Regression of Low Grade Biliary Dysplasia Following Endoscopic Stent Placement: A Case Report

Abstract

Choledochoduodenostomy (CDD) is a surgical technique in which the common bile duct (CBD) is anastomosed to the duodenum to create a fistulous tract. We report a case of biliary dysplasia which reversed, and then reoccurred with the placement of an endoscopic fully covered metal stent (FCMS) then its removal in a patient who underwent remote CDD. A 60-year-old woman with history of cholecystectomy and CDD performed in the 1970s for symptomatic episodes of retained biliary stones presented with recurrent epigastric pain and associated unintentional weight loss over a six month period. MRI of the abdomen showed intrahepatic biliary and common hepatic ductal dilation, a focal moderate grade stricture in the mid-portion of the CBD measuring 0.6 cm in length, and a fistulous tract extending posteriorly from the lower portion of the stricture connecting to the first portion of the duodenum. ERCP with cholangioscopy using Spyglass DS was performed and the mucosa of the bile duct was observed to have been replaced by brush border-like small bowel epithelium. Intestinal contents were seen refluxing into the bile duct through the fistulous tract. Biopsies of the stricture revealed intestinal metaplasia, gastric foveolar hyperplasia, and low grade dysplasia. A FCMS was placed to seal the fistulous tract. Subsequently, the patient reported resolution of her abdominal pain, increased tolerance of oral intake, and weight gain. Four months later, a second ERCP was performed, the FCMS was removed, and biopsies of the bile duct were obtained, with findings of resolution of dysplasia while intestinal metaplasia and gastric hyperplasia persisted. The patient underwent a stent-free trial. Two months later, the patient reported the recurrence of her index pain and food intolerance. Repeated ductal biopsies demonstrated recurrence of dysplasia. Again, a FCMS was placed with repeated symptomatic improvement. The patient was then referred to a surgeon for surgical correction. The mechanism for regression of biliary dysplasia following endoscopic stent placement is not well understood, but one explanation is that cholangiocytes normally remain in a persistent mitotically inactive state, but may proliferate depending on the nature of the stimulus. This case report shows that endoscopic FCMS placement may be useful in treatment of biliary dysplasia in patients with bilioenteric anastomosis or fistula.Figure: Top left - MRI of the abdomen demonstrating the bilioenteric fistula and stricture. Top right - Visualization of the biliary stricture on cholangioscopy. Bottom left - Small bowel-like epithelium in the biliary system seen on cholangioscopy.Figure: Slide 1 - tissue section from the bile duct, an area of fistula showing low grade dysplasia. Slide 2 - same tissue section as Slide 1 stained with P53 immunostain. The epithelial cells show increased P53 staining with features supporting low grade dysplasia (4X, P53 immunostain). Slide 3 - tissue section with goblet cell (arrow) indicating intestinal metaplasia of biliary epithelium (40X, H&E stain). Slide 4 - tissue section showing MUC2 staining of goblet cells in biliary epithelium supporting intestinal metaplasia (4X, MUC2 immunostain). Slide 5 - Alcian blue staining of goblet cells supporting intestinal metaplasia (40X, Alcian blue-PAS stain). Slide 6 - increased MUC5AC staining supporting gastric foveolar hyperplasia (4X, MUC5AC immunostain).