, Utz JP, Kwon-Chung KJ — In: Lea and Febiger, editors. Medical Mycology, 3rd edn.

Philadelphia 1977: 464-70.

2 Franca GV Jr, Gomes CC, Sakano E, Altermeni AM, Shimizu LT— Nasal rhinosporidiosis in children. J Pediatr 1994; 70: 299-301.

3 Makannavar JH, Chavan SS — Rhinosporidiosis: Clinicopathological study of 34 cases. Indian J Pathol Microbiol 2001; 44: 17-21.

4 Khoo JJ, Kumar KS — Rhinosporidiosis presenting as recurrent nasal polyps. Med J Malays 2003; 58: 282-5.

5 Arseculeratne SN, Ajello L — Rhinosporidium seeberi- In:

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RJ, Ajello L,editors. Topley & Wilson’s Microbiology & Microbial Infections, vol.4, 9th ed.London, Edward Arnold 1998: 595-615.

Kennedy FA, Buggage RR, Ajello L — Rhinosporidiosis: A description of an unprecedented outbreak in captive swans(Cygnus spp.) and a proposal for revision of the ontogenic nomenclature of Rhinosporidium seeberi. J Med Vet Mycol 1995; 37: 157-65.

Vukovic A, Bobic-Radovanovic A, Latkovic Z, Radovanovic Z— An epidemiological investigation of the first outbreak of rhinosporidiosis in Europe. J Trop Med Hygiene 1995; 98: 333-57.

Karunaratne WAE — The pathology of Rhinosporidiosis. J Path Bact 1934; XLII: 193-202.

Karunaratne WAE — Rhinosporidiosis in Man: (the Athlone Press, London) 1964.

Kumari R, Nath AK, Rajalakshmi R, Adityan B, Thappa DM — Disseminated cutaneous rhinosporidiosis: varied morphological appearances in the skin. Indian J Dermatol venerol Leprol 2009; 75: 68-71.

Shenoy MM, Girisha BS, Bhandari SK, Peter R — Cutaneous rhinosporidiosis. Indian J Dermatol Venerol Leprol 2007; 73: 179-80.

Tolat SN, Gokhale NR, Belgaumkar VA, Pradhan SN, Birud NR — Disseminated cutaneous rhinosporidiosis in an immunocompetent male. Indian J Dermatol Venerol Leprol 2007; 73: 343-5.

Kini U, Amirtham V, Shetty SC, Balasubramanya AM — Rhinosporidiosis of the parotid duct cyst: cytomorphological diagnosis of an unusual extranasal presentation. Diagn Cytopathol 2001; 25: 244-7.

McClatchie S , Bremner AD — Unusual subcutaneous swellings in African patients. East Afr Med J 1969; 46: 62533.

Arseculeratne SN — An update on rhinosporidiosis and Rhinosporidium seeberi. 2nd SAARC ENT Congress 2000, Kathmandu, Nepal; Guest lecture.

Job A, Venkateswaran S, Mathan M, Krishnaswamy H, Raman R — Medical therapy of rhinosporidiosis with dapsone. J Laryngol Otol 1993; 107: 809-12.

Vascular tumours of the female genital tract : a clinicopathologic study of 11 cases 1

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Sainath K Andola , Uma S Andola

Vascular tumours of the female genital tract are very rare. The aim of this study was to analyse the distribution of vascular tumors in female genital tract and to correlate their clinicopathological features. In a retrospective study of ten years, clinical features, imaging studies, gross and microscopic features of eleven cases of benign vascular tumours of female genital tract were reviewed. The age range in the present study was 22 to 95 years. The presenting complaint was abdominal pain/mass, postcoital bleeding, vaginal and vulval mass. The duration of symptoms varied from 3 months to 10 years. A diagnosis of vascular tumour was not considered in any of these on clinical grounds. The vascular tumours occurred most commonly in ovary (five cases), followed by vulva (three cases), and one each in cervix, vagina and placenta. Clinical diagnoses ranged from cystadenoma in ovaries to endocervical polyp in cervix, Bartholin’s cyst in vulva and carcinoma in vagina. Histologically, all were benign vascular neoplasms, ranging from haemangioma (five cases), lymphangioma (two cases), lymphangioma circumscriptum (one case) and chorangioma (one case). Two recently described very rare vulval soft tissue tumours angiomyofibroblastoma (one case) and aggressive angiomyxoma of the vulva (one case) were also encountered. Benign vascular tumours in the female genital tract can present with symptoms similar to gynaecological tumours and epithelial malignancies and may lead to unwarranted radical surgery.

Pathological examination is necessary in all such cases to exclude the possibility of malignancy.

Angiomyofibroblastoma and aggressive angiomyxoma of the vulva are very rare and both shared similar clinical and histopathologic features causing diagnostic problems.

[J Indian Med Assoc 2016; 114: 25-30]

Key words : Female genital tract, vascular tumours, haemangioma, lymphangioma, angiomyofibroblastoma, aggressive angiomyxoma, chorangioma, lymphangioma circumscriptum.

V

MATERIALS AND METHODS

ascular tumours are rarely found in the female genital tract (FGT). The ovaries have a rich vascular supply and the rarity of the vascular tumours in the ovary is therefore surprising. It is postulated that the rarity of this tumour is due to the cyclic changes that the ovary undergoes during the reproductive years. Most of the vascular tumours are incidental findings due to their small size and asymptomatic nature.

However, large lesions present clinically with features mimicking the common gynaecological tumours, even on ultrasonographic examination. Most literature contain short series of these tumours confined to one organ of FGT. The objective of the present study is to describe the clinical profile and pathological features of eleven cases of benign vascular tumours of the FGT.

