CASE REPORT
Tuberculous Fistulae in Ano: A Case Report and Literature Review Joanne Favuzza DO,* Sheldon Brotman MD,* Daniel M. Doyle MD,† and Timothy C. Counihan MD* *Department of Surgery and †Department of Medicine, Berkshire Medical Center, Pittsfield, Massachusetts Tuberculosis can present anywhere in the gastrointestinal tract; however, anorectal tuberculosis has been reported rarely. We present a case report of tuberculous fistulae in ano and review the extrapulmonary manifestations of tuberculosis. (J Surg 65: 225-228. © 2008 Association of Program Directors in Surgery. Published by Elsevier Inc. All rights reserved.) KEYWORDS: tuberculosis, fistula in ano COMPETENCY: Patient Care
INTRODUCTION Although tuberculosis can present anywhere in the gastrointestinal tract, anorectal tuberculosis is uncommon. We present a rare case of tuberculous fistulae in ano and review the extrapulmonary manifestations of tuberculosis.
CASE REPORT A 41-year-old Hispanic man presented to the surgery clinic with a complaint of 2 months of perianal discharge and pain. The patient recently immigrated to the United States from Guatemala. Perianal inspection revealed 2 draining fistulae in ano (Fig. 1). Digital rectal examination showed no induration or palpable masses, but a fibrous tract was palpable that extended into the anal canal. Fecal occult blood testing was negative. On review of systems, the patient complained of a persistent cough. A plain radiograph of the chest showed right upper lobe reticular nodular infiltrates and a cavitating infiltrate in the left apex (Fig. 2). The sputum culture grew acid fast bacilli, and a direct test identified Mycobacterium tuberculosis. With this confirmation, the patient was admitted to the hospital and started on a 2-month course of combination antituberculosis therapy, which consisted of isoniazid, rifampin, pyrazinamide, and ethambutol followed by an additional 4 months of isoniazid and rifampin. The antituberculosis drugs were well tolerated by the patient without any evidence of drug-induced hepatitis Correspondence: Inquiries to Timothy C. Counihan, MD, Department of Surgery, Berkshire Medical Center, 725 North Street, Pittsfield, MA 01201; fax: (617) 388-1866; e-mail:
[email protected]
FIGURE 1. Fistulae in Ano prior to treatment
or toxicity. During this patient’s hospitalization, a specimen of the anal fistula revealed granulomatous lesions. Acid fast stain showed acid fast bacilli, and culture also confirmed the presence of Mycobacterium tuberculosis. Three months later, the fistula healed completely (Fig. 3). A follow-up chest radiograph showed resolution of the previously observed apical lesions. The patient was discharged by his surgeon and pulmonary medicine after completion of 6 months of drug therapy.
DISCUSSION According to the World Health Organization (2007), up to one third of the world’s population may be infected with Mycobacterium tuberculosis.1 The emergence of the AIDS/HIV epidemic in the mid-1980s has contributed to the prevalence of tuberculosis.2 Even with the decline in the early 1990s, tuberculosis remains a serious health problem. Although tuberculosis is usually a disease limited to the lungs, extrapulmonary manifestations of tuberculosis have increased from 16% to 21% between 1993 and 2005.3 Extrapulmonary tuberculosis has a variety of presentations and is often difficult to diagnose (Table 14-9). The most common form of extrapulmonary tuberculosis is cervical lymphad-
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FIGURE 2. Chest radiograph before therapy
enitis, which presents asymptomatically as a fixed nontender node with a possible sinus tract.4 Pleural effusions, which are observed in approximately 18% of patients, present as pleuritic chest pain and cough.5 Skeletal tuberculosis, which occurs most often in the spine as Pott’s disease, infects the anterior aspect of the vertebral body of the thoracic spine and presents as back pain.6 The central nervous system may also be involved as tuberculous meningitis. Most patients found to have extrapulmonary tuberculosis can be treated successfully with medical therapy. Gastrointestinal tuberculosis is the sixth most frequent site of extrapulmonary tuberculosis.10 The etiology of gastrointestinal tuberculosis is unclear; however, 3 possible routes can explain these manifestations. Gastrointestinal tuberculosis may develop from ingestion of contaminated milk, swallowing infected sputum, or hematogenous spread.11 Any part of the gastrointestinal tract may be affected from the esophagus to the anus as well as the peritoneum.10 The most common location of gastrointestinal tuberculosis is the ileocecal region.6 Patients may present with a right lower quadrant mass or abdominal pain.10 Gastrointestinal lesions can be either ulcerative, hypertrophic, or a combination of both.6 Hypertrophic cecal lesions or ileal strictures may lead to obstruction.12 Another major complication of ileocecal tuberculosis is perforation, which may be related to the disease process or secondary to stricture. Unlike other extrapulmonary manifestations, ileocecal tuberculosis may require surgery along with conventional medical therapy. Before the use of antituberculous drugs, radical surgical procedures such as a right hemicolectomy and extensive bowel resections were performed.10 However, such procedures were not well tolerated in malnourished patients, and now more conservative procedures are used.10 For short intestinal strictures, antituberculous therapy alone is curative; for strictures that exceed 12 cm, strictureplasty is performed.13 If resection is necessary, limited segmental resections have been advocated.10 Pujari et al14 presented the use of ileocecal resections and ileo226
