S2227 Bile Cast Syndrome in a Young Female Following Severe COVID-19 Infection

Introduction: Post-COVID-19 cholangiopathy is a rare entity that remains poorly understood. Bile cast syndrome is an unusual complication typically reported in patients following liver transplant. Here we describe a unique case of bile cast syndrome developed as a late complication of severe COVID-19 in a young, otherwise healthy female and further discuss therapeutic techniques. Case Description/Methods: A 45-year-old female with a history of a severe COVID-19 infection a year ago was referred for evaluation of abnormal liver tests and constant right upper quadrant abdominal pain. Routine blood work showed elevated alkaline phosphatase at 265 U/L, ALT 107 U/L, AST 62 U/L, GGT 479 U/L, and normal total bilirubin of 1 mg/dL. MR cholangiopancreatogram revealed a biliary stricture involving the proximal right hepatic duct (RHD) with associated focal biliary dilatation in liver segments 8 and 5. In the strictured segment, there was intrinsically T1 hyperintense material and mural enhancement. Endoscopic retrograde cholangiopancreatography (ERCP) was performed under general anesthesia. A cholangiogram showed a stricture vs. filling defect in the main RHD (Figure 1A). The common bile duct was normal. The biliary tree was swept with an extraction balloon starting at the right hepatic duct. One large stone and multiple fragments were removed. A single-operator direct cholangioscope was advanced over a wire and into the RHD, revealing multiple dark brown stones and casts. A cholangioscopy-assisted basket was used with minimal success. Cholangioscopy-guided electrohydraulic lithotripsy was then performed for 20 minutes. Shockwaves generated by an electric spark (50-90 W) were produced through the aqueous medium to achieve stone fragmentation (Figure 1B, C). Multiple balloon sweeps were performed. One 7fr straight plastic stent was placed into the RHD. The procedure was well-tolerated without complications. A repeat ERCP was performed after 4 weeks: the prior stent was removed, and RHD clearance was achieved with stricture resolution. Three months later, the patient had complete normalization of liver tests (Table 1) and was symptoms free. Discussion: COVID-19-related bile cast syndrome is rare; however, more cases are identified a few years post-infection. It can mimic choledocholithiasis or malignant biliary strictures in clinical presentation. Early recognition is essential to prevent long-term complications of cholangiopathy and potentially live failure. ERCP is safe and effective for treating post-COVID-19 bile cast syndrome, especially in the early stages.Figure 1.: A: Fluroscopy image of cholangioscopy guided cannulation of the right hepatic duct; B: Cholangioscopy view of bile casts inside the right hepatic duct; C: Cholangioscopy guided lithortripsy of the bile casts. Table 1. - Patient's laboratory values before and after endoscopic retrograde cholangiopancreatography (ERCP) for post-COVID-19 bile cast syndrome Laboratory Values Before ERCP 3 months after ERCP WBC (cells/μL) 5.06 x 10 6 x 10 Hemoglobin (g/dl) 13.9 13.8 Platelet count (per microliter) 236 x 10 250 x 10 Creatinine (mg/dL) 0.7 0.7 BUN (mg/dL) 11 9 AST (U/L) 487 21 ALT (U/L) 383 40 GGT (U/L) 479 38 ALK (U/L) 265 110 Total Bilirubin (mg/dL) 1 1 Albumin (g/L) 2.6 3.1 ALT, alanine transaminase; ALK, Alkaline phosphatase; AST, aspartate aminotransferase; BUN, blood urea nitrogen; GGT, Gamma-glutamyltransferase.