Eleven cases diagnosed as having vascular tumour of FGT in the department of obstetrics and gynaecology and pathology during a period of ten years from 2000-2009, were retrieved. The clinical features, imaging studies and gross findings were analysed and the microscopic slides were reviewed for histopathology features.

1

2

3,4

OBSERVATIONS

Clinical features and physical examination findings have been presented in Table 1. The age of the patient ranged from 22 to 95 years (mean 45.5 years). The duration of symptoms varied between 3 months to 10 years. Five patients had tumours in ovary (3 left, 1 right, 1 bilateral), three in the vulva and one each in cervix, vagina and placenta (Table 2). These cases presented with nonspecific symptoms ranging from abdominal mass and/or pain, postcoital bleeding and vaginal and vulval mass. A diagnosis of vascular tumour was not considered in any of these cases on clinical grounds.

1,3,4

Department of Pathology, Mahadevappa Rampure Medical College, Gulbarga 585105 1 MD, DCP, FICP, FIAMS, Professor and Head of the Department 2 MD, DNB, Associate Professor of Obstetrics and Gynaecology 25

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J INDIAN MED ASSOC, VOL 114, NO 9, SEPTEMBER 2016

The gross and microscopic features have been summarized in Table 3.

Age Presenting complaints Local examination Histopathology diagnosis (Yrs) Three cases with ovarian tumour showed variably 38 Abdominal lump and Pain Left adnexal mass Cavernous haemangioma of Left Ovary enlarged ovaries with 45 Abdominal lump and Pain Left adnexal mass Lymphangioma of Left Ovary honeycomb appearance on 50 Vulval growth Warty Vulval growth.

Lymphangioma Circumscriptum 55 Post menopausal bleeding Cervical growth Ca cervix with metastasis to pelvic cut section with dark brown lymphnodes, small intramural leiomyoma, areas. Microscopy revealed TB lymphadenitis and Bilateral cavernous haemangioma. In ovarian haemangiomas Case 2 and 10, the ovaries 40 Post coital bleeding Cervical growth Cavernous haemangioma cervix 95 Vaginal mass Vaginal growth Cavernous haemangioma vagina were enlarged and cut 35 Growth in Labia Vulval growth section showed multiple majora – 10 years measuring 7x6x4cm Angiomyofibroblastoma of vulva cystic areas and solid areas.

35 Abdominal pain and mass Haemorrhagic cyst Cavernous haemangioema of left overy Microscopy showed 50 Vulval swelling Vulval mass Deep Aggressive Angiomyxoema vulva 35 Pain, abdominal mass Right ovarian mass, Lymphangioma ovary numerous dilated lymphatic in right iliac fossa acute appendicitis spaces filled with lymph 22 Pregnancy Hydramnios with Chorangioma of Placenta fluid and lymphocytic premature separation infiltrates and diagnosis of of placenta lymphangioma was made.

The cervical lesion in case-5 Clinical differential diagnosis in the present series was received as cervical polyp which was haemorrhagic included tubo-ovarian mass (case 1, 2, 10), haemorrhagic and microscopy revealed cavernous haemangioma. In cyst (case 8), endocervical polyp (case 5), Ca vagina (Case case 6, the excision of vaginal mass clinically suspected to 6) Ca cervix with metastasis (case 4) and Bartholin’s cyst be Ca vagina revealed cavernous haemangioma. The vulval lesion in case 3, revealed the features of (case 7, 9). Bilateral ovarian tumour in case 4 was an lymphangioma circumscriptum. incidental finding in the Panhysterectomy specimen in a Case 7, had mass in the labia for last 10 years and case diagnosed as carcinoma cervix. The vulval lesion in clinically diagnosed as Bartholin’s Cyst. As the mass was progressively increasing in size, total excision was done case 7 and 9, were clinically thought to be Bartholin’s cyst. and microscopy revealed tumour consisting of alternate In case 10, USG report showed acute appendicitis with hypo and hypercellular areas with numerous delicate bulky uterus and enlarged right ovary with clinical capillary sized blood vessels lined by endothelial cells.

The stromal cells were plump to spindle cells with diagnosis of acute appendicitis.

USG in three cases with ovarian tumour showed cystic moderate amount of eosinophilic cytoplasm and having round to oval to spindly nucleus with fine chromatin ovarian mass with variable echogenecity. The imaging and inconspicuous nucleoli. These cells were reports of other cases were not available for review. In case numerous in hyper cellular areas and were 4, USG reports suggested enlargement of both ovaries probably because of metastasis. The anatomic distribution of the tumours has been shown in Table 2.

Three of the patients with ovarian tumour underwent Table 2 — Anatomic Distribution of Vascular Tumours total abdominal hysterectomy with salphingoAnatomic Site Histopathologic Number of oophorectomy. In one case, panhysterectomy was Diagnosis Cases (%) performed and bilateral pelvic lymph nodes excised. In Haemangioma 02 (18.18) Left case 2, left sided salphingo-oophorectomy was done.

Lymphangioma 01 (9.09) Ovary Right Lymphangioma 01 (9.09) Endocervical polypectomy was done in case 5 and Bilateral Haemangioma 01 (9.09) Total 05 (45.45) excision of mass (vagina) was done in case 6. In case 7 & 9, Cervix Haemangioma 01 (9.09) Vulval masses were excised completely. In case 3, Vagina Haemangioma 01 (9.09) Lymphangioma 01 (9.09) excision was done. In case-10, appendicectomy was done Vulva Angiomyofibroblastoma 01 (9.09) Deep Aggressive Angiomyxoma 01 (9.09) along with hysterectomy. In case 11, placenta was sent for Total 03 (27.27) examination after delivery as the patient had hydramnios Placenta Chorangioma 01 (9.09) Total 11 (100) with premature separation of placenta.

