Severe intrahepatic cholestasis of pregnancy associated with thiopurines: Case report and review of the literature.
- 1Department of Medicine, University of British Columbia, Vancouver, BC, Canada.
- 2Arthritis Research Canada, Vancouver, Canada.
- 3Department of Obstetrics and Gynecology, University of British Columbia, Vancouver, BC, Canada.
Abstract
Intrahepatic cholestasis of pregnancy (ICP) is associated with significant maternal morbidity and fetal risk. We describe a 31-year-old primiparous patient with quiescent systemic lupus erythematosus and lupus nephritis on azathioprine, hydroxychloroquine, and tacrolimus who developed generalized pruritus at 29 weeks. Workup showed markedly elevated bile acids (234 µmol/L) with mild alanine aminotransferase elevation. She was diagnosed with ICP and started on ursodiol and hydroxyzine, but symptoms persisted. Given reports of thiopurine-associated cholestasis, the medication was discontinued, and 6-methylmercaptopurine levels were found to be supratherapeutic. Within days, bile acids and pruritus improved, with normalization over three weeks. She remained asymptomatic, delivered at 36 weeks, and later resumed azathioprine without issue. We summarize 17 cases of thiopurine-associated cholestasis, typically with early onset, severe bile acid elevation, and poor response to ursodiol. This case highlights the importance of medication review, and assessment for possible secondary contributors in atypical or refractory ICP presentations.
Related
- Case Report: Severe persistent transaminitis during pregnancy: a complex differential diagnosis involving suspected toxoplasmosis, possible drug-induced liver injury, and intrahepatic cholestasis
- Marked Hyperbilirubinemia in a Patient with Intrahepatic Cholestasis of Pregnancy and a UGT1A1 Polymorphism: A Case Report
- Peak Total Bile Acids and Adverse Perinatal Outcomes in Intrahepatic Cholestasis of Pregnancy: A Retrospective Cohort Study with Threshold Analysis
- MOTS-c attenuates placental vascular endothelial cell apoptosis in intrahepatic cholestasis of pregnancy by inhibiting K33-linked deubiquitination of BACH1
- The Epidemiology and Associated Factors of Ectopic Pregnancy in Saudi Arabia: A Literature Review.
BibTeX
@article{benhaddadi2026severe,
title = {{Severe intrahepatic cholestasis of pregnancy associated with thiopurines: Case report and review of the literature.}},
author = {Cecilia Benhaddadi and Jieqing Jessica Xu and Jeremiah Tan and Neda Amiri and Monica Beaulieu and Jessica Liauw and Wee-Shian Chan and Tessa Chaworth-Musters},
journal = {Obstetric medicine},
year = {2026},
month = oct,
doi = {10.1177/1753495x261490375},
url = {https://europepmc.org/article/MED/42968874},
note = {CC BY-NC},
}
RIS
TY - JOUR
TI - Severe intrahepatic cholestasis of pregnancy associated with thiopurines: Case report and review of the literature.
AU - Cecilia Benhaddadi
AU - Jieqing Jessica Xu
AU - Jeremiah Tan
AU - Neda Amiri
AU - Monica Beaulieu
AU - Jessica Liauw
AU - Wee-Shian Chan
AU - Tessa Chaworth-Musters
PY - 2026
DA - 2026/10/30
JO - Obstetric medicine
DO - 10.1177/1753495x261490375
UR - https://europepmc.org/article/MED/42968874
AB - Intrahepatic cholestasis of pregnancy (ICP) is associated with significant maternal morbidity and fetal risk. We describe a 31-year-old primiparous patient with quiescent systemic lupus erythematosus and lupus nephritis on azathioprine, hydroxychloroquine, and tacrolimus who developed generalized pruritus at 29 weeks. Workup showed markedly elevated bile acids (234 µmol/L) with mild alanine aminotransferase elevation. She was diagnosed with ICP and started on ursodiol and hydroxyzine, but symptoms persisted. Given reports of thiopurine-associated cholestasis, the medication was discontinued, and 6-methylmercaptopurine levels were found to be supratherapeutic. Within days, bile acids and pruritus improved, with normalization over three weeks. She remained asymptomatic, delivered at 36 weeks, and later resumed azathioprine without issue. We summarize 17 cases of thiopurine-associated cholestasis, typically with early onset, severe bile acid elevation, and poor response to ursodiol. This case highlights the importance of medication review, and assessment for possible secondary contributors in atypical or refractory ICP presentations.
SN - 1753-495X
SN - 1753-4968
ER -