Background:Hemobilia is an uncommon complication, and when it is related to choledochal varices it becomes exceptionally rare. Diagnosis may be difficult, and management often requires collaboration between endoscopists and interventional radiologists, especially in patients with portal hypertension and complex vascular anatomy.
Case Presentation:We describe a 44-year-old patient with multivisceral sarcoidosis who had developed extensive thrombosis of the splenomesenteric axis and portal vein, resulting in cavernoma formation, chronic liver disease, and portal hypertension-related cholangiopathy. The patient initially presented with cholestatic liver test abnormalities. Imaging showed intrahepatic bile duct dilation above a short distal common bile duct (CBD) stricture. ERCP confirmed these findings, and a plastic biliary stent was placed.
One month later, the patient was admitted with acute cholangitis and septic shock due to stent obstruction. During urgent ERCP, removal of the plastic stent immediately triggered significant hemobilia. Given the patient’s background, bleeding from choledochal varices was suspected. Balloon sweeps up to 11.5 mm allowed evacuation of clots from the CBD and right intrahepatic ducts. A covered self-expandable metal stent (SEMS) of 10 × 80 mm was placed to achieve tamponade, but bleeding persisted. A second SEMS of the same size was deployed at the biliary confluence, again without satisfactory control.
Because the patient remained unstable, the case was reviewed urgently with interventional radiology and hepatobiliary surgery. No surgical approach was considered feasible, so radiologic management with TIPS was arranged. During the TIPS procedure, injection of contrast into a suprahepatic vein resulted in opacification of the biliary tree, confirming hemobilia. A second angiographic phase showed simultaneous filling of the arterial tree, suggesting the presence of arteriovenous fistulas. TIPS placement was successfully completed, leading to hemodynamic stabilization.
Follow-up imaging demonstrated reduced contrast opacification in both biliary and vascular structures and delineated the cavernoma. The patient improved progressively, and biliary stents were removed without further complications. No recurrent cholangitis or bleeding episodes were observed afterward.
Conclusion:This case illustrates a very unusual source of hemobilia related to choledochal varices in the setting of portal hypertension and complex vascular changes. A combination of endoscopic measures for initial control and radiologic TIPS for definitive management proved essential. Recognizing this possibility is important when hemobilia appears unexpectedly during ERCP in patients with portal hypertension or venous cavernoma.