ORIGINAL ARTICLE Tubercular Fistula-in-Ano Iram Bokhari, Syed Sagheer Hussain Shah, Inamullah, Zahid Mehmood, Syed Umer Ali* and Asadullah Khan ABSTRACT Objective: To determine the frequency of tuberculosis in recurrent fistula-in-ano. Study Design: Case series. Place and Duration of Study: This study was conducted in Surgical Ward-3, Jinnah Postgraduate Medical Centre, Karachi, Pakistan, from 1998 to 2007. Patients and Methods: The study included 100 cases of recurrent fistula-in-ano not responding to conventional surgery. Patients with other co-morbidities such as diabetes mellitus, bleeding disorders or with the evidence of pulmonary, abdominal or intestinal tuberculosis were excluded from this study. Fistulogram was performed in all patients. All the patients were subjected to fistulectomy followed by histopathology of the resected specimen. Thereafter, confirmation of the disease, anti-tuberculous treatment was immediately started and response to treatment was observed after 6 months. Results: Out of the 100 studied patients, 11 cases had biopsy proven tuberculosis in the fistula. All the patients were male. The fistulae were low type, single and usually located posteriorly (n=9) with everted margins. Ten were located within 3 cm of anus. Fistulogram revealed single internal opening. Comparative statistics of tuberculous fistula-in-ano with fistulas due to specific inflammation revealed no major differences. The diagnosed patients of tubercular fistulae-in-ano were observed for at least 6 months after starting anti-tuberculous treatment. They all responded well to anti-tubercular treatment and the fistulae healed without any complication such as recurrence or anal stenosis within 6 months. Conclusion: Tuberculosis should be suspected in case of recurrent fistulae-in-ano, so as to avoid unusual delay in the treatment and miseries to the patient. Appropriate anti-tuberculous therapy leads to healing within 6 months. Key words: Tuberculosis. Fistula-in-ano. Recurrence. INTRODUCTION Pakistan ranks sixth in the world in terms of tuberculosis (TB) burden, with an estimated incidence of 181 per 100000, or 286000 new cases annually.1 Majority of the cases present either as pulmonary, abdominal or cervical tuberculosis. Although it is described as one of the cause of granulomatous diseases within the anorectal region,2 yet perianal TB, without the presence of any previous or active pulmonary infection, is extremely rare.3 Resurgence in tuberculosis during the HIV era produces a new spectrum of presentations for the surgeon4 and, therefore, invasion by tubercle bacilli is often seen at unusual sites of the gut and reported in literature.5 The distinct features, which include anal pain or discharge, multiple or recurrent fistulae-in-ano and inguinal lymphadenopathy, are not characteristically distinct from other anal lesions. Undiagnosed cases are associated with high recurrence rates. Due to the varied Department of General Surgery, Jinnah Postgraduate Medical Centre, Karachi. * Medical Student, The Aga Khan University Hospital, Karachi. Correspondence: Dr. Iram Bokhari, A-17, Sea Breeze Super Star Apartments, Block No. 7, Kehkashan, Clifton, Karachi. E-mail: [email protected] Received February 8, 2008; accepted April 16, 2008. presentation of anal TB, it should be suspected in all lesions not responding to the conventional approaches. All such recurrent or complex fistulae i.e. those with more than one external opening should undergo histopathological examination to exclude tuberculosis. When confirmed, the treatment options are surgical for the suppuration and medical for the tuberculosis. The purpose of this study was, therefore, to determine the frequency of tubercular fistulae-in-ano in recurrent or complex fistula, with no evidence of tuberculous foci elsewhere in the body and not responding to conventional surgical treatment. PATIENTS AND METHODS This study was carried out at the Surgical Ward-3, Jinnah Postgraduate Medical Centre, Karachi, from 1998 to 2007, on 100 patients, admitted through surgical OPD with signs and symptoms suggestive of recurrent fistula-in-ano, not responding to conventional surgical treatment i.e. fistulectomy and developed recurrence within 3 months. Those with diabetes mellitus, bleeding disorders, age below 12 years and evidence of pulmonary or systemic tuberculosis were excluded from this study. Type of fistula, low or high, site of fistula, anterior or posterior, number of fistulas, number of external opening, nature of margins and distance from Journal of the College of Physicians and Surgeons Pakistan 2008, Vol. 18 (7): 401-403 401 Iram Bokhari, Syed Sagheer Hussain Shah, Inamullah, Zahid Mehmood, Syed Umer Ali and Asadullah Khan the anus were noted in all cases. A fistulogram was performed in all the patients. All patients underwent standard fistulectomy and the resected specimen was sent for histopathological evaluation of tuberculosis. After confirmation of the disease, antituberculous treatment was started immediately. For atleast six months after commencement of anti-tuberculous