Table 1 — Clinical Presentation of Vascular Tumours of Female Genital Tract

27

Table 3 — Gross and Microscopic Features of the Tumours of Female Genital Tract Surgical Procedure Uterocervix with right Salphingooophorectomy Left Salphingo oophorectomy Excision of lesion Panhysterectomy Polypectomy Excision of vaginal mass Excision of labial mass

Gross

Microscopy

Ovary 5x3x2 cm.

Ovary 8x5x3 cm. C/s multiple cystic spaces Greyish-brown mass 1x0.5 cm in vulva Ovaries – both 6x5x4 cm. c/s dark brown Irregular friable mass- cervix measuring 2x1.5 cm.

Irregular necrotic and purple reddish mass measuring 4x3x2 cm Globular grey-white mass measuring 7x6x5 cm. c/s well encapsulated grey-white homogenous

Cavernous haemangioma (Right) Cystic Lymphangioma (Left) Lymphangioma circumscriptum – vulva Bilateral Cavernous hemangioma Cavernous haemangioma cervix Cavernous haemangioma vagina

Hysterectomy with left salphingo oophorectomy Ovary measuring 4x6 cm. c/s honeycomb appearance.

Vulval mass Pedunculated mass covered with skin. Lesion 6x4 cm, peduncle 3x1 cm. c/s soft gelatinous reddish to grey-brown unencapsulated.

Uterine cervix with adnexa Right Ovary 6x4x3 cm. C/s cystic spaces and solid areas.

Placenta Placenta measuring 15x13x6 cm. with brown area measuring 6x5cm. with myxoid and variegated appearance

clustered around the blood vessels. There was no atypia or mitoses. A diagnosis of angiomyofibroblastoma of vulva was made.

In case 9, a clinical diagnosis of Batholin’s cyst was made and excision done. Grossly it showed soft gelatinous reddish-brown areas. Microscopically, the lesion was moderately cellular with predominant stellate cells and few spindle cells in an abundant myxoematous stroma.

The stromal cells were bland oval, showed no atypia.

Amidst these were seen numerous medium to large sized blood vessels with thickening of the walls and hyalinisation. Plently of pigmented macrophages were present. A diagnosis of deep aggressive angiomyxoma of vulva was made.

In case 11, the placenta was submitted after the delivery of the baby by caesarean section. Placenta was measuring 15x13x6 cms. Cut section showed a large nodular lesion measuring 6x5 cms and dark brown in appearance. Microscopically the lesion composed of numerous thin walled foetal vessels of capillary or sinusoidal calibre with scant intervening stroma with fibrous and myxoid areas.

In case 4, bilateral cavernous haemangioma were detected as an incidental finding during the procedure of panhysterectomy done in a diagnosed case of carcinoma of cervix. Interestingly in this patient, small leiomyoma was present in the myometrium. The pelvic lymph nodes showed metastatic deposits. In addition, the lymph nodes showed necrotising granulomatous inflammation compatible with tuberculous lymphadenitis. Both the ovaries were enlarged and showed features of cavernous haemangioma.

In all the eleven cases, there was no atypia, no mitoses and no necrosis.

Angiomyofibroblastoma of the vulva Cavernous haemangioma Ovary (Left) Deep aggressive angiomyxoma of vulva Cystic Lymphangioma (Right) Chorangioma of placenta

DISCUSSION

Vascular tumours of FGT, especially of ovary constitute a very small percentage of all tumours of FGT. There are only a few case reports and short series of these tumours in the literature. Vascular tumours have been reported in a wide age group ranging from 4 months to 81 years.4 In the present series, age of the patients ranged from 22 years to 95 years with a mean of 45.5 years.

Majority of the patients were in the age group of 35-50 years (8 cases). There was no specific clinical presentation suggestive of vascular tumor noted in the present series.

However, these tumours can mimic other common FGT neoplasms. One of the cases was highly suspicious of malignancy (case6).

4

1,4

Haemangioma of ovary — Haemangioma of ovary was first described by Payne in 1869. Ovarian haemangiomas are commonly discovered incidentally at autopsy or surgery. Sometimes they present with abdominal mass and/or pain, acute abdomen or ascites, simulating commoner ovarian neoplasms. All the cases in the present series were symptomatic except one (case 4), where panhysterectomy was done in a case of primary cervical cancer which showed incidental findings of bilateral ovarian haemangioma (Fig 1). Ovarian haemangiomas are usually unilateral, though bilateral cases have been reported. Present series also showed incidental finding of bilateral haemangioma of ovary.

2

4

2

Ovarian haemangiomas are usually situated in the medulla and hilus. The lesion has smooth outer surface and is red or purplish on cut surface. In contrast to vascular

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J INDIAN MED ASSOC, VOL 114, NO 9, SEPTEMBER 2016

tumours in the other parts of the body, the most common histologic type in ovary is cavernous or mixed cavernouscapillary type. In the present series all the 5 cases were cavernous type (Fig 2, H&E, x 200). Both the cortex and medulla of the ovaries were involved in all the cases.

Histopathologic examination is diagnostic for the lesion.

Pre-operative diagnosis of ovarian haemangioma may be facilitated by radiologic methods, thus avoiding radical surgery. In the present series, 4 cases were diagnosed as ovarian cyst on ultrasound examination and thus underwent radical surgery. Simple oophorectomy is curative for ovarian hemangioma. Thus clinicopathologic correlation is usually essential.

