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Yano K, Nakai G, Matsutani H, Yamada T, Ohmichi M, Yamamoto K, Osuga K. A FSH-secreting pituitary adenoma discovered after ovarian hyperstimulation syndrome: a case report, illustrating pitfalls in the interpretation of serum FSH levels. BMC Womens Health 2024; 24:650. [PMID: 39709410 DOI: 10.1186/s12905-024-03504-2] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/19/2024] [Accepted: 12/09/2024] [Indexed: 12/23/2024] Open
Abstract
BACKGROUND Most cases of ovarian hyperstimulation syndrome (OHSS) are caused by infertility treatment using human menopausal gonadotropin (HMG) and human chorionic gonadotropin (hCG). OHSS is widely known to have a "spoke-wheel" appearance on imaging, presenting as bilateral symmetric enlargement of ovaries with multiple cysts of varying sizes. When this spoke-wheel appearance is observed in patients not undergoing infertility treatment, tumor-derived hormones such as follicle-stimulating hormone (FSH) and hCG should be measured. However, pitfalls exist in the interpretation of FSH levels. CASE PRESENTATION A 29-year-old, gravida 0, para 0 woman visited her local doctor for irregular menstruation and to seek fertility treatment. At the first medical examination, bilateral ovarian tumors were found by ultrasonography, and she was referred to our hospital. Magnetic resonance imaging (MRI) findings of the bilateral ovarian tumors suggested typical OHSS, and thus levels of serum hormones including FSH and hCG were measured to determine whether endogenous follicle-stimulating hormones were the cause. Estradiol was elevated at 737 pg/ml (normal: 28.8-196.8 pg/ml in follicular phase) and luteinizing hormone (LH) was low at < 0.3 mIU/ml (normal: 1.4-15 in follicular phase, 2.1-88 mIU/ml in ovulatory phase). FSH (18.6 mIU/ml; normal: 3.0-14.7 in follicular phase, 4.5-22.5 mIU/ ml) and hCG (< 1.0 mIU/ml) were within normal ranges for non-pregnant women. Initially, since ovarian neoplasms producing estrogen were suspected, surgical resection was scheduled. However, computed tomography of the neck to pelvic region was performed to rule out metastatic ovarian tumors, and indicated a coincidental pituitary lesion, which was pathologically characterized as an FSH-secreting pituitary adenoma. Consequently, the final diagnosis was OHSS caused by an FSH-producing pituitary adenoma and the scheduled ovarian surgery was avoided. CONCLUSIONS Awareness of MRI findings of OHSS is important to avoid unnecessary invasive procedures. When treating patients who have suspected OHSS on imaging but whose serum FSH is in the normal range, it is also important to know that an unsuppressed FSH level despite the negative feedback effect of high estrogen should prompt investigation for a pituitary adenoma as a primary consideration.
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Affiliation(s)
- Keigo Yano
- Department of Diagnostic Radiology, Osaka Medical and Pharmaceutical University, 2-7 Daigaku-machi, Takatsuki City, Osaka, 569-8686, Japan
| | - Go Nakai
- Department of Diagnostic Radiology, Osaka Medical and Pharmaceutical University, 2-7 Daigaku-machi, Takatsuki City, Osaka, 569-8686, Japan.
