Biliary cast syndrome in a non-transplant patient with acute pancreatitis

Abstract

We have read with great interest the paper by Zimmer et al. in Digestive and Liver Disease describing biliary cast syndrome (BCS) in sclerosing cholangitis after orthotopic heart transplantation [ [1] Zimmer V. Raedle J. Treiber G. et al. Biliary cast syndrome in sclerosing cholangitis. Dig Liver Dis. 2011; 43: e4 Abstract Full Text Full Text PDF PubMed Scopus (5) Google Scholar ]. We agree with the authors that cases of BCS are difficult to treat and often require liver transplantation. The occurrence of BCS in the livers of non-transplant patients is extremely rare, with fewer than ten cases described in the literature. Herein, we describe a 37-year-old patient with BCS. Similar to the patient described in Digestive and Liver Disease, our patient presented symptoms of cholangitis, cholestasis and acute pancreatitis (laboratory tests revealed amylase levels of 1550 U/L and lipase levels of 6423 U/L). Magnetic resonance cholangiopancreaticography showed a diffusely destroyed biliary system with multiple filling defects, intrahepatic duct dilation and bile-duct irregularities. Endoscopic retrograde cholangiopancreatography confirmed irregular contours of the biliary duct with strictures and dilations as well as the presence of deposits and biliary sludge. Because of the inability to remove the deposits, a prosthesis and a nasobiliary drainage were inserted into the right and left hepatic ducts respectively. An abdominal CT confirmed the diagnosis of acute pancreatitis with necrosis and abscesses. Due to the serious condition associated with severe acute necrotizing pancreatitis, the first surgical intervention involved only cast removal (Fig. 1) and biliary tract drainage. The abscess and other fluid collections were evacuated and drained, and the necrotic tissue was removed. Morganella morganii and Candida albicans were cultured from the bile. Four months after the operation, during which the patient complained about cholangitis, he was admitted again. Due to destruction of the bile duct, the second surgery, a Roux-en-Y hepaticojejunostomy, was performed. Because changes in the type of biliary cast also include small intrahepatic bile ducts, a recurrence of cholangitis is possible, and a liver transplantation may be necessary.