Lymphangioma of ovary — Lymphangioma of ovary is extremely rare with approximately 16 cases reported in the english literature . In the present series, 2 cases of ovarian lymphangioma were encountered. Clinically they simulated other cystic tumours of the ovary similar to haemangioma. Therefore pathologic examination was necessary to reach the correct diagnosis. Lymphangioma has to be differentiated from teratoma with a prominent vascular component, haemangioma and adenomatoid tumour. The contents in cystic spaces, characteristic morphology with lymphocytic infiltrates and immunohistochemistry may help to differentiate these conditions in difficult cases.

Cervical haemangioma — Fewer than 40 cases of haemangioma of cervix are present in the literature. In the present study, one case of cavernous haemangioma of cervix presented clinically with post coital bleeding and diagnosed as endocervical polyp on examination.

Although cervical haemangiomas are generally asymptomatic, 35% of reported cases were associated with abnormal vaginal bleeding. In one of the reported case, there was rapid growth of the lesion during two pregnancies, necessitating delivery by caesarean section.

3

4

4,5,6

5

6

2,4,7

7

Cavernous haemangioma of vagina — Cavernous hemangioma of the vagina is extremely rare and no cases have been reported in the literature over the past 35 years.

A case of cavernous haemangioma during pregnancy was reported by Rezvani in 1997. The present case was seen in 95-years-old female who presented with a mass in the para-urethral region of the vagina which bled on touch. It was clinically diagnosed as vaginal carcinoma and excised. The mass was vascular, necrotic and friable with a sessile base. Microscopically, the mass revealed hyperplastic squamous epithelium with large dilated cavernous vascular channels lined by flattened endothelium and diagnosed as cavernous haemangioma of vagina (Fig 3, H&E, x 200).

Lymphangioma circumscriptum — Lymphangioma circumscriptum is characterised by clusters of thin walled vessels filled with clear fluid. However epithelial hyperplasia and hyperkeratosis give rise to firm lesions, which are clinically suspected for genital warts or m o l l u s c u m c o n t a g i o s u m . Ly m p h a n g i o m a circumscriptum may be congenital or acquired. To date about 11 cases of congenital and 23 cases of acquired lymphangioma circumscriptum of vulva have been reported in English literature. Acquired cases are mostly seen after radiotherapy to pelvis for carcinoma cervix, hence the cases are diagnosed to exclude metastatic deposits. The present case presented with a small nodular warty lesion in the labia without any previous history of malignancy. Excision biopsy revealed feature of lymphangioma circumscriptum covered by hyperkeratotic hyperplastic squamous epithelium (Fig 4, H&E, x 200).

Angiomyofibroblastoma of vulva — Angiomyofibroblastoma is a rare, benign mesenchymal tumor that occurs mainly in the vulval region of middle aged (35-45years) women.

In 1992, Fletcher et al proposed angiomyofibroblastoma a clinicopathological entity based on the detailed observation of vulval soft tissue

29

8

9

4,9,10

11,12

11

Fig 3 — Microphotograph showing numerous cavernous channels filled with blood seen in sub-epithelial region of vagina. (H&E X 200)

Fig 4 — Microphotograph of lymphangioma circumscriptum showing hyperkeratotic hyperplastic squamous epithelium with multiple fluid filled spaces lined by flat endothelial cells in superficial and deep dermis (H&E X 200)

tumours. Different studies suggest that mesenchymal vulval tumour in women of reproductive age group, angiomyo-fibroblastoma, aggressive angiomyxoema, cellular angiofibroma, fibro-epithelial stromal polyp and superficial angiomyxoema probably arise on a common pluripotential primitive cell located around the vessels of connective tissue, which could show the capacity for modulating its phenotype towards similar but distinctive mature cells. This can result in diagnostic difficulties for pathologists because of the relative rarity and their overlapping morphologic features. There are only over 70 cases reported in the English literature to date.

Angiomyofibroblastoma are well circumscribed and range from 0.5 to 12cm, but usually measure <5cm. They can be adequately treated by wide local excision.

On histologic examination, angiomyofibroblastoma are composed of alternating hypercellular and hypocellular oedematous areas in which numerous thin walled, small to medium sized vessels are regularly distributed. Tumour cells which are described as stromal cells, have a spindle to rounded or epithelioid appearance. Tumour cells are characteristically aggregated around the vessel or loosely dispersed in the hypocellular areas. Nuclear atypia and mitoses are not seen. The oedematous area typically contains wavy collagen fibres but little or no mucin (Fig 5, H&E, x 200). Differentiation between angiomyofibroblastoma and aggressive angiomyxoema may be very difficult both clinically and histologically.

In the present study, one case of angiomyofibroblastoma was diagnosed which presented clinically as Bartholin’s cyst and was present for last 10 years. She came to the hospital because the mass was progressively increasing in size. Grossly, it was globular grey-white mass measuring 7x6x5 cms. Cut section was well encapsulated grey-white with soft rubbery consistency and

histologically diagnosed as angiomyofibroblastoma.

Aggressive angiomyxoema of vulva — Aggressive angiomyxoema was first described by Steeper and Rosai in 1983. This is a rare locally infiltrative tumour that arises in the pelvic and perineal soft tissues of young women.

Approximately 100 cases have been reported. Aggressive angiomyxoema has a high rate of local recurrence because of its infiltrative growth and anatomical location. The treatment of choice is wide local excision. The local recurrence rate in the range of 50-70% is reported.

