Spontaneous ovarian hyperstimulation syndrome – understanding the dilemma

Dey, Amit; Dubey, Abhishek; Mittal, Kartik; Kale, Sunita

doi:10.3109/09513590.2015.1037268

Cited by 17 publications

(13 citation statements)

References 21 publications

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“…Differential diagnoses in women include ovarian hyperstimulation syndrome, hyperandrogenic chronic anovulation syndrome, and ovarian neoplasia. In men, the differential diagnosis includes McCune-Albright syndrome, congenital testicular cysts, and malignant testicular lesions [ 13 , 14 ]. The management of this sellar tumor is primarily surgical.…”

Section: Discussionmentioning

confidence: 99%

Nonfunctioning Pituitary Adenoma That Changed to a Functional Gonadotropinoma

Andino-Ríos

Portocarrero-Ortíz

Rojas-Guerrero

et al. 2018

Case Reports in Endocrinology

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Objective Pituitary adenomas can be classified as clinically functional or silent. Depending on the reviewed literature, these are the first or second place in frequency of the total pituitary adenomas. Even rarer is the presence of a functional gonadotropinoma since only very few case reports exist to date. The conversion of a clinically silent to functional pituitary adenoma is extraordinarily rare; the mechanisms that explain these phenomena are unknown or not fully understood. Methods We report the case of a woman who initially had a nonfunctional gonadotropinoma and in the course of her medical condition showed biochemical changes in her hormonal pituitary profile compatible with a functional gonadotropinoma. Results We considered that the patient had a functional gonadotropinoma due to the hyperestrogenemia in the context of secondary amenorrhea, resolving the hyperestrogenemia after almost complete resection of the tumor. Conclusion It is necessary to point out from a clinical and/or biochemical point of view the change in functionality that a nonfunctional pituitary adenoma may have. In the case of our patient, the suspicion of this change in functionality became evident when we found an increase in the FSH/LH ratio and a progressive increase in serum estradiol concentrations when the patient had amenorrhea.

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Section: Discussionmentioning

confidence: 99%

Nonfunctioning Pituitary Adenoma That Changed to a Functional Gonadotropinoma

Andino-Ríos

Portocarrero-Ortíz

Rojas-Guerrero

et al. 2018

Case Reports in Endocrinology

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“…Patients with spontaneous OHSS related to TSH can be treated with thyroid hormone replacement therapy, so they have a better prognosis [5], [8]. The cysts normally regress 3–6 months after they develop [6].Most gonadotropin-secreting adenomas (mostly FSH-secreting tumors) secrete inactive hormones, but some can secrete high or normal levels of FSH with higher biological activity. High estradiol levels with non-suppressed or even normal FSH or LH should raise suspicion of autonomous activity.…”

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

“…Normally iatrogenic, OHSS mainly occurs in the context of infertility treatments and is extremely rare in spontaneous pregnancies [1], [2], [3], [4], [5]. In contrast with iatrogenic forms, which typically appear earlier [2], [4], [6], [7], spontaneous OHSS usually develops between the 8th and 14th weeks of gestation; it is sometimes familial and recurrent [6], and it can occur in non-pregnant women [5], [7], [8].…”

Section: Introductionmentioning

confidence: 99%

“…Complications are rare, but should always be considered [1], [6]. Ovarian torsion occurs in 12%–20% of women with spontaneous OHSS [1], [2]; right adnexal torsion is more common than left [2].…”

Section: Introductionmentioning

confidence: 99%

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Ovarian torsion and spontaneous ovarian hyperstimulation syndrome in a twin pregnancy: A case report

Navarro

Grau

Pérez

et al. 2017

International Journal of Surgery Case Reports

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HighlightsSpontaneous ovarian hyperstimulation syndrome (OHSS) can result from hyperstimulation of the follicle-stimulating hormone (FSH) receptor by FSH or other glycoprotein hormones such as chorionic gonadotrophin hormone (hCG), thyroid-stimulating hormone (TSH), and luteinizing hormone (LH); mutations of the FSH receptor gene can make these receptors abnormally sensitive to hCG, TSH, or both.In patients with ovarian cysts and suspected spontaneous OHSS, it is important to determine whether hydatidiform mole, multiple pregnancie, hypothyroidism, glycoprotein-secreting adenoma or FSH receptor mutation are present.Although rare, in the context of spontaneous OHSS and abdominal pain, ovarian complications such as follicular rupture, cyst hemorrhage, or ovarian torsion should always be considered.

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Rare genetic variants suggest dysregulation of signaling pathways in low- and high-risk patients developing severe ovarian hyperstimulation syndrome

Borgwardt

Olsen

Rossing

et al. 2020

J Assist Reprod Genet

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Purpose To investigate if rare gene variants in women with severe ovarian hyperstimulation syndrome (OHSS) provide clues to the mechanisms involved in the syndrome. Methods Among participants in a prospective randomized study (Toftager et al. 2016), six women with predicted low and six women with predicted high risk of OHSS developing severe OHSS (grades 4 and 5, Golan classification) were selected. In the same cohort, six plus six matched controls developing no signs of OHSS (Golan grade 0) were selected. Whole-exome sequencing was performed. Analysis using a predefined in silico OHSS gene panel, variant filtering, and pathway analyses was done. Results We found no convincing monogenetic association with the development of OHSS using the in silico gene panel. Pathway analysis of OHSS variant lists showed substantial overlap in highly enriched top pathways (p value range p < 0.0001 and p > 9.8E-17) between the low-and high-risk group developing severe OHSS, i.e., "the integrin-linked kinase (ILK) signaling pathway" and the "axonal guidance signaling pathway," both being connected to vasoactive endothelial growth factor (VEGF) and endothelial function. Conclusion Rare variants in OHSS cases with two distinct risk profiles enrich the same signaling pathways linked to VEGF and endothelial function. Clarification of the mechanism as well as potentially defining genetic predisposition of the high vascular permeability is important for future targeted treatment and prevention of OHSS; the potential roles of ILK signaling and the axonal guidance signaling need to be validated by functional studies.

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Spontaneous ovarian hyperstimulation syndrome – understanding the dilemma

Cited by 17 publications

References 21 publications

Nonfunctioning Pituitary Adenoma That Changed to a Functional Gonadotropinoma

Nonfunctioning Pituitary Adenoma That Changed to a Functional Gonadotropinoma

Ovarian torsion and spontaneous ovarian hyperstimulation syndrome in a twin pregnancy: A case report

Rare genetic variants suggest dysregulation of signaling pathways in low- and high-risk patients developing severe ovarian hyperstimulation syndrome

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