Post written by Erica Park, MD, from the Division of Gastroenterology, Hepatology, and Nutrition, The Ohio State University Wexner Medical Center, Columbus, Ohio, USA.

We describe a case of a patient who underwent an orthotopic liver transplantation from a donation after circulatory death. Dehiscence of the biliary anastomosis complicated her post-transplant course, requiring return to the operating room with creation of a Roux-en-Y hepaticojejunostomy.

A couple months later, the patient was found to have elevated results of liver function tests, including a total bilirubin of 2.6 mg/dL and alkaline phosphatase of 374 U/L. She subsequently underwent single-balloon enteroscopy—assisted ERCP, and sludge was swept from the biliary tree and two 7F stents were placed in the left and right hepatic ducts.

A repeat ERCP was performed 2 months later for stent removal. After removal of the first stent, a grasping forceps was used to remove what was thought to be the second biliary stent but found to be a large biliary cast measuring 6 cm in length. After cast removal, the hepaticojejunostomy was widely patent, with resolution of the irregularities seen on prior cholangiogram.

This case shows a remarkable example of biliary cast syndrome, a rare adverse event after liver transplantation occurring in 4% to 18% of transplant patients. Biliary casts are thought to be the result of mucosal necrosis after ischemia and may lead to adverse events including biliary obstruction causing jaundice and cholangitis. Biliary cast syndrome after liver transplant also is associated with increased morbidity, mortality, and graft rejection.

Therefore, it is important to recognize and treat biliary casts. Treatment of biliary cast syndrome is primarily with ERCP with biliary sphincterotomy and balloon extraction of the cast. This case was particularly unique, given the Roux-en-Y hepaticojejunostomy anatomy, which necessitated use of a single-balloon enteroscope with which endoscopic techniques and devices are significantly limited. Fortunately, the cast was able to be successfully removed in this case.

Although they are rare, it is crucial to consider biliary casts in post—liver transplant patients who develop abnormal liver function test results, especially in a cholestatic pattern. It is vital to recognize that nearly half of these patients relapse and may require multiple ERCP procedures for successful treatment of biliary casts.

Large biliary cast removed with a grasping forceps.

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