| | - Hiroki Matsutani
- Department of Diagnostic Radiology, Osaka Medical and Pharmaceutical University, 2-7 Daigaku-machi, Takatsuki City, Osaka, 569-8686, Japan
| | - Takashi Yamada
- Department of Pathology, Osaka Medical and Pharmaceutical University, 2-7 Daigaku-machi, Takatsuki City, Osaka, 569-8686, Japan
| | - Masahide Ohmichi
- Department of Obstetrics and Gynecology, Osaka Medical and Pharmaceutical University, 2-7 Daigaku-machi, Takatsuki City, Osaka, 569-8686, Japan
| | - Kazuhiro Yamamoto
- Department of Diagnostic Radiology, Osaka Medical and Pharmaceutical University, 2-7 Daigaku-machi, Takatsuki City, Osaka, 569-8686, Japan
| | - Keigo Osuga
- Department of Diagnostic Radiology, Osaka Medical and Pharmaceutical University, 2-7 Daigaku-machi, Takatsuki City, Osaka, 569-8686, Japan
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Daolio J, Sperduti S, Casarini L, Falbo A, Materazzo C, Aguzzoli L, Villani MT. Spontaneous and iatrogenic ovarian hyperstimulation syndrome in the absence of FSHR mutations: a case report of two unexpected cases. BMC Med Genomics 2023; 16:45. [PMID: 36882810 PMCID: PMC9990314 DOI: 10.1186/s12920-023-01473-3] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/23/2022] [Accepted: 02/27/2023] [Indexed: 03/09/2023] Open
Abstract
BACKGROUND Ovarian hyperstimulation syndrome (OHSS) is a complication of controlled ovarian hyperstimulation (COH). It is a potentially life-threatening condition that usually occurs either after human chorionic gonadotropins (hCG) administration in susceptible patients or as a result of an implanting pregnancy, regardless of whether it was achieved by natural conception or infertility treatments. Despite many years of clinical experience regarding the adoption of preventive measures and the identification of patients at high risk, the pathophysiology of OHSS is poorly understood and no reliable predictive risk factors have been identified. CASES PRESENTATION We report about two unexpected cases of OHSS following infertility treatments, occurring after freeze-all strategy with embryo cryopreservation approaches. The first case developed spontaneous OHSS (sOHSS), despite efforts to prevent its manifestation by a segmentation approach, including frozen embryo replacement cycle. The second case developed a late form of iatrogenic OHSS (iOHSS), even though the absence of any risk factors. No mutations in the follicle-stimulating hormone (FSH) receptor (FSHR)-encoding gene were detected, suggesting that the high levels of hCG due to the twin implanting pregnancies could be the only triggering factor of OHSS outbreak. CONCLUSION Freeze-all strategy with embryo cryopreservation cannot entirely prevent the development of OHSS, which may occur in its spontaneous form independently from the FSHR genotype. Although OHSS remains a rare event, all infertile patients requiring ovulation induction or controlled ovarian stimulation (COS) may be at potential risk of OHSS, either in the presence or in the absence of risk factors. We suggest closely monitoring cases of pregnancy following infertility treatments in order to provide early diagnosis and adopt the conservative management.
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Affiliation(s)
- Jessica Daolio
- Quality and Accreditation Office, Medical Directorate ASMN, Azienda Unità Sanitaria Locale - IRCCS di Reggio Emilia, viale Umberto I 50, 42123, Reggio Emilia, Italy.
| | - Samantha Sperduti
- Unit of Endocrinology, Department of Biomedical, Metabolic and Neural Sciences, University of Modena and Reggio Emilia, via Campi 287, 41125, Modena, Italy
- Center for Genomic Research, University of Modena and Reggio Emilia, via Campi 287, 41125, Modena, Italy
| | - Livio Casarini
- Unit of Endocrinology, Department of Biomedical, Metabolic and Neural Sciences, University of Modena and Reggio Emilia, via Campi 287, 41125, Modena, Italy
- Center for Genomic Research, University of Modena and Reggio Emilia, via Campi 287, 41125, Modena, Italy
| | - Angela Falbo
- Department of Obstetrics & Gynaecology, Azienda Unità Sanitaria Locale - IRCCS di Reggio Emilia, viale Risorgimento 80, 42123, Reggio Emilia, Italy
| | - Caterina Materazzo
- Department of Obstetrics & Gynaecology, Azienda Unità Sanitaria Locale - IRCCS di Reggio Emilia, viale Risorgimento 80, 42123, Reggio Emilia, Italy
| | - Lorenzo Aguzzoli
- Department of Obstetrics & Gynaecology, Azienda Unità Sanitaria Locale - IRCCS di Reggio Emilia, viale Risorgimento 80, 42123, Reggio Emilia, Italy