treatment, responses like decrease in the discharge and closure of the fistulae were observed. All these observations were recorded on the prescribed proforma. Descriptive statistics in terms of frequency and proportions were used. RESULTS Out of the 100 patients, the majority were males (92%). The median age was 35 years ranging from 15-65 years. Biopsy revealed non-specific inflammation in majority of the cases. Only in 11 patients, the diagnosis turned out to be tubercular fistula-in-ano. All of the fistulas were low type, single in number and mostly located posteriorly, usually within 3 cms of anal verge. In one case of fistula, due to non specific inflammation, the external openings were multiple. Table I shows the comparative statistics of tubercular fistulas with fistulas due to non-specific inflammation. There were no significant major difference regarding age, gender, type, site, number, external openings and distance from the anus. Fistulogram revealed single internal opening in all the tubercular cases. However, in only one case of nonspecific inflammation, external openings were multiple. These patients were then kept under follow-up for at Table I: Comparative statistics of tuberculous fistulas with non-specific fistulas. Tuberculosis Non-specific inflammation Total no. of patients 11 89 Age (years) mean + SD 33.50 + 7.5 35.49 +12.27 11 81 Gender Male Female 0 8 11 89 High 0 0 Low 11 89 Recurrence* Type of fistula Site of fistula Anterior 2 5 Posterior 9 84 Number of fistula Multiple Single 0 0 11 89 11 88 External openings Single Multiple Margins everted 0 1 11 89 10 80 1 9 Distance from anus Within 3 cms More than 3 cms *Recurrence within 3 months of previous operation 402 least 6 months and in all cases complete healing within 6 months after the start of antituberculous treatment was observed. DISCUSSION Gastrointestinal tuberculosis represents 1% of extrapulmonary tuberculosis and only sporadic cases of anal tuberculosis have been reported in the literature.6,7 Tuberculosis is a neglected cause of anal sepsis. Often it is not recognized and, therefore, is not treated properly.8 This results in recurrence of fistulas after routine surgical treatment. The clinical features of anal tuberculosis, which include anal pain or discharge, multiple or recurrent fistulaein-ano and inguinal lymphadenopathy are not characteristically distinct from other anal lesions.9 Similarly in this study, there was no characteristic clinical picture for tubercular fistulae-in-ano. This series showed that there was no difference in the clinical picture including age and gender distribution and clinical features of anal lesions between the 11 cases of tubercular fistula-in-ano or 89 cases of non-tubercular fistula-in-ano. In one case of non-specific inflammation fistulas, external openings were multiple. A case reported by Gupta,10 showed multiple (eight) external openings in tubercular anal fistulae with evidence of tuberculosis in one of the tracts. Therefore, it seems that histological examination of the excised fistula is mandatory for the diagnosis of anal tuberculosis.11,12 As excised fistula-in-ano are not invariably subjected to histopathological examination, it may also be agreed that some cases of tubercular fistula-in-ano are missed and the incidence of anal or perianal tuberculosis may not be as low as reported in literature.11,13-14 However, non-recurrent fistulas may also be tuberculous. So, to prevent recurrence, all fistulas should be sent for histology. It is also concluded that a tuberculous origin must be considered when the cause of perianal lesion is unclear to avoid undesirable delay in the diagnosis and treatment.15 The main differential for gastrointestinal tuberculosis remains the Crohn’s disease. Crohn's disease is a debilitating expensive disease that is growing in incidence in both developing and developed countries.16 Clinical manifestations varies from asymptomatic skin tags to severe, debilitating perineal destruction and sepsis. However, the histological differential diagnosis of Crohn's disease and intestinal tuberculosis can be very challenging, as both are chronic granulomatous disorders with overlapping histological features.17 In this study, none of the biopsy reports showed the evidence of Crohn’s disease or malignancy. This may be explained by the fact that malignancy is already diagnosed because of its clinical features before the development of fistulous communications at a later Journal of the College of Physicians and Surgeons Pakistan 2008, Vol. 18 (7): 401-403 Tubercular fistula-in-ano drugs in Pakistan. Int J Tuberc Lung Dis 2008; 12: 326-31. stage. So, biopsy is seldom required in such patients and fistulae usually assumed as their sequel. Similarly, Crohn’s disease is not common in this country, therefore, none of the patients were diagnosed with this disease in the biopsy reports. Although abdominal and pulmonary tuberculosis is commoner in women,11 but in this study, none of the resected specimens from the females showed evidence of tuberculosis. This reversal of the gender incidence is difficult to explain unless it is a simple reflection of a generally increased incidence of fistula-in-ano in males compared to females in most series.18 In some cases, pulmonary or other tuberculosis is accompanied with anal tuberculosis. However, in this study, all of these fistulas were primary and there was no evidence of any underlying systemic or gastrointestinal causative factor. So, it further confirms that isolated perianal tuberculosis can occur in the absence of any tuberculous foci elsewhere in the body. Therefore, it further concludes that primary tuberculosis of the perianal region should always be kept in mind when encountering a case of recurrent fistulae or multiple fistulae not healing despite conventional surgical treatment. 