A Case of Spontaneous Ovarian Hyperstimulation Syndrome in Pregnancy

doi:10.12280/gjszjk.20210541

Abstract

Abstract:

Ovarian hyperstimulation syndrome is a common complication in assisted reproductive technology. It usually occurs after two weeks or more of induced ovulation. However, the spontaneous ovarian hyperstimulation syndrome (sOHSS) is extremely rare. Some clinical cases showed that the etiology of sOHSS was related to the mutations of follicle stimulating hormone receptor gene, the high level of human chorionic gonadotropin (hCG), the high level of thyroid-stimulating hormone (TSH), and gonadotropin pituitary adenoma. In this paper, we report a case of twin pregnancy complicated with sOHSS, and further study the possible etiology, diagnosis and treatment of sOHSS.

Key words: Spontaneous ovarian hyperstimulation syndrome, Pregnancy, twin, Etiology, Ovulation induction, Reproductive techniques, assisted, Case reports

ZHOU Duo, LI Heng-jing, LU Guang-mei, HUANG Qiu-fang, ZHANG Yi. A Case of Spontaneous Ovarian Hyperstimulation Syndrome in Pregnancy[J]. Journal of International Reproductive Health/Family Planning, 2022, 41(4): 298-301.

Figures/Tables 2

References 32

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参考文献	病理特点	FSH受体基因突变位置	FSH受体突变位置	hCG/TSH 水平	发病机制
Smits等^[5]	家族复发性妊娠早期sOHSS	c.1699G>A p.Asp567Asn	外显子10 （位于第6个跨膜结构域的胞质末端）, 在3个糖蛋白激素受体高度保守	正常	突变受体基础活性增加,对hCG、TSH敏感性增加;细胞实验表明,在表达突变受体的细胞中cAMP异常积累
Vasseur等^[6]	家族复发性妊娠早期sOHSS	c.1346C>T p.Thr449Ile	外显子10 （位于受体第3个跨膜结构域的上部）, 在3个糖蛋白激素受体高度保守	正常	对hCG敏感性增加;外源性hCG使cAMP在表达突变受体的细胞中异常积累
Montanelli等^[7]	家族复发性妊娠早期sOHSS	c.1345A>C p.Thr449Ala	外显子10 （位于第6个跨膜结构域的细胞外末端）,在3个糖蛋白激素受体高度保守	正常	对hCG、TSH异常敏感;细胞实验表明,在表达突变受体的细胞中cAMP异常积累
De Leener等^[8]	妊娠早期sOHSS	c.1636T>C p.Ile554Thr	外显子10 （位于受体第5个跨膜螺旋结构）	正常	对hCG、TSH异常敏感;细胞实验表明,在表达突变受体的细胞中cAMP异常积累
De Leener等^[9]	妊娠早期sOHSS	c.383C>A p.Ser128Tyr	外显子5 （细胞外氨基末端结构域）	正常	对hCG异常敏感;细胞实验表明,外源性hCG使cAMP在表达突变受体的细胞中异常积累
Uchida等^[10]	FSH垂体腺瘤	c.1536G>A p.Met512Ile	外显子10 （位于跨膜结构域）	FSH轻度升高, 生物活性正常	细胞实验表明,显著减少cAMP的产生以及PI3K的磷酸化水平
Chauhan等^[11]	妊娠早期sOHSS	c.1346C>A p.Thr449Asn	外显子10 （位于受体第3个跨膜结构域）	正常	对hCG异常敏感
Hugon-Rodin等^[12]	卵巢蒂扭转	c.1901G>A p.Arg634His	位于受体细胞质的末端	正常	细胞实验表明,在表达突变受体的细胞中cAMP产生减少

参考文献	病理特点	FSH受体基因突变位置	FSH受体突变位置	hCG/TSH 水平	发病机制
Smits等^[5]	家族复发性妊娠早期sOHSS	c.1699G>A p.Asp567Asn	外显子10 （位于第6个跨膜结构域的胞质末端）, 在3个糖蛋白激素受体高度保守	正常	突变受体基础活性增加,对hCG、TSH敏感性增加;细胞实验表明,在表达突变受体的细胞中cAMP异常积累
Vasseur等^[6]	家族复发性妊娠早期sOHSS	c.1346C>T p.Thr449Ile	外显子10 （位于受体第3个跨膜结构域的上部）, 在3个糖蛋白激素受体高度保守	正常	对hCG敏感性增加;外源性hCG使cAMP在表达突变受体的细胞中异常积累
Montanelli等^[7]	家族复发性妊娠早期sOHSS	c.1345A>C p.Thr449Ala	外显子10 （位于第6个跨膜结构域的细胞外末端）,在3个糖蛋白激素受体高度保守	正常	对hCG、TSH异常敏感;细胞实验表明,在表达突变受体的细胞中cAMP异常积累
De Leener等^[8]	妊娠早期sOHSS	c.1636T>C p.Ile554Thr	外显子10 （位于受体第5个跨膜螺旋结构）	正常	对hCG、TSH异常敏感;细胞实验表明,在表达突变受体的细胞中cAMP异常积累
De Leener等^[9]	妊娠早期sOHSS	c.383C>A p.Ser128Tyr	外显子5 （细胞外氨基末端结构域）	正常	对hCG异常敏感;细胞实验表明,外源性hCG使cAMP在表达突变受体的细胞中异常积累
Uchida等^[10]	FSH垂体腺瘤	c.1536G>A p.Met512Ile	外显子10 （位于跨膜结构域）	FSH轻度升高, 生物活性正常	细胞实验表明,显著减少cAMP的产生以及PI3K的磷酸化水平
Chauhan等^[11]	妊娠早期sOHSS	c.1346C>A p.Thr449Asn	外显子10 （位于受体第3个跨膜结构域）	正常	对hCG异常敏感
Hugon-Rodin等^[12]	卵巢蒂扭转	c.1901G>A p.Arg634His	位于受体细胞质的末端	正常	细胞实验表明,在表达突变受体的细胞中cAMP产生减少