Exploring Maralixibat for Treatment-Resistant Pruritus in Intrahepatic Cholestasis of Pregnancy and Primary Biliary Cholangitis: A Case Report

BACKGROUND Intrahepatic cholestasis of pregnancy (ICP) is a rare cholestatic condition that occurs during the second and third trimesters, characterized by pruritus and elevated serum bile acid levels. It is associated with a high risk of adverse outcomes for both mother and fetus. Ursodeoxycholic acid remains the only validated treatment for ICP but shows limited efficacy in alleviating pruritus. Ileal bile acid transporter inhibitors, such as maralixibat, have recently been approved for the treatment of pruritus in patients with Alagille syndrome and progressive familial intrahepatic cholestasis type 2. These agents are currently undergoing evaluation for the treatment of primary biliary cholangitis (PBC) and other cholestatic disorders, but data regarding their safety and efficacy in pregnancy - particularly ICP - remain limited. CASE REPORT This report describes a 34-year-old woman who developed jaundice and pruritus in the second trimester of pregnancy due to ICP/PBC overlap. Maralixibat was administered to reduce severe pruritus after prior therapies had failed to achieve symptom control. CONCLUSIONS In this case, maralixibat appeared to be well tolerated and effective as adjunct therapy for severe, treatment-resistant pruritus in a pregnant woman with ICP/PBC overlap; no adverse effects were evident in the mother or the infant during short-term follow-up. However, prior treatments may have contributed to symptom improvement. Although the findings are encouraging, the use of ileal bile acid transporter inhibitors during pregnancy warrants further evaluation in controlled studies before definitive conclusions can be established.