Grossly, Aggressive angiomyxoema is non-encapsulated gelatinous tumour with infiltrative edge. Histologic examination shows hypocellular tumour with small ovoid, spindled or stellate cells that exhibit minimal nuclear atypia if any (Fig 6, H&E, x 400). Mitotic figures are not common. Numerous blood vessels are present and vary

13,14

14

12,13,14

15

15

14

15,16

14

Fig 1 — Gross Specimen showing incidental bilateral cavernous hemangiomas in ovaries

Fig 2 — Microphotograph showing numerous cavernous vascular channels in ovaries. Normal ovary is seen on left side. (H&E X 200)

Fig 5 — Microphotograph of angiomyofibroblastoma of vulva with many delicate capillary sized blood vessels surrounded by plump to spindle stromal cells with fusiform nuclei. No atypia and no mitosis.

(H&E X 400)

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GP Forum

J INDIAN MED ASSOC, VOL 114, NO 9, SEPTEMBER 2016

particu larly those exceeding 5cm in diameter can cause foetal hydrops, congestive cardiac failure and thrombocytopaenia. In the present series, a case of chorioangioma was seen in the placenta submitted from a patient who had hydramnios.

19

Fig 6 — Microphotograph of aggressive angiomyxoma of vulva showing abundant myxoid stroma with bland oval stromal cells with no atypia. Also seen is large blood vessel with thickening and hyalinization of the walls. (H&E X 400)

from thin walled capillary like vessels to large vessels with thick muscular walls. There is no specific immunohistochemical marker of aggressive angiomyxoema yet.

Tumour cells uniformly express vimentin and heterogeneously express muscle specific actin and desmin.

Srinivasan et al reported aggressive angiomyxoma presenting as a vulval polyp. A case of Aggressive angiomyxoema in a 50-year-old-female with a clinical presentation as Bartholin’s cyst was encouned in this study. Grossly it was a pedunculated mass covered with skin measuring 6x4 cm with gelatinous cut section appearance. Based on the characteristic histologic features a diagnosis of deep aggressive angiomyxoema was made.

There was no recurrence upto 9 months of follow-up.

The case of angiomyofibroblastoma and aggressive angiomyxoema in the present series illustrate that the differential diagnosis can be difficult. The tumour was rather similar in clinical presentation as well as at surgery and on histopathologic examination. Both cases presented as a soft non-tender swelling in the vulva and were preoperatively diagnosed as Bartholin’s cyst. Atypical and diagnostically misleading clinical features were the large size of angiomyofibroblastoma and near absence of local infiltration of aggressive angiomyxoema. Similar features were observed by Schiotz et al . These tumours are so rare that many gynaecological surgeons may never see one.

Placental chorangioma — The chorangioma (haemangioma or so called angiomyxoma of the placenta) is a benign neoplasm present in approximately 1% of placenta. Most chorangiomas are solitary firm solid nodules upto several cms in diameter, located in the superficial parenchyma near the foetal surface. Cut section reveals a smooth myxoid consistency and tan, dark red or variegated appearance. Small solitary chorangioma are usually clinically insignificant. Large chorangioma 16

13,14

17

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18

REFERENCES 1 Uppal S, Heller DS, Majmudar B — Ovarian hemangiomaReport of three cases and review of the literature. J Arch Gynecol Obstet 2004; 270: 1-5.

2 Talerman A — Hemangioma of the ovary and the uterine cervix. Obstet Gynecol 1967; 30: 108-13.

3 Gerbie AB, Hirsch MR, Greene RR — Vascular tumors of female genital tract. Obstet Gynecol 1955; 6: 499-507.

4 Gupta R, Singh S, Nigam S, Khurana N — Benign vascular tumors of female genital tract. Int J Gynecol Cancer 2006; 16: 1195-200.

5 Ahluwalia J, Girish V, Saha S, Dey P — Lymphangioma of the Ovary, Acta Obstet Gynecol Scand 2000; 79: 894-5.

6 Evans A, Lytwyn A, Urbach G, Chapman W — Bilateral Lymphangiomas of the ovary: an immunohistochemical characterization and review of the literature. Int J Gynecol Pathol 1999; 18: 87-90.

7 Cherkis RC, Kamath CP — Hemangioma of the uterine cervix and pregnancy: a case report. J Reprod Med 1988; 33: 393-5.

8 Rezvani FF — Vaginal cavernous hemangioma in pregnancy. Obstet Gynecol 1997; 89: 824-5.

9 Vlastos AT, Malpica A, Follen M — Lymphangioma circumscriptum of the vulva: a review of the literature. Obstet Gynecol 2003; 101: 946-54.

10 Kondi-Pafiti A, Kairi-Vassilatou E, Spanidou-Carvouni H, Kontogianni K, Dimopoulou K, Goula K — Vascular tumors of the female genital tract: a clinicopathologic study of nine cases. Eur J Gynaecol Oncol 2003; 24: 48-50.

11 F l e t c h e r C D M , T s a n g W Y , F i s c h e r C — Angiomyofibroblastoma of the vulva. Am Surg Path 1992; 16: 373-82.

12 Fukunaga M, Nomura K, Matsumoto K, Doi K, Endo Y, Ushigome S — Vulval angiomyofibroblastoma: Clinicopathologic analysis of six cases. Am J Clin Pathol 1997; 107: 45-51.

13 Ducarme G, Valentin M, Davitian C, Felce-Dachez M, Luton D — Angiomyofibroblastoma: a rare vulvar tumor. Arch Gynecol Obstet 2010; 281: 161-2.

