LAUSR

A 64-year-old woman presented with a 5-day duration of fever, jaundice, and productive cough of green colored sputum. She had undergone more than 20 sessions of endoscopic removal of recurrent choledocholithiasis, a cholecystectomy for cholelithiasis, and a right hepatectomy for hepatolithiasis at the outside hospital. The laboratory results showed aspartate aminotransferase of 73 U/L, alkaline phosphatase of 159U/L, gamma-glutamyl transpeptidace of 141 U/L, and bilirubin of 4.0 mg/dL. Bilioptysis was confirmed with urinary dipstick test (Fig. 1a). Chest computed tomography (CT) revealed a tunnel between the right bronchus and the biliary tract (Fig. 1b). Endoscopic retrograde cholangiography (ERC) revealed the contrast medium was leaked from the common bile duct to the right bronchus (Fig. 1c and video in electronic supplementary material), which confirmed the diagnosis of bronchobiliary fistula (BBF). After endoscopic transpapillary embolization with 10 mL of autologous blood, bilioptysis was temporarily subsided. She subsequently underwent video-assisted thoracoscopic surgery and had no further episodes of bilioptysis. In adult, BBF develops when bile is inadequately drained via the biliary system, leading to inflammatory reactions in the subdiaphragmatic space and subsequent rupture through the bronchial system. The biliary obstruction can be caused by lithiasis, tumor, hydatid cyst, or postoperative stricture [1]. Clinical symptoms include bilioptysis (100%), fever (53%), jaundice (29%), abdominal pain (20%) and respiratory disorder (12%) [2]. However, in some cases, bilioptysis can present with yellow sputum and make it difficult to recognize clinically [3]. Detection of a bile component in sputum by dipstick is helpful to make a diagnosis which can be confirmed by CT, ERC, magnetic resonance cholangiopancreatography, or cholescintigraphy. Management includes endoscopic or percutaneous drainages, embolization, pharmacotherapy and surgery [2]. In the present case, the fistula formation was promoted by both recurrent choledocholithiasis and postoperative changes. Clinicians should be aware of BBF in patients presented with bilioptysis as well as having long history of biliary tract disease and/or biliary tract-related surgery.