Severe intrahepatic cholestasis of pregnancy due to a Sertoli-Leydig cell tumour in a woman with polycystic ovary syndrome: a case report
Abstract Background Intrahepatic 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 patholo...
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| Format: | Article |
| Language: | English |
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BMC
2022-11-01
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| Series: | BMC Pregnancy and Childbirth |
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| Online Access: | https://doi.org/10.1186/s12884-022-05159-z |
| _version_ | 1811233724003516416 |
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| author | Feng Yun Leyi Fu Dong Xu Fan Qu Fangfang Wang |
| author_facet | Feng Yun Leyi Fu Dong Xu Fan Qu Fangfang Wang |
| author_sort | Feng Yun |
| collection | DOAJ |
| description | Abstract Background Intrahepatic 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 presentation Here, 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. Conclusion This 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. |
| first_indexed | 2024-04-12T11:25:32Z |
| format | Article |
| id | doaj.art-899bb8741aba405e8578cdf81e74c727 |
| institution | Directory Open Access Journal |
| issn | 1471-2393 |
| language | English |
| last_indexed | 2024-04-12T11:25:32Z |
| publishDate | 2022-11-01 |
| publisher | BMC |
| record_format | Article |
| series | BMC Pregnancy and Childbirth |
| spelling | doaj.art-899bb8741aba405e8578cdf81e74c7272022-12-22T03:35:15ZengBMCBMC Pregnancy and Childbirth1471-23932022-11-012211510.1186/s12884-022-05159-zSevere intrahepatic cholestasis of pregnancy due to a Sertoli-Leydig cell tumour in a woman with polycystic ovary syndrome: a case reportFeng Yun0Leyi Fu1Dong Xu2Fan Qu3Fangfang Wang4Women’s hospital, School of Medicine, Zhejiang UniversityWomen’s hospital, School of Medicine, Zhejiang UniversityWomen’s hospital, School of Medicine, Zhejiang UniversityWomen’s hospital, School of Medicine, Zhejiang UniversityWomen’s hospital, School of Medicine, Zhejiang UniversityAbstract Background Intrahepatic 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 presentation Here, 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. Conclusion This 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.https://doi.org/10.1186/s12884-022-05159-zIntrahepatic cholestasis of pregnancyPolycystic ovary syndromeSertoli–leydig cell tumourHyperandrogenismCase report |
| spellingShingle | Feng Yun Leyi Fu Dong Xu Fan Qu Fangfang Wang 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 Intrahepatic cholestasis of pregnancy Polycystic ovary syndrome Sertoli–leydig cell tumour Hyperandrogenism Case report |
| title | Severe intrahepatic cholestasis of pregnancy due to a Sertoli-Leydig cell tumour in a woman with polycystic ovary syndrome: a case report |
| title_full | Severe intrahepatic cholestasis of pregnancy due to a Sertoli-Leydig cell tumour in a woman with polycystic ovary syndrome: a case report |
| title_fullStr | Severe intrahepatic cholestasis of pregnancy due to a Sertoli-Leydig cell tumour in a woman with polycystic ovary syndrome: a case report |
| title_full_unstemmed | Severe intrahepatic cholestasis of pregnancy due to a Sertoli-Leydig cell tumour in a woman with polycystic ovary syndrome: a case report |
| title_short | Severe intrahepatic cholestasis of pregnancy due to a Sertoli-Leydig cell tumour in a woman with polycystic ovary syndrome: a case report |
| title_sort | severe intrahepatic cholestasis of pregnancy due to a sertoli leydig cell tumour in a woman with polycystic ovary syndrome a case report |
| topic | Intrahepatic cholestasis of pregnancy Polycystic ovary syndrome Sertoli–leydig cell tumour Hyperandrogenism Case report |
| url | https://doi.org/10.1186/s12884-022-05159-z |
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