Case Presentation:

A 59 year-old man with a history of hepatitis C virus infection and hepatocellular carcinoma who received an orthotopic liver transplant 8 years ago, presented with acute onset epigastric pain and melena 6 days after percutaneous liver biopsy was performed to assess for recurrent HCV allograft infection. He had epigastric tenderness with guarding and new onset jaundice. Laboratory tests showed anemia (Hgb 9.7 g/dl); aspartate aminotransferase (82 IU/L); alanine aminotransferase (95 IU/L) alkaline phosphatase (320 IU/L); total bilirubin (6.3 mg/dl); direct bilirubin (4.3 mg/dl); and elevated pancreatic enzymes (amylase 218 U/L; lipase 326 U/L). Abdominal computer tomography (CT) scan showed early enhancement of the left portal vein (Fig. A, arrow), consistent with an arterial-portal fistula.  Endoscopic retrograde cholangiopancreatography disclosed a large clot protruding from the papilla (Fig. B). Cholangiogram revealed dilated intrahepatic bile ducts with numerous filling defects throughout the common hepatic and common bile duct (Fig. C). Sphincterotomy was performed with multiple balloon sweeps yielding blood-tinged bile and coagulated blood. Hepatic angiogram confirmed the arterial-portal fistula (Fig. D, arrow) arising from a branch of the left hepatic artery, supplying lateral segments of the left liver lobe. Urgent hepatic angiography with embolization of the arterial-portal fistula was performed. Angiogram following embolization showed complete fistula occlusion with preserved arterial flow to the lateral lobe segments (Fig. E). The patient’s abdominal pain resolved, and he was discharged two days later.  Laboratory values returned to baseline 4 weeks after discharge.

Discussion:

While percutaneous liver biopsy is a relatively safe procedure, hemobilia is a rare complication with a reported incidence of 0.059%. Acute hemobilia is typically the result of arterial bleeding from direct injury of intrahepatic vessels or bile ducts. Although arterial-portal fistula formation in liver transplant patients after percutaneous hepatic intervention is not uncommon (up to 5.4%), such fistula causing hemodynamically significant bleeding is very unusual (only 0.2%). Arterial-portal fistula causing hemobilia and acute chemical pancreatitis is even more uncommon, having been reported in only twelve cases in published literature.  None of these cases were liver transplant patients. 

Conclusions:

Doppler ultrasound, contrast enhanced CT, as well as magnetic resonance imaging (MRI) scans play important roles in the diagnosis of hemobilia. Angiographic intervention of active hemobilia is the preferred method of treatment for hemodynamic instability, with a success rate over 95%. Although some cases have reported successful resolution with sphincterotomy alone, our patient required angiographic embolization once conservative treatment failed.