| | - Maria Teresa Villani
- Department of Obstetrics & Gynaecology, Azienda Unità Sanitaria Locale - IRCCS di Reggio Emilia, viale Risorgimento 80, 42123, Reggio Emilia, Italy
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Muacevic A, Adler JR, Elmoheen R. Spontaneous Ovarian Hyperstimulation Syndrome Associated With Primary Hypothyroidism. Cureus 2023; 15:e33247. [PMID: 36606102 PMCID: PMC9808876 DOI: 10.7759/cureus.33247] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 01/02/2023] [Indexed: 01/03/2023] Open
Abstract
Ovarian hyperstimulation syndrome is a rare condition in pregnant women. Most cases are associated with the use of ovulation induction and stimulation medications. Some studies have reported cases of this condition in non-pregnant women or women undergoing ovulation therapy. In this case report, we report the case of a 27-year-old pregnant Saudi woman presenting with a picture of severe spontaneous ovarian hyperstimulation syndrome secondary to severe undiagnosed hypothyroidism. Treatment with Eltroxin (thyroxine) led to complete improvement and regression of ovarian enlargement after empirical titrating thyroxin replacement therapy, which proved the presence of this causation. The diagnosis was confirmed by laboratory and imaging findings, which helped prompt management and prevented complications of unneeded surgical intervention.
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Alqurashi RM, Alsuwat SA, Yamani MA, Omar SY. Onset of Spontaneous Ovarian Hyperstimulation Syndrome in the Third Trimester: Case Report. Cureus 2022; 14:e20940. [PMID: 35004087 PMCID: PMC8727944 DOI: 10.7759/cureus.20940] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 01/04/2022] [Indexed: 11/25/2022] Open
Abstract
Ovarian hyperstimulation syndrome (OHSS) is a rare and occasionally fatal complication of ovulation induction. However, OHSS has occurred without interventional ovulation induction and in spontaneous ovulatory cycles. In most reported cases, physiological production of human chorionic gonadotropin was high, as in cases of multiple gestation, molar pregnancy, polycystic ovarian syndrome, and hypothyroidism. We report a very rare case of spontaneous OHSS in a healthy 36-year-old woman, gravida 5 para 2 + 2, 35 weeks pregnant, who had become pregnant naturally. According to our research, this is the first report of a case that occurred in the third trimester.
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Oliveira e Souza L, Innecco Arêas JV, Rezende Campos MC, Innecco Arêas I, Martins Resende BA. Spontaneous ovarian hyperstimulation syndrome in a pregnant woman with hypothyroidism: a case report. F S Rep 2021; 2:433-439. [PMID: 34934984 PMCID: PMC8655400 DOI: 10.1016/j.xfre.2021.07.004] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/12/2021] [Revised: 07/17/2021] [Accepted: 07/19/2021] [Indexed: 01/19/2023] Open
Abstract
Objective To study a rare case of spontaneous ovarian hyperstimulation syndrome in a naturally conceived pregnancy associated with overt hypothyroidism. Design Case report. Setting Endocrinology private practice. Patient(s) A 32-year-old woman who was 13 weeks pregnant with bilaterally enlarged ovaries presumed to be secondary to hypothyroidism. Intervention(s) Administration of levothyroxine and titration of the dose. Main Outcome Measure(s) Regression of signs and symptoms of spontaneous ovarian hyperstimulation syndrome after 12 weeks of therapy. Result(s) The patient was diagnosed with severe hypothyroidism, as confirmed by her elevated thyroid-stimulating hormone level. Ultrasound evaluation revealed ovarian enlargement secondary to multiple contiguous cysts with anechoic content. The patient was administered levothyroxine 175 μg/day. Results of hormonal studies demonstrated thyroid function normality at week 12 after treatment. Incomplete regression of ovarian cysts was also noticed within this period. At week 37, the patient developed preeclampsia, and cesarean delivery was recommended. An 8-month postpartum ultrasound evaluation revealed complete regression of the cysts. Conclusion(s) Spontaneous ovarian hyperstimulation syndrome secondary to hypothyroidism may be the cause of ovarian enlargement, and levothyroxine replacement seems an appropriate primary therapeutic option. Proper endocrinological assessment of patients is recommended as it may avoid unfavorable outcomes.
