Ovary hyperstimulation syndrome accompanying molar pregnancy: case report and review of the literature

Suzuki, Hiroyoshi; Matsubara, Shigeki; Uchida, Shin-ichiro; Ohkuchi, Akihide

doi:10.1007/s00404-014-3319-0

Cited by 9 publications

(12 citation statements)

References 11 publications

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“…In one case report, a woman presented in the 15th week of pregnancy with a partial (triploid) hydatidiform mole, and ongoing and severe OHSS even after termination of pregnancy. VEGF levels were very high despite low levels of hCG, supporting the hypothesis that VEGF might cause OHSS 11 12. In our case, since VEGF can be produced by PMD macrophages and has been associated with OHSS, we postulate that PMD-derived VEGF was responsible for the spontaneous OHSS.…”

Section: Discussionsupporting

confidence: 84%

“…However, mutated FSH receptors expressed in the developing follicles may be hyperstimulated by pregnancy-derived hCG and result in enlarged follicles. In addition, luteinising hormone receptors expressed by granulosa cells may also be stimulated by hCG to induce follicular luteinisation and the secretion of vasoactive molecules such as VEGF, angiotensin II and various interleukins that are responsible for the development of the syndrome 10 12. In one case report, a woman presented in the 15th week of pregnancy with a partial (triploid) hydatidiform mole, and ongoing and severe OHSS even after termination of pregnancy.…”

Section: Discussionmentioning

confidence: 99%

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Placental mesenchymal dysplasia associated with spontaneous ovarian hyperstimulation syndrome

Davoudian

2015

BMJ Case Reports

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Placental mesenchymal dysplasia (PMD) is a rare disorder of unknown aetiology characterised by placentomegaly, serpiginous surface blood vessels and large cystic villi. Although a mimic of molar pregnancies and other entities, it can be distinguished from them by its association with intrauterine growth restriction or death of a relatively normal fetus and its characteristic histopathology. We report the case of a 20-year-old primigravida who presented at 16 weeks with heavy clotty vaginal bleeding and subsequent miscarriage. The placenta was grossly abnormal and resembled a partial molar pregnancy. PMD was confirmed by microscopic examination. Postmiscarriage, ultrasound revealed enlarged and multicystic ovaries similar to those seen in ovarian hyperstimulation syndrome (OHSS), which returned to normal a few weeks after miscarriage. This is the first report of PMD associated with OHSS, and we hypothesise that the most likely pathogenesis is ovarian stimulation from PMD-derived vascular endothelial growth factor.

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Section: Discussionsupporting

confidence: 84%

Section: Discussionmentioning

confidence: 99%

Placental mesenchymal dysplasia associated with spontaneous ovarian hyperstimulation syndrome

Davoudian

2015

BMJ Case Reports

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“…Factors that increase the incidence of ovarian pregnancy are younger age, endometriosis, pelvic inflammatory disease, intrauterine devices, use of ovulatory medications and assisted reproduction techniques. Ectopic molar gestation is very rare; the incidence is estimated to be only 1.5 per million births 2 3…”

Section: Discussionmentioning

confidence: 99%

Suspected ovarian molar pregnancy after assisted reproductive technology conception: a diagnostic challenge

Obeidi¹,

Tchrakian

Saadeh

et al. 2015

BMJ Case Reports

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A 32-year-old patient with primary infertility received in vitro fertilisation (IVF) therapy. Four weeks later she developed intermittent left iliac fossa pain. Transvaginal ultrasound showed an empty uterus and an adnexal mass adjacent to the right ovary. Serum β-human chronic gonadotropin was 33 492 IU/L. At laparoscopy a mass attached to right ovary, suggestive of a right ovarian ectopic pregnancy, was excised. Histological examination confirmed an ovarian ectopic gestation, but noted enlarged chorionic villi and trophoblastic atypia, which raised the suspicion of molar pregnancy. Subsequent p57 immunohistochemistry and DNA ploidy studies excluded a mole, however. Cases of suspected molar disease in ectopic pregnancy present a diagnostic challenge for both clinicians and histopathologists, and establishing a definitive diagnosis may be difficult.