14 Schiotz HA, Myhr SS, Chan KF, Klingen TA — Angiomyofibroblastoma and Aggressive angiomyxoma: Two Rare Tumors of the Vulva. J Pelvic Med Surg 2006; 12: 2258.

15 Steeper TA, Rosai J — Aggressive angiomyxoma of the female pelvis and perineum. Report of nine cases of a distinctive type of gynecologic soft-tissue neoplasm. Am J Surg Pathol 1983; 7: 463-75.

16 Begin LR, Clement PB, Kirk ME, Jothy S, McCaughey WT, Ferenczy A — Aggressive angiomyxoma of the pelvic soft parts: A clinicopathologic study of nine cases. Human Pathol 1985; 16: 621-8.

17 Srinivasan R, Mohapatra N, Malhotra S, Rao SK — Aggressive angiomyxoma presenting as a vulval polyp. Ind J Cancer 2007; 44: 87-9.

18 Wallenburg HCS, Chorangioma of the Placenta: Thirteen new cases and review of literature from 1939 to 1970 with special reference to clinical complications. Obstet Gynecol Survey 1971; 26: 411-25.

19 Elder Geva T, Hochner Celnikier D, Ariel — Fetal high output cardiac failure and acute hydramnios caused by large placental chorangioma: case report. Br J Obstet and Gynecol 1988; 95: 1200-2.

Dupatta injuries : an identifiable hazardous entity in a variety of work place and social scenarios 1

2

3

1

2

4

Ashok Kumar , Pritish Singh , S K Babhulkar , Pramod Jain , Bhavya Sirohi , C M Badole

Dupatta is a long loose piece of cloth worn around neck .It is flaunted by both males and females. Despite the cultural association of this apparel, it is part of numerous injury episodes of varying enormity. Severity and enormity of dupatta related injuries range from mild contusions to strangulations, paraplegias, scalp avulsions, miscarriages, traumatic amputations, and burns to even death. These common forms of injury patterns associated with dupatta are observed both in social and industrial milieu. In all such injuries, common component observed is that the long loose piece of cloth gets entangled in a machine part or the wheels and result in a entanglement of hair, limb part etc. Its constant and devastating consequences in cases dictate for a prevention plan comprising both educational and legislative measures.

[J Indian Med Assoc 2016; 114: 31-2]

Key words : Dupatta, scalp avulsion, traumatic amputations, industrial milieu.

D

thresher machine his neck cloth got entangled. He was alone at the moment, and the pulling force of the thresher hit him hard against the rotating machine and avulsed his scalp and hair from the head before helping hands arrived. He was rescued but suffered from mutilating and ghastly scalp avulsion. (Fig 2).

upatta (scarf), a piece of long cloth worn around neck, is part of a traditional apparel in eastern culture. It is uniformly attired by masses in Indian subcontinent due to its cultural associations. The loose and long floating ends of this attire invite trouble in varying common unforeseen situations in the work place, at home, infarmyards, public places etc. The spectrum of harm may range from cautionary abrasions to dire quadriplegias and even unattended strangulations to death .This sporadic but constantly reported hazardous entity calls for a diversified intervention both in the social and occupational surroundings.

Bilateral Forearm Amputation :

1,2,3

A sugar factory daily wedge labourer was feeding raw material in a cane crushing machine. He was distracted for a minute and his loose neck cloth got entangled in the rotating machine part along with sugarcanes and rotating momentum of the machine stopped only after severely crushing both his forearms.

His life was saved but he underwent bilateral fore-arm amputation leaving him devoid of both his forearms for life (Fig 3).

School Girl Strangulation : 14-year-old girl student of the 7th standard was returning from her school along with her school friends riding on a cycle rickshaw. She lost attention to her dupatta and it got entangled in wheel spokes. The moment in waiting was horrible enough to remind her of some impending doom. She was strangulated hard with neck and face thrust against metal bars and dupatta choking her neck.

Fortunately she was rescued by timely efforts and all this left her with a circumferential constriction mark and frightening memories (Fig 1).

Pregnancy Loss :

A young expecting mother with advanced gestation was travelling by a cycle rickshaw on her way back home.

Events turned on the wrong side soon, and her loose long scarf got entangled in uncovered wheel spokes and the jerk she Fig 1 — Showing Schoolgirl with Deep Scalp Avulsion : Strangulation Marks Sustained after endured before the rickshaw halted gave A 32-year-old male farmyard labourer Dupatta Related Injury her a severe pain abdomen and a was preparing to feed his cattle. While cutting green crop for cattle by a motorised circumferential constriction mark. She was immediately hospitalised to a trauma centre, where a sonographic scan revealed a dead foetus, which was evacuated on emergency Department of Orthopedics & Traumatology, Mahatma Gandhi Institute basis. of Medical Sciences, Sewagram 442102 MS (Orth), Associate Professor 2 MS (Orth), Postgraduate Student 3 MCh (Neurosurg), Associate Professor 4 MS (Orth), Professor and Head of the Department

Conclusion :

1

Dupatta is an integral part of the traditional attire of women 31

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of Indian subcontinent. It is a long piece of cloth worn like a scarf around the neck attired by masses owing to its cultural association. It is popularly worn with long loose ends hanging from shoulders to back on both sides. This long loose piece of cloth is part of many continental types of attire like saree. Males are not shy of using a similar type of cloth around neck or head with clandestine thoughts of social supremacy, averting heat etc. Turban is part of traditional attire of males in the Indian society.

These articles of clothing are not only part of social surroundings but are equally part of occupational surroundings. Persons use such clothes in differing proportions depending on Fig 3 — Showing Bilateral forearm their work place situations also.