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Affiliation(s)
- Lucas Oliveira e Souza
- Department of Medicine, Minas Gerais Faculty of Medical Sciences, Belo Horizonte, Brazil
- Reprint requests: Lucas Oliveira e Souza, M.S., Al. Ezequiel Dias, 275 Centro, Belo Horizonte, Brazil.
| | | | | | - Isabela Innecco Arêas
- Department of Medicine, Minas Gerais Faculty of Medical Sciences, Belo Horizonte, Brazil
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Gui J, Zhang J, Xu WM, Ming L. Spontaneous ovarian hyperstimulation syndrome: Report of two cases. World J Clin Cases 2019; 7:4384-4390. [PMID: 31911922 PMCID: PMC6940325 DOI: 10.12998/wjcc.v7.i24.4384] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/05/2019] [Revised: 11/01/2019] [Accepted: 11/23/2019] [Indexed: 02/05/2023] Open
Abstract
BACKGROUND Spontaneous 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 SUMMARY Here 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.
CONCLUSION Patients 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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Affiliation(s)
- Juan Gui
- Reproductive Center, Renmin Hospital of Wuhan University, Wuhan 430060, Hubei Province China
- Assisted Reproduction and Embryogenesis Clinical Research Center of Hubei Province, Wuhan 430060, Hubei Province, China
| | - Jie Zhang
- Reproductive Center, Renmin Hospital of Wuhan University, Wuhan 430060, Hubei Province China
| | - Wang-Ming Xu
- Reproductive Center, Renmin Hospital of Wuhan University, Wuhan 430060, Hubei Province China
- Assisted Reproduction and Embryogenesis Clinical Research Center of Hubei Province, Wuhan 430060, Hubei Province, China
| | - Lei Ming
- Reproductive Center, Renmin Hospital of Wuhan University, Wuhan 430060, Hubei Province China
- Assisted Reproduction and Embryogenesis Clinical Research Center of Hubei Province, Wuhan 430060, Hubei Province, China
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Morotti E, Battaglia C. Oligosypthomatic ovarian hyperstimulation syndrome in a spontaneous uneventful pregnancy. A case report. Gynecol Endocrinol 2019; 35:657-660. [PMID: 30806108 DOI: 10.1080/09513590.2019.1579791] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 10/27/2022] Open
Abstract
Spontaneous hyperstimulation syndrome (spOHSS) is a rare event in pregnancies following natural conception. Only a few cases are reported in the scientific literature and result associated with massive ovarian enlargement with multiple cysts, dehydration, and fluid accumulation in the "third space". We report a novel case of spOHSS in a naturally conceived singleton pregnancy. The spOHSS was characterized by massive ovarian enlargement with multiple cysts. No dehydration and no signs of ascites were observed. The pregnancy uneventful evolved up to term.