3. Akgun E, Tekin F, Ersin S, Osmanoglu H. Isolated perianal tuberculosis. Neth J Med 2005; 63:115-7. 4. Clarke DL, Thomson SR, Bissetty T, Madiba TE, Buccimazza I, Anderson F. A single surgical unit's experience with abdominal tuberculosis in the HIV/AIDS era. World J Surg 2007; 31:1087-96. 5. Lax JD, Haroutiounian G, Attia A, Rodriguez R, Thayaparan R, Bashist B. Tuberculosis of the rectum in a patient with acquired immune deficiency syndrome: report of a case. Dis Colon Rectum 1988; 31: 394-7. 6. Romelaer C, Abramowitz L. Anal abscess with a tuberculous origin: report of two cases and review of the literature. Gastroenterol Clin Biol 2007; 31: 94-6. Myers SR. Tuberculous fissure-in ano. J R Soc Med 1994; 87:46. 8. Kraemer M, Gill SS, Seow-Choen F. Tuberculous anal sepsis: report of clinical features in 20 cases. Dis Colon Rectum 2000; 43:1589-91. 9. Gupta PJ. Ano-perianal tuberculosis- solving a clinical dilemma. Afr Health Sci 2005; 5:345-7. 10. Gupta PJ. A case of multiple (eight external openings) tubercular anal fistulae: a case report. Eur Rev Med Pharmacol Sci 2007; 11:359-61. 11. Shukla HS, Gupta SC, Singh G, Singh PA. Tubercular fistula-inano. Br J Surg 1988; 75:38-9. 12. Ohse H, Ishii Y, Saito T, Watanabe S, Fukai S, Yanai N, et al. A case of pulmonary tuberculosis associated with tuberculous fistula of anus. Kekkaku 1995; 70:385-8. 13. Chung CC, Choi CL, Kwok SP, Leung KL, Lau WY, Li AK. Anal and perianal tuberculosis: a report of three cases in 10 years. J R Coll Surg Edinb 1997; 42:189-90. 14. Alvarez Conde JL, Gutiérrez Alonso VM, Del Riego Tomás J, García Martínez I, Arizcun Sánchez-Morate A, Vaquero Puerta C. Perianal ulcers of tubercular origin: a report of 3 new cases. Rev Esp Enferm Dig 1992; 81:46-8. 15. Gupta PJ. Ano-perianal tuberculosis. Bratisl Lek Listy 2005; 106: 351-4. CONCLUSION 16. Panés J, Gomollón F, Taxonera C, Hinojosa J, Clofent J, Nos P. Crohn's disease: a review of current treatment with a focus on biologics. Drugs 2007; 67:2511-37. Tuberculosis was responsible for 11% cases in recurrent fistula-in-ano responding standard surgery. Antituberculous therapy led to healing within 06 months. A tubercular fistula-in-ano is seldom diagnosed preoperatively on the basis of clinical picture. Therefore- in all cases of recurrent fistula-in-ano, histopathological examination of the excised fistula is mandatory and once tuberculosis is confirmed, antituberculous treatment should be immediately started to ensure early healing and cure of the disease. 17. Kirsch R, Pentecost M, Hall Pde M, Epstein DP, Watermeyer G, Friederich PW. Role of colonoscopic biopsy in distinguishing between Crohn's disease and intestinal tuberculosis. J Clin Pathol 2006; 59: 840-4. 18. Marks CG, Ritchie JK. Anal fistulas at St. Mark’s Hospital. Br J Surg 1977; 64:84-91. 19. Puri AS, Vij JC, Chaudhary A, Kumar N, Sachdev A, Malhotra V, et al. Diagnosis and outcome of isolated rectal tuberculosis. Dis Colon Rectum 1996; 39:1126-9. REFERENCES 20. Rocha M, Carrasco C, Naquira N, Venegas J, Kauer G, Coñoman H. Anal tuberculosis: report of a case. Rev Med Chil 1997; 125:1199-203. Javaid A, Hasan R, Zafar A, Ghafoor A, Pathan AJ, Rab A, et al. Prevalence of primary multi-drug resistance to anti-tuberculosis ● ● ● ● ● Shan YS, Yan JJ, Sy ED, Jin YT, Lee JC. Nested polymerase chain reaction in the diagnosis of negative Ziehl-Neelsen stained Mycobacterium tuberculosis fistula-in-ano: report of four cases. Dis Colon Rectum 2002; 45:1685-8. 7. The treatment is two-fold in such patients of tubercular anal fistulas i.e. surgical for the suppuration and medical for the tuberculosis. Therefore, a good response was seen when healing occurred in all of the cases of tubercular fistulae-in-ano, after anti-tuberculous treatment was started, following fistulectomy. Response to anti-tubercular chemotherapy is uniformly good, and surgery is seldom required in these patients,19 as shown in literature in such cases of perianal tuberculosis.20 Recurrences are unusual after the start of anti-tuberculous therapy. Therefore, an early diagnosis is a must in such patients to prevent recurrences as well as further surgeries of such an easily curable disease. 1. 2. ✯ ● ● ● ● ● Journal of the College of Physicians and Surgeons Pakistan 2008, Vol. 18 (7): 401-403 403
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