Fig 2 — Showing Severe Scalp crush sustained in a sugar factory related to Avulsion Sustained after being Pulled The persons wearing this piece of cloth are neck cloth salvaged by a bilateral forearm in Farmyard Grass Chopping Machine vulnerable to varieties of injuries at both social amputation scenarios and workplace situations . This peculiar form of injury is sporadic but with it dic tates scope for intervention . constantly reported. This particular variety of occupational The intervention plan has to be directed both in public and hazard gains importance not only by virtue of its enormity but occupational domains. There is a need to raise awareness level of also by its occurrence in common unforeseen situations and it's the public about increased alertness for such avoidable injuries. scope of prevention.

Incriminating factors like public transport system, poorly There are various scenarios from which such incidences have shielded machines for households can be deemed safe with a been reported. Uncovered wheel spokes of cycle rickshaws, blend of education and legislation. motor cycles, poorly protected thresher machines, grass Industrial environment can be made safer with the help of chopping machines, sugarcane juice extracting machines and uniform safety measures, awareness of personal protection, flour mills are potential origin for such injuries . Roller protective barrier application in roller machines, conveyer belts machines, and conveyer belts are part of numerous etc. Increasing the amount of automation in high risk industrial manufacturing units. Protective measures prescribed in processes will also be a promising step against this bizarre form of legislation are practiced in a compromised manner in workplace harm. More and more participation of semi-organised manufacturing units whether they are small scale or large scale small scale industrial units will promise more prevention. To units. Small scale units in semi-organised sector put their summarise, this particular form of injury needs to be identified as workers more at risk. Cohorts at risk are many eg, household a separate entity in public and industrial work fields and members, school children, mill workers etc. intervention plans should be accordingly directed towards it.

Mode of injury is identical in almost all situations. The loose long piece of cloth is often entwined in rotating component of the REFERENCES machinery and momentum of the moving part pulls body part 1 Aggarwal NK, Agarwal BB — Accidental strangulation in a towards it causing a variety of injuries to victims. The limb parts cycle rickshaw. Med Sci Law 1998; 38: 263-5. are often crushed after futile efforts of escape from the accident.

2 Kohli A, Verma SK, Agarwal BB — Accidental strangulation Spectrum of injuries by this mechanism is diverse. One end in a rickshaw. Forensic Sci Int 1996; 78: 7-11. of the spectrum constitutes simpler injuries like abrasions, 3 Siddiqui AA, Shamim MS, Jooma R, Enam SA — Long scarf contusions, or some blunt trauma to the body part. But graver part injuries. J Coll Physicians Surg Pak 2006; 16: 152-3. of spectrum has injuries like crush injuries of limbs, traumatic 4 Shetty M, Shetty BS — Accidental ligature strangulation due amputations, strangulations near to death, scalp avulsions, to electric grinder. J Clin Forensic Med 2006;13:148–50. traumatic quadriplegias, post-traumatic foetal demise etc.

5 Taff ML, Boglioli LR — Variants of long scarf syndrome. Am J Deaths are also in list from such bizarre incidents .

Forensic Med Pathol 1991; 12: 359-61.

The history also describes the strangulation by a long scarf of 6 Habal MB, Meguid MM, Murray JE — “The long-scarf the world famous dancer Isadora Duncan in 1929. She is syndrome”—a new health hazard. N Engl J Med 1971; 284: acclaimed as a respected figure in modern dance and was fond of 734-36. flaunting long scarves. She died due to accidental strangulation 7 Gupta BD, Jani CB, Datta RG — Accidental strangulation: a and laryngeal rupture by a long loose scarf which got entangled case report. Med Sci Law 2004; 44: 359-62. in the spokes of her car .

8 Gowens PA, Davenport RJ, Kerr J, Sanderson RJ, Marsden This peculiar type of injury with almost singular common AK — Survival from accidental strangulation from a scarf mechanism is very amenable for preventive actions. The ever resulting in laryngeal rupture and carotid artery stenosis: the ‘‘Isadora Duncan syndrome’’. A case report and review of increasing number of such incidents both from public and literature. Emerg Med J 2003; 20: 391-3. industrial milieu and amount of morbidity and burden attached 4

7,8

1-6

5,6,7

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Case Note Recurrent cryptomenorrhoea — a successful outcome Pradip Kr Saha1, Dipak Kr Giri2, Haricharan Roy3, Satabdi Majhi4 Primary amenorrhoea with well developed secondary sexual character with cyclical lower abdominal pain in a postpubertal girl points more towards concealed menstruation. Imperforate hymen and transeverse vaginal septum are the two most common congenital causes of cryptomenorrhoea. Thick transverse vaginal septum if improperly treated, may recur. Successful surgery with proper postoperative care lead to a good permanent outcome.

[J Indian Med Assoc 2016; 114: 33-4]

Key words : Cryptomenorrhoea, transverse vaginal septum.

A

complete transverse vaginal septum leads to haematocolpometra in the pubescent girls. Though small but important component of gynaecological practice is correction of congenital anomalies . Transeverse vaginal septum is rare and its coexistence with cervical dysgenesis is even more rare. Transverse vaginal septum results from incomplete fusion between the vaginal components of the Mullerian ducts and the urogenital sinus . The septum varies in thickness and may be located at any level in the vagina, although most are found in the upper and mid-vagina. Clinical presentation depends on whether it is complete or partial. With complete septa, menstrual blood accumulates and distends structures above the septum after puberty, resulting in hematocolpos and haematometra. Such patients usually present with cyclic lower abdominal pain and ultrasonic findings of haematocolpometra. Occasionally a lower abdominal mass (haematometra) is palpable. Clinical diagnosis is usually difficult before surgery.

