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Abstract

IntroductionBile cast syndrome (BCS) is a complication of orthotopic liver transplantation (OLT). It is characterized by the presence of biliary casts and debris causing biliary obstruction. It occurs in 4 %-18 % of OLT recipients [1]. It can present as cholangitis and graft damage or loss. Twenty-two percent of patients with BCS require repeat OLT [1]. Symptoms include high fever, jaundice and cholestatic liver enzyme elevation [2]. We describe 2 patients with BCS who were successfully diagnosed with endoscopic retrograde cholangio pancreatography (ERCP) and one of whom was successfully treated endoscopically. Case Reports Patient 1A 65-year-old female underwent OLT 6 months prior to presentation for chronic hepatitis C and hepatocellular carcinoma. Her intraoperative course during liver transplantation was complicated by a significant amount of blood loss from hepatic veins. She had a complicated postoperative course including hemorrhagic shock, abdominal compartment syndrome and acute renal failure which required hospitalization for 2 months after the transplantation. She developed an anastomotic bile leak which was treated with placement of a 10 Fr stent. At 4 weeks and 8 weeks after initial ERCP, the CBD stents were exchanged and secondary sclerosing cholangitis was suspected based on cholangiographic appearance. Her immunosuppression regimen included tacrolimus 9 mg BID and mycofenolic acid 180 mg BID.Laboratory data showed bilirubin of 1. Fig. 1) revealed near complete resolution of the previously described anastomotic stricture. Balloon sweeps yielded a small amount of debris, but cholangiographic findings persisted that were concerning for filling defects or marked ductal irregularity in the region of the common hepatic duct and bifurcation. Cholangioscopy (▶ Fig. 2) was performed with the SpyGlass TM single operator biliary visualization system (Boston Scientific, Natick, MA, USA). A tubular biliary cast, in the shape of the bile duct, was seen in the donor bile duct extending from the anastomosis into the intrahepatic ducts. Attempts to extract the cast with a snare, basket, mechanical lithotripter, balloon, SpyBite TM (Boston Scientific, Natick, MA, USA) biopsy forceps, and standard EGD forceps were all unsuccessful due to inability to grasp the cast and tethering of the cast to intrahepatic extension. A 10 Fr 12-cm CBD stent was placed. In subsequent months, periodic stent changes with continued attempts of removing the bile cast were unsuccessful. The patient's anastomotic stricture has resolved with endoscopic treatment. Patient 2A 58-year-old male with a history of hepatitis C and cirrhosis presented with fevers, chills, and a headache. He also complained of diffuse itching which was affecting his quality of life. He underwent deceased donor liver and kidney transplan-