Severe intrahepatic cholestasis of pregnancy due to a Sertoli-Leydig cell tumour in a woman with polycystic ovary syndrome: a case report

BMC Pregnancy and Childbirth

卷:22

Case Report

Fan Qu¹  Leyi Fu¹  Feng Yun¹  Dong Xu¹  Fangfang Wang¹ 

[1] Women’s hospital, School of Medicine, Zhejiang University, 310006, Hangzhou, China;

关键词: Intrahepatic cholestasis of pregnancy;    Polycystic ovary syndrome;    Sertoli–leydig cell tumour;    Hyperandrogenism;    Case report;   

DOI  :  10.1186/s12884-022-05159-z

 received in 2022-05-19, accepted in 2022-10-26,  发布年份 2022

来源: Springer

【 摘 要 】

BackgroundIntrahepatic cholestasis of pregnancy (ICP) is a common gestational complication characterized by pruritus and elevated bile acids, usually occurring in the third trimester when the serum estrogen and progesterone levels are highest. Hyperandrogenism during pregnancy is a pathological state that is mostly induced by polycystic ovary syndrome (PCOS) but rarely by concomitant androgen-secreting ovarian tumours. To date, no correlation has been drawn between ICP and hyperandrogenism.Case presentationHere, we present a rare case of early-onset severe ICP in a PCOS patient conceived via in vitro fertilization-embryo transfer, with worsening hirsutism and acne due to high levels of testosterone and dehydroepiandrosterone sulphate, both of which were produced by a fast-growing ovarian Sertoli–Leydig cell tumour. Her serum estradiol was also very high, which was speculated to be converted from the circulating androgens by the placenta. She had preterm premature rupture of membranes and delivered at 30 weeks, followed by a rapid remission of ICP as her serum estradiol dropped. However, the excessive androgens did not retreat until the large ovarian tumour was surgically removed.ConclusionThis unusual case highlights the concurrence of original hyperandrogenism and subsequent hyperestrogenism during pregnancy and the resultant confounding manifestations. Obstetricians should be aware of the potential association between androgen excess and ICP via placental aromatization.

【 授权许可】

CC BY   
© The Author(s) 2022

【 图 表 】

Fig. 1