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Pancreaticobiliary maljunction (PBMj) is a congenital dysplasia in which the pancreatic duct and the bile duct join outside the duodenal wall. In PBMj cases, the sphincteric action of the ampulla of Vater cannot reach the junction of the pancreatic duct and the bile duct, thereby causing a reflux of the pancreatic juice into the bile duct. The phenomenon persists, resulting in chronic inflammation in the bile duct epithelium, followed by an atypical change, which is known to lead to the development of biliary tract cancer at a high rate . PBMj is characteristically associated with biliary tract cancer in a relatively large number cases; there have been previous reports in Japan as well on cases of synchronous or metachronous overlap of the two diseases. We recently encountered an extremely rare case in which a patient developed pre-malignant biliary neoplasms simultaneously in three sites: the gallbladder, extrahepatic bile duct, and ampulla of Vater, associated with PBMj. Herein, we report this case, as it will help understand the mechanism of the malignant transformation in PBMj and the multistep carcinogenesis of biliary tract cancer.

Triple Synchronous Neoplasms in the Biliary Tract associated with Pancreaticobiliary Maljunction: A Case Report and Brief Literature Review