A 49-year-old male was admitted to the local emergency room (ER) with right upper abdominal pain that occurred after excessive drinking a day earlier. He was a heavy alcoholic. There was no family history. Enhanced abdominal CT scan showed a distal CBD stone and acute cholecystitis with multiple gallbladder stones. Endoscopic biliary sphincterotomy was performed followed by stone removal (Fig. 1A). After that, his jaundice persisted. His serum bilirubin and alkaline phosphatase levels continued to increase. Follow-up CT scan showed persist dilated CBD, dilated both intrahepatic bile ducts, and thrombus in the left portal vein (Fig. 1B). After 3 days, percutaneous transhepatic biliary drainage (PTBD) was performed and drainage catheter interposition was performed. Follow-up cholangiography revealed a long intraluminal filling defect (Fig. 1C). A large amount of stone debris was removed using a stone basket. After that, about a month after the initial symptom, the patient was transferred to the emergency center of our hospital. At the time of the visit to the ER, vital signs were normal and jaundice was observed in the sclera and skin. Laboratory data showed bilirubin of 6.22 mg/dL (reference range: 0.2-1.3 mg/dL), alanine aminotransferase of 107 IU/L (reference range: 13-69 IU/L), aspartate aminotransferase of 101 IU/L (reference range: 15-46 IU/L), alkaline phosphatase of 613 IU/L (reference range: 42-140 IU/L), gamma-glutamyl transferase of 261 IU/L (reference range: 5-55 IU/L). Hepatitis c was confirmed in the patient with viral test and hepatitis C virus RNA positive were 4462906 IU/mL in quantitative tests (real-time quantitative polymerase chain reaction).
Three days after hospitalization, a follow-up cholangiography was performed through an 8F drainage catheter that was previously inserted. On cholangiography, a smooth cast like material, filling of the extrahepatic duct extending into both intrahepatic ducts, and marked ductal irregularity were identified. The patient was diagnosed with biliary cast syndrome and the cast was extracted using a basket through the PTBD catheter (Fig. 1D). The cast was composed of inflammatory exudates with pigmented material (Fig. 1E). The pathology report was negative for malignancy. The patient was discharged with PTBD after conservative treatment.
After a month, the patient visited the emergency center again for abdominal pain and melena. He was diagnosed with cholecystitis and cystic artery pseudoaneurysm (Fig. 1F). He was discharged after cholecystectomy and CBD repair. Over the next two years, there had been intermittent episodes of abdominal pain and general weakness. Each time he was admitted, he received ERCP or PTBD for recurrent biliary cast. Finally, he received liver transplantation. The patient was symptom-free at one-month follow-up.
This case report was exempt from the institutional review board standards (IRB No. KUGH 2021-06-017).