An ectopic opening of the common bile duct (CBD) into the duodenal bulb is a very rare congenital anomaly of the biliary system, which may cause recurrent duodenal ulcer or biliary diseases such as choledocholithiasis and cholangitis. Endoscopic retrograde cholangiopancreatography (ERCP) plays a major role in the diagnosis of this anomaly. We report two such cases: one in a 61-year-old man and the other in a 57-year-old man. In the first case, this anomaly caused acute cholangitis with multiple CBD stones, which were successfully treated by ERCP. In the second case, abdominal computed tomography showed pneumobilia, which was further evaluated using ERCP. Besides, this patient was diagnosed with an ectopic opening of the CBD associated with gallbladder cancer. We report these unusual cases and review the relevant medical literature.

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