Spontaneous Ovarian Hyperstimulation Syndrome with FSH Receptor Gene Mutation: Two Rare Case Reports

Yılmaz, Emsal Pınar Topdağı; Yapça, Ömer Erkan; Topdaği, Yunus Emre; Topdagi, Seray Kaya; Kumtepe, Yakup

doi:10.1155/2018/9294650

Cited by 3 publications

(2 citation statements)

References 17 publications

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“…Because CA125 increases during the first trimester, it is not accurate for the diagnosis of ovarian tumor during pregnancy. Currently, conservative treatment is the primary management option, and surgery is only needed for cases of ovarian rupture, ovarian torsion, intra-abdominal hemorrhage, or ectopic pregnancy[13]. Pregnancy with sOHSS is a rare disease, and is not easy to detect in the early phase, so obstetricians and gynaecologists should have a comprehensive understanding of sOHSS to make correct diagnosis and treatment, avoiding premature termination of pregnancy and unnecessary surgery damaging the fertility of the patients.…”

Section: Discussionmentioning

confidence: 99%

“…Spontaneous OHSS (sOHSS) has the similar clinical presentations as OHSS and is a rare event. During the past 30 years, sOHSS has been reported in women with polycystic ovarian syndrome[2], hypothyroidism[3], hydatidiform mole[4], invasive mole[5], gonadotropin-producing pituitary adenoma[6,7], multiple gestation[8], disturbed liver function[9], and follicle-stimulating hormone receptor ( FSHR ) gene mutation[10-13]. Previous studies have postulated that multiple pregnancy, gestational trophoblastic diseases, hypothyroidism, gonadotropin adenoma, and FSHR gene mutations are responsible for sOHSS[10].…”

Section: Introductionmentioning

confidence: 99%

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Spontaneous ovarian hyperstimulation syndrome: Report of two cases

Gui

Zhang

et al. 2019

WJCC

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BACKGROUNDSpontaneous ovarian hyperstimulation syndrome (sOHSS) is extremely rare. It can be divided into four types according to its clinical manifestations and follicle stimulating hormone receptor mutations.CASE SUMMARYHere we report two cases of sOHSS in Chinese women, one with a singleton gestation developing sOHSS in the first trimester who conceived naturally and the other with a twin pregnancy developing sOHSS in the second trimester after a thawed embryo transfer cycle. Both patients were admitted to the hospital with abdominal distension, ascites, and enlarged ovaries. Conservative treatment was the primary option of management. The first patient had spontaneous onset labor at 40 wk of gestation and underwent an uncomplicated vaginal delivery of a male newborn. The second patient delivered a female baby and a male baby by caesarean section at 35 wk and 1 d of gestation.CONCLUSIONPatients with a history of ovarian hyperstimulation syndrome should be closely monitored. Single embryo transfer might reduce the risk of this rare syndrome.

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Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Spontaneous ovarian hyperstimulation syndrome: Report of two cases

Gui

Zhang

et al. 2019

WJCC

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Spontaneous ovarian hyperstimulation syndrome in the postpartum period

Rudenko

Зазерская

et al. 2022

Journal of obstetrics and women's diseases

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Spontaneous ovarian hyperstimulation syndrome is an extremely rare complication that develops during or after a spontaneous pregnancy and requires timely diagnosis and appropriate intensive care. This article presents a rare clinical case of spontaneous ovarian hyperstimulation syndrome with the development of its severe clinical picture in the postpartum period. The article focuses on the fact of the difficult diagnosis of this syndrome in the postpartum period and the difficulties of differential diagnosis with systemic inflammatory response syndrome associated with current infection process. The correct diagnosis changed the treatment landscape, when the administration of the right therapeutic doses of low molecular weight heparin provided positive changes in the disease course with a benign outcome.

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Glycoprotein G-protein Coupled Receptors in Disease: Luteinizing Hormone Receptors and Follicle Stimulating Hormone Receptors

et al. 2020

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Signal transduction by luteinizing hormone receptors (LHRs) and follicle-stimulating hormone receptors (FSHRs) is essential for the successful reproduction of human beings. Both receptors and the thyroid-stimulating hormone receptor are members of a subset of G-protein coupled receptors (GPCRs) described as the glycoprotein hormone receptors. Their ligands, follicle-stimulating hormone (FSH) and luteinizing hormone (LH) and a structurally related hormone produced in pregnancy, human chorionic gonadotropin (hCG), are large protein hormones that are extensively glycosylated. Although the primary physiologic functions of these receptors are in ovarian function and maintenance of pregnancy in human females and spermatogenesis in males, there are reports of LHRs or FSHRs involvement in disease processes both in the reproductive system and elsewhere. In this review, we evaluate the aggregation state of the structure of actively signaling LHRs or FSHRs, their functions in reproduction as well as summarizing disease processes related to receptor mutations affecting receptor function or expression in reproductive and non-reproductive tissues. We will also present novel strategies for either increasing or reducing the activity of LHRs signaling. Such approaches to modify signaling by glycoprotein receptors may prove advantageous in treating diseases relating to LHRs or FSHRs function in addition to furthering the identification of new strategies for modulating GPCR signaling.

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Spontaneous Ovarian Hyperstimulation Syndrome with FSH Receptor Gene Mutation: Two Rare Case Reports

Cited by 3 publications

References 17 publications

Spontaneous ovarian hyperstimulation syndrome: Report of two cases

Spontaneous ovarian hyperstimulation syndrome: Report of two cases

Spontaneous ovarian hyperstimulation syndrome in the postpartum period

Glycoprotein G-protein Coupled Receptors in Disease: Luteinizing Hormone Receptors and Follicle Stimulating Hormone Receptors

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