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Affiliation(s)
- Elena Morotti
- a Department of Obstetrics and Gynecology , Alma Mater Studiorum-University of Bologna , Bologna , Italy
| | - Cesare Battaglia
- a Department of Obstetrics and Gynecology , Alma Mater Studiorum-University of Bologna , Bologna , Italy
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8
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Hirshberg B, Rheinboldt M. Multimodality imaging of acute locoregional and systemic complications in the setting of assisted reproduction. Emerg Radiol 2019; 26:205-219. [PMID: 30631995 DOI: 10.1007/s10140-018-01665-w] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/26/2018] [Accepted: 12/13/2018] [Indexed: 02/02/2023]
Abstract
Over the past 40 years since the first in vitro fertilization was performed, both the role of assisted reproductive technology (ART) in establishing viable pregnancy as well as the available treatment options have expanded enormously. Annually in the USA, nearly 2% of pregnancies now employ some form of ART assistance, with in vitro fertilization (IVF) being the most commonly utilized methodology. Both maternal and fetal risks are elevated in ART pregnancies, the latter including adverse outcome due to both increased gestational number as well as advanced maternal age. Maternal risks may be divided into locoregional and systemic complications. Adverse pelvic complications include those relating to gamete harvesting and transfer, ovarian hyperstimulation, the sequela of ectopic and heterotopic pregnancies, as well as ovarian torsion, all of which are elevated in the ART cohort. Ovarian hyperstimulation syndrome is the most commonly encountered complication, with both systemic and pelvic features relating to increased vascular permeability, hemoconcentration, and ascites. While life-threatening cases are relatively rare, moderate and severe manifestations may occur in up to 10% of ART cycles and, as such, are a not infrequent cause for ER visitation. Familiarity with the clinical and imaging manifestations of ART complications as well as their prognostic implications will facilitate a timely diagnosis and assist the interpreting radiologist in best expediting appropriate clinical care. In this article, we will briefly discuss the current methodology of ART then present an imaging-based multimodality review of the potentially encountered adverse maternal sequela, highlighting key diagnostic features and differential considerations as well as potential prognostic implications.
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9
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Kim SJ, Yoon JH, Kim HK, Kang HC. Spontaneous ovarian hyperstimulation syndrome in a young female subject with a lingual thyroid and primary hypothyroidism. Korean J Intern Med 2017; 32:559-562. [PMID: 27510824 PMCID: PMC5432798 DOI: 10.3904/kjim.2015.372] [Citation(s) in RCA: 8] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/17/2015] [Revised: 03/29/2016] [Accepted: 05/15/2016] [Indexed: 12/16/2022] Open
Affiliation(s)
- Soo Jeong Kim
- Department of Internal Medicine, Chonnam National University Hwasun Hospital, Hwasun, Korea
| | - Jee Hee Yoon
- Department of Internal Medicine, Chonnam National University Hwasun Hospital, Hwasun, Korea
| | - Hee Kyung Kim
- Department of Internal Medicine, Chonnam National University Hwasun Hospital, Hwasun, Korea
| | - Ho-Cheol Kang
- Department of Internal Medicine, Chonnam National University Hwasun Hospital, Hwasun, Korea
- Correspondence to Ho-Cheol Kang M.D. Department of Internal Medicine, Chonnam National University Hwasun Hospital, 322 Seoyang-ro, Hwasun 58128, Korea Tel: +82-61-379-7620 Fax: +82-61-379-7628 E-mail:
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10
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Cabar FR. Ovarian hyperstimulation syndrome in a spontaneous singleton pregnancy. ACTA ACUST UNITED AC 2017; 14:231-4. [PMID: 27223308 PMCID: PMC4943359 DOI: 10.1590/s1679-45082016rc3429] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/30/2015] [Accepted: 09/30/2015] [Indexed: 11/21/2022]
Abstract
The ovarian hyperstimulation syndrome is the combination of increased ovarian volume, due to the presence of multiple cysts and vascular hyperpermeability, with subsequent hypovolemia and hemoconcentration. We report a case of spontaneous syndrome in a singleton pregnancy. This was a spontaneous pregnancy with 12 weeks of gestational age. The pregnancy was uneventful until 11 weeks of gestational age. After that, the pregnant woman complained of progressive abdominal distention associated with abdominal discomfort. She did not report other symptoms. In the first trimester, a routine ultrasonography showed enlarged ovaries, multiples cysts and ascites. Upon admission, the patient was hemodynamically stable, her serum β-hCG was 24,487mIU/mL, thyroid-stimulating hormone was 2.2µUI/mL and free T4 was 1.8ng/dL. All results were within normal parameters. However, levels of estradiol were high (10,562pg/mL). During hospitalization, she received albumin, furosemide and prophylactic dose of enoxaparin. The patient was discharged on the sixth hospital day.
