A Case of Juvenile Adenomyomatosis of the Gallbladder Associated with Congenital Bile duct Dilatation and Anomalous Pancreatico-Biliary Union

Hideyuki Ishida, Takeo Iwama, Kuniaki Kitago, Zenro Nihei, Yoshio Mishima

Second Department of Surgery, Tokyo Medical and Dental University

A rare case of juvenile adenomyomatosis of the gallbladder associated with congenital bile duct dilatation and anomalous pancreatico-biliary dilatation is reported. A 13-year-old girl complaining of epigastralgia was admitted and was diagnosed as having adenomyomatosis of the gallbladder, an anomolous pancreatico-biliary union, and cylindrical dilatation of the common bile duct with intrahepatic involvement, by abdominal echography and endoscopic retrograde cholangiopancreatography. She underwent cholecystectomy and resection of the extrahepatic bile duct with hepatico-jejunostomy. Histological examination of the removed gallbladder revealed proliferation of fibromuscular tissue and RokitanskyAschoff sinuses. Including this case, there are 5 cases of adenomyomatosis of the gallbladder associated with an anomalous pancreatico-biliary union in the Japanese literature. The age at diagnosis ranged from 13-35 years. The generalized type of adenomyomatosis and type IIa of anomalous pancreatico-biliary union according to Komi's new classification were characteristics common to all cases. Our case and one other were also associated with Todani's type IVa of congenital bile duct dilatation. From the analysis of the reported cases, it is suggested that one of the etiologies of juvenile adenomyomatosis of the gallbladder may be an anomalous pancreatico-biliary union.

Key words
juvenile adenomyomatosis of the gallbladder, congenital biliary ductal dilatation, anoma-anomalouspancreatico-biliary union

Reprint requests
Hideyuki Ishida Second Department of Surgery, Tokyo Medical and Dental University
1-5-45 Yushima, Bunkyo-ku, Tokyo, 113 JAPAN

Accepted
March 6, 1996

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