Successful surgical treatment of extrahepatic biliary papillomatosis diagnosed with endoscopic retrograde cholangiopancreatography: a case report

Abstract

Introduction: Biliary papillomatosis is a condition characterized by multiple papillary tumors of variable distribution and extent within the biliary tract. Papillary carcinoma can develop in these lesions. It is a rare biliary pathological entity and its clinical features and outcome are not well known.

Case presentation: We experienced a case of biliary papillomatosis in a 51-year-old North African man who presented with obstructive jaundice. Laboratory tests showed elevated bilirubin, alkaline phosphatase and gamma-glutamyl transpeptidase levels. Imaging (ultrasound and magnetic resonance imaging) was suggestive of Klatskin tumor associated to common bile duct stones. After endoscopic retrograde cholangiopancreatography, a balloon sweep retrieved friable tissue from his bile ducts. Histology demonstrated papillary adenomatous proliferation showing high-grade dysplasia and he was referred for surgical management.

Conclusions: Although biliary papillomatosis is rare, it is a premalignant condition that should be well known and considered in all diagnoses of obstructive jaundice. We report a new case of biliary papillomatosis and highlight the contribution of endoscopic retrograde cholangiopancreatography in the diagnosis of this condition.

Figure 1

Magnetic resonance image showing a complete stop at the biliary bifurcation compatible with a Klatskin tumor associated to common bile duct stones.

Figure 2

After endoscopic retrograde cholangiopancreatography, balloon sweep retrieved friable, polypoid soft tissue from bile ducts.

Figure 3

Post operative view of common bile duct containing papillary material.

Figure 4

Surgical specimen showing polylobed papillary mass within the common bile duct.