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“…We present an exceptionally rare case of a 19-year-old female with a partial molar pregnancy, thyrotoxicosis, and OHSS. Suziki et al completed a 2014 literature review describing the eight ever-reported molar pregnancy OHSS cases, three of which were partial molar pregnancies [2]. Of these three partial molar OHSS cases, only one was stated to have occurred with concurrent thyrotoxicosis [2].…”

Section: Discussionmentioning

confidence: 99%

“…Clinically evident OHSS has an incidence of 1%-5% in patients who undergo assisted reproductive technology, with 0.5%-1.0% of women requiring hospital admission for OHSS management [1]. OHSS has infrequently been reported to be caused by the following etiologies: high human chorionic gonadotropin (hCG) levels in a multifetal pregnancy, high hCG levels in a complete/partial/invasive molar pregnancy, or hypothyroidism with high thyroid stimulating hormone (TSH) levels acting similarly as hCG [2]. The classic signs and symptoms of OHSS include nausea, vomiting, bloating, abdominal pain, tachycardia, tachypnea, and dyspnea.…”

Section: Introductionmentioning

confidence: 99%

A Comparison of the Clinical Presentation of Ovarian Hyperstimulation Syndrome in a Partial Molar Pregnancy Case Versus a Fertility Treatment Case

Cohen

Lanzer

Mittal

2019

Cureus

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Ovarian hyperstimulation syndrome (OHSS) is ovarian enlargement secondary to hormones overstimulating ovarian growth. It can be associated with a spectrum of other clinical findings, including ascites, hemoconcentration, hypercoagulability, and electrolyte imbalances. OHSS most commonly occurs as a complication of treatment with in vitro fertilization medications, such as human chorionic gonadotropin (hCG) or gonadotropin-releasing hormone agonists. OHSS has infrequently been reported to be caused by high hCG levels in complete, partial, or invasive molar pregnancies. The classic signs and symptoms of OHSS include nausea, vomiting, bloating, abdominal pain, tachycardia, tachypnea, and dyspnea. Further positive diagnostic studies for OHSS include enlarged ovaries, ascites, hemoconcentration, hyponatremia, hyperkalemia, and oliguria. OHSS due to molar pregnancies is extremely rare. Suziki et al. performed a literature review in 2014 and describe the eight ever-reported molar pregnancy-associated OHSS cases, three of which were partial molar pregnancies. We present a two-case comparison that first examines an exceptionally rare OHSS case presentation of a 19-year-old female with a partial molar pregnancy that was also complicated by hCG-induced thyrotoxicosis. Following this, we discuss a case of the more classic presentation of OHSS caused by fertility treatments. This case report is of novel interest because we present a case comparison that emphasizes a rare, paradoxical association between OHSS and dilation-evacuation procedures that is important for physicians to be aware of - OHSS is not an adverse event of molar pregnancies that can be eliminated by declining hCG levels after a dilation and evacuation procedure; rather, in a molar pregnancy, OHSS occurs after the dilation and evacuation.

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Ovary hyperstimulation syndrome accompanying molar pregnancy: case report and review of the literature

Abstract: We must be aware that OHSS can occur during molar pregnancy, and can be exacerbated after D&E.

Cited by 9 publications

References 11 publications

Placental mesenchymal dysplasia associated with spontaneous ovarian hyperstimulation syndrome

Placental mesenchymal dysplasia associated with spontaneous ovarian hyperstimulation syndrome

Suspected ovarian molar pregnancy after assisted reproductive technology conception: a diagnostic challenge

A Comparison of the Clinical Presentation of Ovarian Hyperstimulation Syndrome in a Partial Molar Pregnancy Case Versus a Fertility Treatment Case

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