Benign Intrahepatic Bile Duct Stricture Undiscriminated from Intrahepatic Cholangiocarcinoma

Impaired liver function and increased serum CEA levels were detected in a 63-year-old man with bilateral pedal edema. Contrast-enhanced CT showed dilatation of the left intrahepatic bile duct in the S2/S3 segments of the liver. ERCP showed complete obstruction of the left intrahepatic bile duct right after the B1 branch. We suspected intrahepatic cholangiocarcinoma and examined the bile and brushing cytology of the stricture, but both results showed no malignancy. In August 2008, we performed left hepatectomy including the caudate lobe. The resected specimen showed stricture of the intrahepatic bile duct in the S2 segment of the liver and a multilocular cystic lesion. There was no connection between the cystic lesion and the intrahepatic bile duct. The bile duct stricture was encircled by cuboidal epithelia with no cellular atypia, and there was fibrosis of the cystic wall, glandular proliferation, and mild lymphocyte infiltration. The patient is still alive without recurrence at 44 months following surgery. Benign intrahepatic bile duct stricture is very rare and only 12 cases have been reported to date in Japan. It is difficult to distinguish between benign intrahepatic bile duct stricture and intrahepatic cholanciocarcinoma, therefore, surgical resection should be performed after thorough explanation.