BackgroundSevere early-onset pregnancy-associated cholestasis with atypical features may reflect an intrahepatic cholestasis of pregnancy (ICP) component superimposed on underlying hepatobiliary disease. Evidence for bicyclol use during pregnancy is very limited.Case presentationA 34-year-old pregnant woman, developed palmoplantar pruritus and severe cholestasis at 15 weeks of gestation. She had previously undergone cholecystectomy and common bile duct exploration. The working diagnosis was pregnancy-associated cholestasis with a possible ICP component superimposed on suspected chronic hepatobiliary dysfunction. Biochemical abnormalities persisted despite treatment with ursodeoxycholic acid, ademetionine, glutathione, and polyene phosphatidylcholine. Off-label bicyclol at 50 mg three times daily was added at 29 weeks and continued for approximately 5 weeks. Before bicyclol was added, alanine aminotransferase (ALT) level ranged from 724 to 1,492 U/L, and total bile acids (TBA) level ranged from 111.2 to 224.6 μmol/L. Following bicyclol initiation, ALT ranged from 137 to 464 U/L. TBA remained markedly elevated, ranging from 96.7 to 146.6 μmol/L. Total bilirubin ranged from 42.8 to 46.5 μmol/L. Overall, aminotransferase levels were lower than the pretreatment range, whereas TBA and total bilirubin remained markedly elevated. Cesarean delivery was performed at 34+1 weeks of gestation because of worsening maternal symptoms and persistent hyperbilirubinemia. Grade III meconium-stained amniotic fluid was noted during surgery. A liveborn female infant was delivered with Apgar scores of 10, 10, and 10 at 1, 5, and 10 min, respectively. No early neonatal complications were observed. At 42 days postpartum, liver tests remained mildly abnormal, which suggested persistent hepatobiliary dysfunction rather than cholestasis limited to pregnancy.ConclusionA temporal association was observed between bicyclol initiation and lower aminotransferase levels. However, this temporal association should not be interpreted as evidence of causality because multiple factors, including substantial pretreatment biochemical variability, concomitant multidrug therapy, advancing gestation, and possible underlying hepatobiliary disease, may have contributed to the observed changes. This case does not establish efficacy, pregnancy prolongation, or maternal and fetal safety. Instead, it provides a hypothesis-generating observation and highlights the need for further structural and genetic evaluation and systematic studies with long-term infant follow-up.