2018 Volume 43 Issue 6 Pages 1102-1105
A 37-year-old woman presented to our hospital with jaundice. Clinical results were as follow: serum total bilirubin concentration, 16 mg/dL; serum direct bilirubin, 13 mg/dL; serum alkaline phosphatase, 1,781 IU/L; and γ-glutamyltranspeptidase, 399 IU/L. Abdominal computed tomography (CT) revealed expansion from the common bile duct to the intrahepatic bile ducts of both hepatic lobes and a solid tumor in a multilocular cyst of the left hepatic lobe with neighboring lymphadenopathy. Magnetic resonance cholangiopancreatography and endoscopic retrograde cholangiopancreatography revealed choledocholithiasis in the lower bile duct. The biliary cytology was class I. The multilocular cyst was reduced in size after biliary drainage, but CT revealed a residual solid tumor with a contrasting effect. These findings indicated a intraductal papillary neoplasm of the bile duct (IPNB) or cholangiocarcinoma. Left lobectomy with lymph nodes dissection was performed. Pathological findings indicated no neoplastic lesion in the expanded bile duct; however xanthogranulomatous inflammation throughout the neighboring fat tissue was observed. These results indicated, xanthogranulomatous cholangitis (XGC), which is rare. Reports suggest that differentiation between cholangiocarcinoma and bile duct stenosis is needed because of xanthogranulomatous cholecystitis. In our case, XGC occurred in the intrahepatic bile duct and required differentiation from IPNB.
