Biliary papillomatosis in the common bile duct

A 62-year-old woman was admitted to our hospital with abdominal pain. Clinical examination revealed definite tenderness in the right hypochondrium only. Laboratory assessment on admission was normal. An abdominal ultrasound was performed, which showed an irregular hyperechoic pseudomass of the gallbladder.

An abdominal contrast-enhanced computed tomography (CT) scan ([Fig. 1]) and a magnetic resonance imaging (MRI) scan of the liver ([Fig. 2]) revealed the presence of an abnormal tissue mass at the hilum of the liver. The suspected diagnosis was confirmed by biliopancreatic endoscopic ultrasound (EUS), which showed an abnormal piece of tissue floating at the upper pole of the common bile duct ([Fig. 3]). The tissue was not invading the common bile duct wall and the endoluminal choledochal surface was intact. These results suggested a benign endoluminal lesion so no biopsy was performed.

Fig. 1 Computed tomography (CT) scan of a 62-year-old woman with abdominal pain and an irregular hyperechoic pseudomass of the gallbladder on ultrasound showing an abnormal tissue mass at the hilum of the liver (1, common bile duct; 2, tumor mass).

Fig. 2 Biliary magnetic resonance imaging (MRI) scan confirming the presence of the abnormal tissue mass at the hilum of the liver (1, common bile duct; 2, tumor mass; 3, gall bladder).

Fig. 3 Biliopancreatic endoscopic ultrasound (EUS) showing an abnormal piece of tissue floating at the upper pole of the common bile duct (1, tumor mass).

The case was discussed by the multidisciplinary oncological team who decided that surgery should be performed. The resected tissue included the gallbladder, extrahepatic bile ducts, and hilar and retropancreatic lymph nodes ([Fig. 4]). A hepaticojejunal anastomosis (Roux-en-Y) was created. The postoperative period was uncomplicated. At microscopic examination, the intraductal region of papillomatosis was found to be surrounded by an invasive tumor, a typical well-differentiated cholangiocarcinoma that was classified as pT1N0M0.

Fig. 4 Macroscopic appearance of the resection specimen showing the tumor mass in the common bile duct (1, common bile duct; 2, tumor mass; 3, cystic duct; 4, gall bladder).

Biliary papillomatosis is an extremely rare disorder [1]. We here report a case of biliary papillomatosis that manifested as vague abdominal pain only. Our treatment option was based on the potential for malignant transformation.

Most patients with this condition present with clinical symptoms of jaundice and acute cholangitis [2] [3] [4]. Nevertheless, biliary papillomatosis can also remain latent many years [5]. Medical imaging can sometimes help in the diagnosis with retrograde cholangiography showing multiple filling defects and a dilated biliary tree with serrated irregularity of the bile duct wall [2] [3]. The diagnosis can be confirmed by intraductal biopsies or by studying the cytology of any secretions. The best treatment is radical liver and biliary surgery with an R0 resection.

Although rare, biliary papillomatosis must be kept in mind when examining a patient suffering from obstructive jaundice who does not have gallstones. Before the final choice of treatment is made, the possibility of a malignant transformation must be assessed with endoscopic retrograde cholangiography, brush biopsy, and core biopsy.

Endoscopy_UCTN_Code_CCL_1AZ_2AN

Competing interests: None

• References

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