Gaurav Garg, Soumyadip Sain, Subhashish Das, Seema Rao, Sunayana Misra, Samiran Nundy
This case documents a previously unreported coexistence of EC and VBDS, identified through a systematic literature review. RUCAM-based causality assessment yielded a score of 4 (Possible), supporting drug-induced liver injury from complementary and alternative medications as the probable cause of VBDS. Persistent postoperative cholestasis in EC should prompt evaluation for secondary causes, particularly drug-induced hepatobiliary injury. Multidisciplinary management is essential for optimal outcomes.
BACKGROUND: Eosinophilic cholecystitis (EC) is a rare histopathological variant, defined by dense transmural eosinophilic infiltration (>90% of inflammatory cells). It is typically an incidental postoperative diagnosis, as its clinical and radiological features mimic those of conventional acute cholecystitis.
CASE SUMMARY: A 38-year-old female with recurrent pregnancy loss presented with acute cholecystitis and markedly deranged liver function tests in a cholestatic pattern. Magnetic resonance cholangiopancreatography revealed cholelithiasis with an impacted neck calculus without biliary obstruction. She underwent laparoscopic cholecystectomy, and histopathology confirmed EC. The postoperative course was complicated by persistent and worsening cholestasis. Extensive evaluation, including autoimmune, parasitic, and eosinophilic workup, was negative. Endoscopic ultrasound-guided liver biopsy demonstrated vanishing bile duct syndrome (VBDS) with ductopenia, attributed to drug-induced liver injury from complementary and alternative medications. Following withdrawal of the offending agents and initiation of ursodeoxycholic acid and ademetionine, liver function tests progressively improved.
CONCLUSION: This case documents a previously unreported coexistence of EC and VBDS, identified through a systematic literature review. RUCAM-based causality assessment yielded a score of 4 (Possible), supporting drug-induced liver injury from complementary and alternative medications as the probable cause of VBDS. Persistent postoperative cholestasis in EC should prompt evaluation for secondary causes, particularly drug-induced hepatobiliary injury. Multidisciplinary management is essential for optimal outcomes.