1

2,3

4

3

Fig 1 — Showing mild haematometra and large haematocolpos

CASE REPORT

Miss MD aged 16 years old unmarried girl was referred from a subdivision hospital on 10.2.2011 with a hypogastric mass of 14 weeks size with well developed secondary sexual character.

Speculam examination revealed, a blind short vagina of 1 cm depth. On per rectal examination, a soft, globular mass at pouch of douglas, with overlying free rectal mucosa. She had normal complete haemogram, blood sugar, urea, chest x-ray and ECG report. She gives history of drainage of haematocolpos on 10-82010, at private nursing home without any prior USG report.

Following drainage she did not menstruate in the next cycle. She visited gynaecologist of subdivision hospital and advised USG (2-11-2010) which shows (Fig 1), mild collection at endometrial cavity and huge thick collection in distended endocervical canal suggestive of mild haematometra & large haematocolpos but both ovaries & kidneys were normal. She underwent re-drainage there and discharged 10 days after the drainage but unfortunately she does not menstruate in the next cycle.

Following admission to our medical college, a provisional diagnosis of Transverse Vaginal septum with vaginal atresia was made & she underwent operation for the 3rd time on 26-2-2012.

Her HIV screening was negative. Keeping Foley's catheter in bladder and urethra, and exerting traction on catheter, probing of thick vaginal septum is done by stillet of IV cannula. Once the sac of haematocolpos is reached, a verres needle followed by gradually increasing size of Hegars dilator upto size no. 8 is introduced in the same tract towards posterosuperior direction, keeping a finger guide at rectum. A 14 Fr Foley's catheter is placed in the sac of haematocolpos and a continuous catheter drainage of accumulated blood is allowed for 4 days (Fig 2). The vagina was atretic and dissection was done in the loose areolar space between the bladder and rectum towards the cervix and vaginal septal excision and vaginoplasty was carried out keeping the Foley's catheter in situ. A 20ml syringe half cut out on nozzle-side, which is gauze and condom wrapped, over which amnion graft is placed and through inner side of syringe the Foley's catheter is passed and this mould structure is placed in the neo-vagina (Fig 3), and kept for 7 days. A follow up USG was done for re-evaluation (Fig 4). The mould is removed after 7 days when amnion graft is taken up but the Foley's

Department of Obstetrics and Gynaecology, Midnapore Medical College, Paschim Midnapore 721101 1 MBBS, DCH, MD (Obstet and Gynaecol) Assistant Professor 2 MBBS, MD, DNB, RMO cum Clinical Tutor 3 MBBS, DGO, MD (Obstet and Gynaecol), RMO cum Clinical Tutor 4 MBBS House Surgeon 33

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J INDIAN MED ASSOC, VOL 114, NO 9, SEPTEMBER 2016

Branch Activities

Fig 2 — Showing continuous catheter drainage of collection of haematocolpos

catheter is kept in situ for 3 months. OC pill was given for 3 cycles and withdrawl bleeding occured at regular interval. The menstrual blood comes out 80% through foley's catheter & 20% through neo-vagina in 1st cycle, 50% each in 2nd cycle, Whereas in 3rd cycle, 75% blood comes out through neo-vagina and only 25% through Foley's catheter. After 3 month, catheter was taken out but OC pill was continued for another 3 month to enhance regeneration of endometrium and to have regular menstruation.

She was under cover of Doxycycline & Metronidazole. After 6 month, a satisfactory vaginal length of 3 inches was achieved.

Wide area above neo-vagina but no clearly demarcated external os like structure. After a year, a HSG was done, which shows elongated cervix & uterine cavity and without any bilateral peritoneal spillage. She is followed up at our gynaecology clinic.

DISCUSSION

The case presented a transverse vaginal septum which get cured after successful management. Diagnosis of such case is usually arrived at from the history of cyclic pain and primary amenorrhea with or without the clinical finding of a bulging vaginal septum . Bulge in the vagina on rectal examination raised the suspicion of haematocolps. This was confirmed by ultrasound findings. A simple excision and dissection through loose tissue was done to create a neo-vagina. A simpler flap method is applied . To prevent stenosis during the phase of epithelialization, a male condom filled with gauze was inserted in the vagina 5

6

Fig 4 — Showing follow up USG with no collection inside

and removed after one week. Canalization procedures for the cervix have been documented to be successful especially where the cervical body is intact as evidenced by HSG. The neo-vagina created has remained patent for 12 months without need for any dilatation.

REFERENCES

1 Stewart AD — Cryptomenorrhoea due to Transeverse vaginal septum. Online library. Wiley.com/doi/10.1111/j.

1479-828X. 1968.....x/pdf.

2 Rock JA, Azziz R — Genital anomalies in childhood. Clin Obstet Gynecol 1987; 30: 682.

3 Deppisch LM — Transverse vaginal septum: histologic and embryologic considerations. Obstet Gynecol 1972; 39: 1938.

4 Levy G, Warren M, Maidman J. Transverse vaginal septum: case report and review of literature. Int Urogynecol J 1997; 8: 173-6.

5 Brenner P, Sedis A, Cooperman H — Complete imperforate transverse vaginal septum. Obstet Gynaecol 1965; 25: 1358.

6 Wenof M, Reniak JV, Novendstern J, Castadot MJ — Transverse vaginal septum. Obstet Gynecol 1979; 54: 60454.

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Fig 3 — Showing the mould structure which is placed in neo-vagina 35

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