An unusual etiology of biliary hilar obstruction and the potential role ofacidic fibroblast growth factor in the development of a biliary neuroma

Neuroma of the biliary tract is a rare condition thought to be caused by tr auma secondary to cholecystectomy. More rare is the occurrence that causes symptomatic biliary obstruction. A 65-year-old woman was hospitalized becau se of abdominal pain, nausea, vomiting, and general malaise of 1 to 2 month s duration. Cholecystectomy had been performed 40 years before. Ultrasound revealed hepatomegaly and dilated intrahepatic ducts. CT showed intra- and extrahepatic ductal dilatation with questionable intraductal mass. Endoscop ic retrograde cholangiopancreatography and percutaneous transhepatic cholan giography demonstrated stricture of the hepatic duct bifurcation. The bilia ry bifurcation was resected, and hepaticojejunostomy was performed. The pat ient's postoperative course was unremarkable. Histological examination of t he surgical specimen revealed positive staining for the S-100 antigen of th e obstructing luminal stricture (without evidence of cholangiocarcinoma), w hich was consistent with a biliary neuroma. Positive staining was also foun d for acidic land not basic) fibroblast growth factor (FGF) and two of its high affinity receptors (FGFR-1 and FGFR-4). This study supports the appare nt association between biliary neuromas and cholecystectomy as well as the potential role of an established angiogenic and neurogenic growth factor in the formation of this tumor. Finally, this case is also unique in that it represents the longest interval between cholecystectomy and presentation of a biliary neuroma, 40 years after surgery.