Successful Diagnosis and Management of Biliary Cast Syndrome in a Liver Transplant Patient Using Single Operator Cholangioscopy

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Purpose: Biliary cast syndrome is an unusual complication of orthotopic liver transplantation (OLT) with serious clinical implications. Biliary cast formation is usually associated with biliary strictures and/or hepatic ischemia. Surgical management is the mainstay of treatment. Endoscopic techniques have been recently described in the successful removal of biliary casts. We report a post OLT patient who underwent successful endoscopic removal of biliary cast using single operator cholangioscopy. Our patient is a 68 year-old male with history of OLT seven months before for hepatitis C induced cirrhosis who came for a follow up visit. He had no complaints and physical exam was normal. He had cholestatic liver enzyme elevation (Alkaline phosphatase 580 IU/L, Total bilirubin 2.3 mg/dl) with normal aminotransferases. Hepatitis C RNA was undetectable. Ultrasound demonstrated normal common bile duct (CBD) diameter with normal Doppler flow. Liver biopsy showed no rejection with suggestion of cholestasis. Endoscopic retrograde cholangiopancreatoscopy (ERCP) was performed and cholangiogram showed multiple linear filling defects in the common hepatic duct (CHD) proximal to the anastomosis with extension into the intrahepatic system. After failure of the balloon to remove the filling defect, a single-operator choledochoscope was passed into the CHD for diagnostic and possible therapeutic purposes. A dark brown tubular structure was seen resembling a cast of the bile duct. Using a basket, the distal aspect of the cast was secured and a 6 cm long cast was successfully removed in a single piece. (Image) Cholangiogram showed improvement with excellent biliary drainage both fluoroscopically and endoscopically. Liver function tests have subsequently normalized and the patient remains normal with a follow period of 10 months without recurrence. In conclusion, Biliary cast syndrome is a rare complication of OLT which should be thought of when patients present with cholestatic liver enzyme elevation usually within the first year of transplant. Single operator cholangioscopy provides a safe means of diagnosing and treating patients with Biliary cast syndrome particularly in cases with intrahepatic duct casts where surgery or percutaneous techniques are often required. Methods: N/A Results: N/A Conclusion: N/A