Case Reports
Bronchobiliary fistula due to alveolar hydatid disease: Report of three cases

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Abstract

Bronchobiliary fistula is a serious complication of echinococcosis of the liver. Surgical and endoscopic treatments have been used successfully in the management of bronchobiliary fistula due to hepatic hydatid cysts. However, very little information exists on the management of bronchobiliary fistula due to alveolar hydatid disease. We report here the efficacy of various potential therapies in three cases.

Introduction

The cardinal symptom of bronchobiliary fistula (BBF) is the presence of bile in sputum, so-called biloptysis. BBF due to hydatid cysts is infrequent, and successful results with endoscopic and surgical treatments have been reported 1, 2, 3, 4, 5. Alveolar hydatid disease caused by echinococcus alveolaris is rare, and survival is <10 yr in untreated cases 6, 7. Most patients are not amenable to surgical resection (8). Benzimidazole derivatives (albendazole and mebendazole) have been reported to increase survival 6, 9. Orthotopic liver transplantation is very successful, but recurrence is possible 10, 11. Resection of small lesions, generally detected by screening, is often curative. Very little information exists on BBF as a complication of alveolar hydatid disease. Here we report the efficacy of various therapies with long term prognosis in three cases.

Section snippets

Case 1

A 53-yr-old woman was admitted with fatigue, abdominal pain, and weight loss. The patient had a history of laparotomy 1 yr previously for a mass invading the liver and lower lobe of the right lung. The mass was considered unresectable and biopsies showed alveolar hydatid disease (AHD) (Fig. 1). Severe iron deficiency anemia (hemoglobin, 66 g/L) was present. The patient was treated with iron replacement and 15 4-wk courses of albendazole (10 mg/kg/day) with 2-wk intervals. Lung lesions

Discussion

To the best of our knowledge this is the first report that describes the efficacy of various therapies and long term prognosis in BBF due to AHD.

Previous reports on BBF in hydatidosis are restricted to hydatid cysts 1, 2, 3, 4, 5. AHD is more invasive, rendering an accompanying BBF more difficult to treat. Invasion of the right lung was present in all of our patients on admission. Although all had a history of previous surgery, the possibility of an iatrogenic fistula is unlikely because of the

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    Echinococcosis

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