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Hepatobiliary and pancreatic: Biliary cast syndrome

A 74-year-old man was treated by liver transplantation because of alcoholic cirrhosis and the development of a small hepatocellular carcinoma. Three weeks after surgery, his postoperative course was complicated by the development of cholangitis and intrahepatic abscesses. These complications were treated by repeated endoscopic retrograde cholangiography with biliary washouts and biliary stents. After a further 3 weeks, his clinical condition was unchanged with refractory biliary obstruction, cholangitis and persistent abscesses. A non-contrast computed tomography scan showed a cavity in the right lobe of the liver, a stent in the hilum of the liver and a drain tube (Fig. 1). The diagnosis of a biliary cast syndrome was made and further surgery was undertaken with drainage of the abscesses and an hepaticojejunostomy. Debridement of the biliary tree at the time of surgery yielded a near-complete cast of the intrahepatic ductal system (Fig. 2). Biliary cast syndrome occurs in 4–18% of liver transplant recipients. The majority of the casts are composed of bile components with high concentrations of bilirubin but a minority have high concentrations of collagen, perhaps because of necrosis of biliary epithelium. The diagnosis should be suspected when jaundice or cholangitis is associated with dilated ducts on abdominal imaging studies. Therapeutic options initially involve endoscopic or percutaneous cholangiography with a variety of therapeutic procedures including extraction of casts, biliary washouts, biliary irrigation and use of stents. These procedures are successful in the majority of patients (60–80%) within 3– 4 months although multiple procedures may be required. The cause of the biliary cast syndrome is still debated but possible predisposing factors include acute rejection, hepatic ischemia, prolonged cold ischemia time, biliary obstruction and biliary infections. Of these, hepatic ischemia and biliary strictures appear to be the most important. Those who develop casts have an increase in early mortality.