plasties in 79 patients with ileocecal tubercular lesions. In this series, few complications were noted, including 2 deaths and 2 fecal fistulas that subsequently healed.14 More recently, a prospective trial examined the treatment of intestinal tuberculous stricture. In 3 of 39 patients who had long strictures or multiple areas of involvement, surgery was required. In the remaining 91% of patients in this study, conventional antituberculous therapy was curative.13 With multidrug antituberculous therapy, surgery is required in only a small percentage of patients. Anorectal manifestations of tuberculosis are exceedingly rare; most cases occur in the presence of active pulmonary disease.15 Anorectal tuberculosis has been classified by Nepomuceno et al16 into 4 categories: ulcerative, verrucous, lupoid, and miliary. The most common type is ulcerative tuberculosis, which usually is a result of a primary source often found in the lung or intestines. The verrucous type is characterized by a warty appearance. The lupoid type starts as a reddish-brown nodule and eventually forms an ulcer in its center, whereas miliary lesions are associated with a systemic process.17 Fistulae may develop from any of these initial types.
FIGURE 3. Fistulae in Ano after 3 months of treatment
Journal of Surgical Education • Volume 65/Number 3 • May/June 2008
TABLE 1. Manifestations of Extrapulmonary Tuberculosis Type (% Frequency) Lymphadenitis (42) Pleural (18) Bone and joint (11)
Symptoms
Treatment
Slowly enlarging nontender lymph node. Pleuritic chest pain, nonproductive cough, dyspnea, and fever. Slowly progressive localized pain, muscle spasm, weakness, fever, night sweats, and anorexia.
Meningeal (6)
Irritability, anorexia, behavioral changes progresses to headache, vomiting, stupor, and coma.
Peritoneal (6) Genitourinary (5)
Ascites, abdominal pain, and fever Dysuria, painless hematuria, flank pain, renal mass, sterile pyuria, and recurrent urinary tract infections. Abdominal pain, diarrhea, constipation, weight loss, anorexia, and malaise.
Gastrointestinal (3)
Data derived from references
4-9
Antituberculous drug therapy Antituberculous drug therapy Corticosteroid therapy Antituberculous drug therapy Surgery for bony destruction, instability, or abscess formation Antituberculous drug therapy Surgery for hydrocephalus Corticosteroid therapy Antituberculous drug therapy Antituberculous drug therapy Antituberculous drug therapy Surgery (strictureplasty, segmental resections) for strictures ⬎12 cm in length or multiple areas of involvement
.
Tuberculous fistulae in ano represent a small subset of anorectal cases of tuberculosis. Since the 1980s, tuberculous fistulae in ano have been reported in India, France, Africa, Japan, Australia, Germany, Turkey, and the United Kingdom. In the United States, only 1 case of tuberculous fistula in ano has occurred, which was reported in 1992.18 Tuberculous fistulae in ano can mimic fistulae from cryptoglandular origin, inflammatory bowel disease, foreign body reaction, sarcoidosis, lymphoma, lymphogranuloma venereum amebiasis, and syphilis.19 Clinically, the distinction between anorectal tuberculosis and Crohn’s disease may be difficult, and a histology and culture is required to diagnose tuberculosis. In this study, an immigrant from Guatemela presented with an anal fistula and cough. Pulmonary tuberculosis was suspected after a plain radiograph of the chest revealed bilateral infiltrates, and diagnosis was confirmed by sputum culture. To make the diagnosis of anorectal tuberculosis, Logan15 recommends confirming acid fast bacillus by culture or histology that showed caseating granulomas with clinical or radiographic evidence of tuberculosis elsewhere. Biopsy obtained from this patient’s fistula tract revealed granulomas, and acid fast stain showed acid fast bacilli. Confirmation of this diagnosis led to routine multidrug treatment. Unlike fistulae from common etiologies, surgical intervention is rarely indicated. The fistulae most often resolve with proper antituberculous therapy, as observed in this patient.20
CONCLUSION Although tuberculous fistulae in ano have been observed in many other countries, we present the first case reported since 1992 in the United States. Clinicians who treat patients with fistulae in ano should have a high index of suspicion in immigrants from countries where tuberculosis is endemic or in patients with known pulmonary tuberculosis. This case empha-
sizes the importance of a thorough history and physical examination by all physicians who examine patients with fistulae in ano as well as the extrapulmonary manifestations of tuberculosis.
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