CASE REPORT: Ovarian Hyperstimulation Syndrome Complicating a Spontaneous Singleton Pregnancy: A Case Report

University of Ulsan, Asan Medical Center
Journal of Assisted Reproduction and Genetics (Impact Factor: 1.72). 01/2001; 18(2):120-123. DOI: 10.1023/A:1026543027300


It has been known that most cases of ovarian hyperstimulation syndrome (OHSS) are associated with the use of exogenous gonadotropins to induce multiple ovulation. However, OHSS is infrequently associated with a spontaneous ovulatory cycle, usually in the case of multiple gestations, hypothyroidism, or polycystic ovarian syndrome. We report a case of severe OHSS in a spontaneously pregnant woman with no underlying disease.

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Available from: Yoon-Seok Chang
    • "Elevated TSH in combination with normal serum FSH levels may get activated with FSH receptors, causing profound stimulation of ovarian follicles. Although it is not clear whether a similar action is operative in humans, the occurrence of spontaneous ovarian hyperstimulation syndrome in pregnant1528–30 and nonpregnant patients122930 with hypothyroidism supports this theory. "
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    ABSTRACT: Primary hypothyroidism may be associated with ovarian enlargement and/ or cyst formation. We evaluated the effect of thyroid hormone replacement therapy on hormonal changes, ovarian volume and sonographic appearance. Open, prospective study of women admitted to university gynecology clinic. The study included 26 patients with untreated hypothyroidism who had polycystic (n=10) or normal-appearing (n=16) ovaries and 20 euthyroidic controls. Basal serum total testosterone, free testosterone, androstenedione, dehydroepiandosterone-sulfate, prolactin, estradiol, luteinizing hormone, follicle-stimulating hormone, free T3, free T4 and thyroid-stimulating horone, together with ovarian volumes, were determined and repeated after euthyroidism was achieved. Ovarian volumes of patients with hypothyroidism were significantly greater compared with controls, and their magnitudes diminished significantly during thyroid hormone replacement therapy. Hypothyroidic patients with polycystic ovaries had significantly higher serum free testosterone and dehydroepiandosterone-sulfate, but lower androstenodione levels compared with those who had normal-appearing ovaries. Serum total testosterone concentrations were significantly higher in hypothyroidic patients without polycystic ovaries, and thyroid hormone replacement therapy achieved a significant reduction in total as well as free testosterone. Severe longstanding hypothyroidism leads to increased ovarian volume and/or cyst formation. A decrease in ovarian volume, resolution of ovarian cysts and reversal of the polycystic ovary syndrome-like appearance, together with improvement in serum hormone levels, occurred after euthyroidism was achieved.
    No preview · Article · Mar 2011 · Annals of Saudi medicine
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    • "previously described byChae et al. (2001). In the present study, the patients with spontaneous HL had no activating FSHR mutations (Thr449Ile/Ala, Ile545Thr or Asp567Asn/ Gly). "
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    ABSTRACT: Severe ovarian hyperstimulation syndrome (OHSS) is a potentially life-threatening complication during assisted reproduction technology (ART). The aetiology of this condition is still not fully understood. Several gene variations in the FSH receptor (FSHR) gene have been identified for the very rare cases of spontaneous OHSS. There are only few published data on gene variations in sterility and iatrogenic OHSS and no data regarding aromatase (cytochrome P450 19A1; CYP19A1). Ninety-one ART patients with OHSS, eighty-eight ART patients without OHSS and ninety-seven women with assumed normal fecundity were analysed for the FSHR single nucleotide polymorphism (SNP) gene variations Asn680Ser (rs6166), Ala189Val, Ile160Thr, Thr449Ile (rs28928870) and the CYP19A1 rs10046 locus using real-time PCR. In addition, exon 10 of FSHR of two patients with spontaneous hyperreactio luteinalis (HL) was sequenced. Significantly lower frequencies of homozygous Ser680/Ser680 (P=0.035) and heterozygous Thr160/Ile160 (P=0.039) were found in patients with normal fecundity than those undergoing ART. The Ile160Thr SNP with a frequency of 6.7 and 6.1% in ART patients with and without OHSS respectively does not represent a rare mutation as previously published. There were no differences in the frequencies of all other gene variations. Of two patients with HL, both had homozygous point mutations for Ser680/Ser680 and one was heterozygous for Ile160Thr and CYP19A1 rs10046. The FSHR gene variations Asn680Ser as well as Ile160Thr may be contributing factors in unexplained sterility. The other FSHR coding gene variations and CYP19A1 rs10046 investigated are most likely not involved in the aetiology of iatrogenic OHSS or sterility.
    Preview · Article · Feb 2008 · Reproduction (Cambridge, England)
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    • "In severe OHSS, massive ovarian enlargement, ascites, pleural effusion, haemoconcentration, oliguria, hypovolaemia, renal failure, thromboembolic episodes , adult respiratory distress syndrome, and death have been reported [1]. Spontaneous OHSS has been reported in women with hypothyroidism [2] [3] [4] [5] [6], polycystic ovary syndrome and pregnancy [7], gonadotroph pituitary adenoma [8], and normal pregnancy [9]. To our knowledge, only three cases of spontaneous OHSS have been described in non pregnant women with primary hypothyroidism [2] [3] [4]. "
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    ABSTRACT: A 22-year-old patient presented with abdominal pain and massive ovarian enlargement secondary to severe long standing hypothyroidism, mimicking an ovarian cancer. Treatment with L-thyroxin caused marked regression of the tumor.
    Full-text · Article · Feb 2004 · European Journal of Obstetrics & Gynecology and Reproductive Biology
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