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11
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Halupczok J, Bidzińska-Speichert B, Lenarcik-Kabza A, Zieliński G, Filus A, Maksymowicz M. Gonadotroph adenoma causing ovarian hyperstimulation syndrome in a premenopausal woman. Gynecol Endocrinol 2014; 30:774-7. [PMID: 24971660 DOI: 10.3109/09513590.2014.934668] [Citation(s) in RCA: 13] [Impact Index Per Article: 1.2] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/13/2022] Open
Abstract
INTRODUCTION Gonadotroph adenomas occur commonly in middle-aged adults without any specific endocrinological symptoms. To date, only 30 cases of gonadotropinoma causing ovarian hyperstimulation syndrome in pre-menopausal women have been reported. CASE REPORT A 37-year old woman with pituitary macroadenoma and hyperprolactinaemia was admitted to the Department of Endocrinology, Diabetology and Isotope Therapy. She presented with recurrent ovarian cysts, menstrual disturbances, headaches, visual impairment and galactorrhea. Her endocrine profile showed normal values of FSH, elevated concentrations of estradiol and suppressed LH levels. Transsphenoidal resection of the tumor tissue resulted in normalization of the hormone values and improvement in the clinical picture. CONCLUSIONS Gonadotroph adenomas should be considered in the differential diagnosis in premenopausal women with OHSS.
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Affiliation(s)
- Jowita Halupczok
- Department of Endocrinology, Diabetology and Isotope Therapy, Wroclaw Medical University , Wroclaw , Poland
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12
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Ovarian Hyper Stimulation Syndrome in Two Spontaneous Pregnancies. RAZAVI INTERNATIONAL JOURNAL OF MEDICINE 2014. [DOI: 10.5812/rijm.14245] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/16/2022] Open
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13
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Sridev S, Barathan S. Case report on spontaneous ovarian hyperstimulation syndrome following natural conception associated with primary hypothyroidism. J Hum Reprod Sci 2013; 6:158-61. [PMID: 24082659 PMCID: PMC3778607 DOI: 10.4103/0974-1208.117164] [Citation(s) in RCA: 17] [Impact Index Per Article: 1.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/15/2012] [Revised: 02/07/2013] [Accepted: 04/13/2013] [Indexed: 11/04/2022] Open
Abstract
Spontaneous OHSS is a rare event in pregnancy following natural conception and only a few cases have been reported in the literature so far. This report is a case of spontaneously conceived pregnancy with Spontaneous OHSS and Primary hypothyroidism.
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Affiliation(s)
- Smisha Sridev
- Department of Reproductive Medicine, Parvathi Nursing Home, KK Nagar, Chennai, Tamil Nadu, India
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14
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Uchida S, Uchida H, Maruyama T, Kajitani T, Oda H, Miyazaki K, Kagami M, Yoshimura Y. Molecular analysis of a mutated FSH receptor detected in a patient with spontaneous ovarian hyperstimulation syndrome. PLoS One 2013; 8:e75478. [PMID: 24058690 PMCID: PMC3772932 DOI: 10.1371/journal.pone.0075478] [Citation(s) in RCA: 27] [Impact Index Per Article: 2.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/13/2012] [Accepted: 08/20/2013] [Indexed: 11/18/2022] Open
Abstract
Spontaneous ovarian hyperstimulation syndrome (sOHSS) is a rare event that may result from a FSH-producing pituitary adenoma (FSHoma), activating mutations of the FSH receptor (FSHR), and cross-reactivity of the FSHR to elevated hCG and TSH in the setting of pregnancy or hypothyroidism. The objective of this study was to investigate whether an aberrant FSHR was present in a woman with sOHSS and a non-surgically diagnosed FSHoma whose serum FSH levels and FSH bioactivity were nearly normal. Sequencing of the patient's FSHR gene revealed a heterozygous novel missense mutation c. 1536G>A resulting in an amino acid substitution M512I. We asked whether this mutant FSHR affected FSHR-mediated signaling pathways involving cAMP/protein kinase A (PKA), phosphatidylinositol-3 kinase (PI3K)/protein kinase B (AKT) and v-src sarcoma (Schmidt-Ruppin A-2) viral oncogene homolog kinase (SRC)/ p42/p44 extracellular signal-regulated protein kinases (ERK1/2). Thus, 293T cells expressing wild-type (FSHRwt), the mutant FSHR (FSHRmt), or both (FSHRwt/mt) were treated with FSH and subjected to