The clinical presentations of ectopic biliary drainage into duodenal bulbus and stomach with a thorough review of the current literature

Abstract

Background: Ectopic biliary drainage is a rare congenital anomaly on which we have scarce data in the current literature.

Methods: The data were collected from the records of 400 endoscopic retrograde cholangio-pancreatography (ERCP). In this report, we present 10 cases (male/female: 9/1, mean age 54 years, range 38-74) with ectopic biliary openings into the duodenum and/or stomach diagnosed by endoscopic retrograde cholangio-pancreatography (ERCP).

Results: In our series, the frequency of ectopic biliary drainage is 2% (10 out of 400 ERCPs). Recurrent attacks of cholangitis and complicated ulcer formation in the distal stomach and bulbar duodenum were the most common signs in the present series. The sites of ectopic biliary drainage were the stomach in 1 case, the duodenum bulbus in 7 cases and the postbulbar duodenum in 2 cases. Bulbar ulcer, deformed pylorus and bulbus were present in 7 cases, apical bulbar stricture in 2, gastric ulcer in 1, pyloroplasty and/or gastroenterostomy in 3 cases. One case had had previous bleeding episode. Some of them had undergone previous surgeries for gall-stone disease (cholecystectomy in 5 cases, bile duct operation in 3 cases) and ulcer complications (pyloroplasty/gastroenterostomy in 3 cases). ERCP revealed dilatation of the biliary tree and hook shaped distal choledochus in all cases, choledocholithiasis in 7 and Mirizzi syndrome in 1. Endoscopic balloon dilatations for gastric outlet obstruction, extraction of bile stones after balloon dilating the ectopic site, surgery for difficult cases with large bile duct stones or with gastric outlet obstruction were preferred methods in this series of patients.

Conclusion: With this report, we have to remind that ectopic biliary drainage must be considered in the differential diagnosis when the clinician faces cases with gastric outlet obstruction due to peptic ulcer formation accompanied by cholangitis/cholestasis.

Figure 1

A case with ectopic bilio-pancreatic drainage into the stomach. Biliary orifice is indicated by taller arrow. The pancreatic duct orifice is shown by smaller arrow. It is clear to see small amount of bile staining the biliary orifice.

Figure 2

Associated gastric abnormalities. Large antral ulcer is indicated by an arrow. We also see the ERCP catheter placed in the bile duct through the orifice in the stomach.

Figure 3

ERCP appearance. Dilated biliary tree and choledocholithiasis on cholangiography (long arrows). Distal end of choledochus is seen as hook shaped structure (short arrow).

Figure 4

ERCP appearance. Dilated choledochus and unclaved bile duct stone (nearly 2.5 cm in size) at distal end (arrows).

Figure 5

CT appearance. Pneumobilia in CT.

Figure 6

MRCP appearance. An MRCP examination reveals dilated whole biliary tree and hook shaped distal choledochus. MRCP did not delineate bile duct stones.

Figure 7

Therapeutic endoscopy. Dilatation of biliary orifice with pyloric dilatation balloon (arrow) in a case with ectopic drainage of biliary tree into the stomach and choledochus.

Figure 8

Therapeutic ERCP. Stone extraction after biliary balloon dilatation (arrow indicate stone extractor balloon)

Figure 9

A therapeutic flow-chart. An algorithm of practical approach to cases with ectopic biliary drainage is provided.