Abstract. Follicular cholangitis may lead to severe focal biliary stricture, which is difficult to distinguish from cholangiocarcinoma. This is the report of a rare case of follicular cholangitis associated with an intrahepatic focal biliary stricture in a 69-year-old woman. The patient visited our hospital for upper abdominal pain in 2006. Computed tomography revealed common bile duct stones and focal stricture of the left intrahepatic duct. Endoscopic retrograde cholangiopancreatography revealed a severe B3 stricture and dilation of the peripheral biliary tract. Repeated cytology revealed no malignancy. The patient was followed up for 8 years and repeated attacks of cholangitis severely compromised her quality of life. A left hepatectomy was performed, and the pathological findings were consistent with follicular cholangitis. It is generally difficult to make a definitive diagnosis after identifying a biliary stricture, and the stricture is often treated with surgical resection. The clinical presentation and radiological findings of follicular cholangitis are similar to those of cholangiocarcinoma; therefore, if a conservative approach is selected, frequent follow-up is recommended. The present case suggests that follicular cholangitis should be considered in the differential diagnosis of biliary stricture.
IntroductionFollicular cholangitis is difficult to diagnose, as it is particularly challenging to distinguish from primary sclerosing cholangitis, immunoglobulin G4 (IgG4)-related sclerosing cholangitis and hilar cholangiocarcinoma (1-4). Follicular cholangitis is relatively rare; it was first reported by Aoki et al (5) in 2003, and only a few cases have been reported to date (5-9). Imaging findings representative of this disease include biliary stricture associated with dilatation of the peripheral bile duct and lymph node enlargement. Certain patients have undergone surgical treatment for preoperative diagnosis of cholangiocarcinoma, which was finally diagnosed as a benign condition (10).It was previously demonstrated that the majority of the cases are diagnosed as cholangiocarcinoma due to the severe biliary stricture and the presence of symptoms, which commonly include abdominal pain and jaundice. Follicular cholangitis has recently been reported to be among several diseases that cause biliary stricture. The reported pathological findings of this disease are fibrotic thickening of the wall of the hilar bile duct; prominent hyperplastic lymph follicles with germinal centers in the subserosal layer, without tumorous growth, which were considered to be reactive hyperplastic follicles, with localization of the lesions in the proximal extrahepatic bile duct and hepatic hilum, and with almost intact peripheral intrahepatic bile ducts (8).We herein report the case of a 69-year-old woman with follicular cholangitis associated with a focal biliary stricture, who underwent left hepatectomy after 8 years of follow-up.
Case reportA 69-year-old woman was referred to our hospital due to repeated hepatolithiasis and c...