measurements of intracellular cAMP, cAMP-induced CRE (cAMP response element)-mediated luciferase assays and immunoblot analyses of phosphorylated PI3K and ERK1/2. There were no differences in luciferase activities or phosphorylation levels of ERK1/2 among FSHRwt, FSHRmt cells and FSHwt/mt cells. However, FSHRmt cells showed a significant reduction in both cAMP production and PI3K phosphorylation levels with unchanged phosphorylation of ERK1/2 upon FSH stimulation in comparison to FSHwt cells. Also, FSH treatment did not provoke PI3K phosphorylation in FSHwt/mt cells. These results indicate that the novel missense M512I FSHR mutation identified herein did not participate in hyperactivation of FSHR-mediated signaling pathways but rather in hypoactivation of the FSH-mediated PI3K/AKT pathway. Thus, this study demonstrates a new functional property of this novel mutatnt FSHR, which, however, might not be involved in the pathogenesis of sOHSS in this FSHoma patient.
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Affiliation(s)
- Sayaka Uchida
- Department of Obstetrics and Gynecology, Keio University School of Medicine, Tokyo, Japan
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15
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Di Carlo C, Savoia F, Ferrara C, Tommaselli GA, Bifulco G, Nappi C. Case report: a most peculiar family with spontaneous, recurrent ovarian hyperstimulation syndrome. Gynecol Endocrinol 2012; 28:649-51. [PMID: 22313155 DOI: 10.3109/09513590.2011.650763] [Citation(s) in RCA: 14] [Impact Index Per Article: 1.1] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/13/2022] Open
Abstract
Ovarian hyperstimulation syndrome (OHSS) mostly occurs as a complication of induction of ovulation. However, rarely, it may occur spontaneously at the beginning of a natural pregnancy and in the absence of any assisted reproductive treatment. Spontaneous OHSS has been reported in pregnant women affected by hypothyroidism, polycystic ovary syndrome, gonadotropin-producing pituitary adenoma, but also in normal pregnancies. The aetiology of this condition is still unclear. We hereby report on a case of spontaneous, familial, recurrent OHSS in a 26-year-old primipara whose first-degree cousin, paternal grandmother and a number of other members of her father's family had suffered from a similar condition. This case seems to support the concept of a genetic predisposition for spontaneous OHSS thereby suggesting the need for preventive and therapeutic strategies.
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Affiliation(s)
- Costantino Di Carlo
- Department of Obstetrics and Gynecology, University of Naples Federico II, Naples, Italy.
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16
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Irvine LM. Spontaneous ovarian hyperstimulation syndrome (OHSS): a rare but important differential diagnosis for abdominal distension in early pregnancy. J OBSTET GYNAECOL 2011; 31:338-9. [PMID: 21534759 DOI: 10.3109/01443615.2011.560299] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/13/2022]
Affiliation(s)
- L M Irvine
- Department of Obstetrics and Gynaecology, Watford General Hospital, Watford, UK.
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Abstract
Hyperandrogenic states in pregnancy are almost always the result of a condition that arises during pregnancy. The onset of virilization symptoms is often very fast. The mother is protected against hyperandrogenism by a high level of SHBG, by placental aromatase and a high level of progesterone. The fetus is protected from the mother’s hyperandrogenism partly by the placental aromatase, that transforms the androgens into estrogens, and partly by SHGB. Nevertheless there is a significant risk of virilization of the female fetus if the mother’s hyperandrogenic state is serious. The most frequent cause of hyperandrogenic states during pregnancy are pregnancy luteoma and hyperreactio luteinalis. Hormonal production is evident in a third of all luteomas, which corresponds to virilization in 25-35 % of mothers with luteoma. The female fetus is afflicted with virilization with two thirds of virilized mothers. Hyperreactio luteinalis is created in connection with a high level of hCG, e.g. during multi-fetus pregnancies. This condition most frequently arises in the third trimester, virilization of the mother occurs in a third of cases. Virilization of the fetus has not yet been described. The most serious cause of hyperandrogenism is represented by ovarian tumors, which are fortunately rare.
