Recurrent hepatic hydatid disease presenting with biliptysis and suspected bronchobiliary fistula: successful management via thoracotomy, lung wedge resection, and diaphragmatic/subdiaphragmatic cyst evacuation.

backgroundBronchobiliary fistula (BBF) is a rare acquired communication between the biliary system and the bronchial tree. Biliptysis (bile-stained sputum) is the most characteristic clinical clue and should prompt evaluation for underlying hepatobiliary pathology, particularly in endemic regions where echinococcosis remains prevalent. CASE PRESENTATION: A 35-year-old Arab male presented with recurrent biliptysis, right upper quadrant pain, nausea/vomiting, reduced appetite, and mild exertional dyspnea. He had a history of hepatic hydatid cyst surgery 18 years earlier followed by albendazole therapy. Chest imaging showed right lower lobe posterobasal atelectasis/inflammatory consolidation adjacent to the diaphragm, with cystic lesions involving the right hemidiaphragm and multiple cysts at the hepatic dome. Abdominal CT demonstrated a well-defined right hepatic cystic lesion compatible with hydatid disease. Hydatid serology was negative and laboratory tests, including liver enzymes and bilirubin, were within normal limits. Biochemical confirmation of bile was not performed, and bronchoscopy was declined by the patient. CASE DISCUSSION: Given clinically apparent bile-stained sputum with imaging evidence of right basilar lung involvement and recurrent hydatid disease near the diaphragm, surgical management was pursued. Right posterolateral thoracotomy revealed bile-stained fluid containing hydatid membranes at the presumed fistulous site. As no communication with a main bronchus was identified, a right lower lobe wedge resection was performed. Intraoperative findings suggested erosion into adjacent lung parenchyma and smaller bronchi. Multiple diaphragmatic cysts and a subdiaphragmatic cavity containing daughter cysts were evacuated with scolicidal measures, followed by diaphragmatic repair and drainage.

conclusionSuspected BBF should be considered in patients with biliptysis, even with negative serology or normal liver tests. Definitive management may require a tailored surgical approach addressing both thoracic and hepatobiliary components, particularly in hydatid-related disease. CLINICAL TRIAL NUMBER: Not applicable.