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Affiliation(s)
- N. KAŇOVÁ
- Institute of Endocrinology, Prague, Czech Republic
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Muderris II, Boztosun A, Oner G, Bayram F. Effect of thyroid hormone replacement therapy on ovarian volume and androgen hormones in patients with untreated primary hypothyroidism. Ann Saudi Med 2011; 31:145-51. [PMID: 21403408 PMCID: PMC3102473 DOI: 10.4103/0256-4947.77500] [Citation(s) in RCA: 33] [Impact Index Per Article: 2.4] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/11/2022] Open
Abstract
BACKGROUND AND OBJECTIVES 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. DESIGN AND SETTING Open, prospective study of women admitted to university gynecology clinic. PATIENTS AND METHODS 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. RESULTS 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. CONCLUSION 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.
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Affiliation(s)
- Iptisam Ipek Muderris
- From the Department of Obstetrics and Gynecology, Erciyes University, Faculty of Medicine, Kayseri, Turkey
| | - Abdullah Boztosun
- Department of Obstetrics and Gynecology, Cumhuriyet University, Faculty of Medicine, Sivas, Turkey
| | - Gokalp Oner
- From the Department of Obstetrics and Gynecology, Erciyes University, Faculty of Medicine, Kayseri, Turkey
| | - Fahri Bayram
- Department of Endocrinology, Erciyes University, Faculty of Medicine, Kayseri, Turkey
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Baba T, Endo T, Kitajima Y, Kamiya H, Moriwaka O, Saito T. Spontaneous ovarian hyperstimulation syndrome and pituitary adenoma: incidental pregnancy triggers a catastrophic event. Fertil Steril 2009; 92:390.e1-3. [DOI: 10.1016/j.fertnstert.2009.02.071] [Citation(s) in RCA: 15] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/13/2009] [Revised: 02/16/2009] [Accepted: 02/25/2009] [Indexed: 10/20/2022]
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Abstract
Ovarian hyperstimulation syndrome (OHSS) is an iatrogenic complication induced by exogenous administration of substances that are used to stimulate follicular growth and ovulation. There has only been one reported incidence of a spontaneous occurrence of OHSS and that was in 1992. The crucial event in the development of the syndrome is the administration of human chorionic gonadotropin (HCG), although several studies have reported the onset of OHSS after gonadotropin stimulation despite withholding HCG. OHSS is represented by a broad spectrum of clinical and laboratory manifestations which, in the severest form, can induce a life-threatening condition.
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21
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Angioni S, Portoghese E, Milano F, Melis GB, Fulghesu AM. Hirsutism and hyperandrogenism associated with hyperreactio luteinalis in a singleton pregnancy: a case report. Gynecol Endocrinol 2007; 23:248-51. [PMID: 17558681 DOI: 10.1080/09513590701214513] [Citation(s) in RCA: 16] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 10/23/2022] Open
Abstract
The incidence of hyperandrogenism during pregnancy is low, although the incidence of some of the ovarian diseases that can cause it is higher. Hyperreactio luteinalis is a rare benign condition that may mimic ovarian and trophoblastic malignancies. A 23-year-old woman at 20 weeks' gestational age presenting with severe hirsutism and ovarian masses was treated conservatively and subsequently gave birth to a healthy female neonate. Final diagnosis was hyperreactio luteinalis. Conservative management with close monitoring of patients with hyperreactio luteinalis represents the best approach in such rare cases. Counseling should be provided to reassure the patient as to the transient effects of hyperandrogenism on the mother and the fetus.
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Affiliation(s)
- Stefano Angioni
- Division of Gynecology, Obstetrics and Pathophysiology of Human Reproduction, Department of Surgery, Maternal-Fetal Medicine, and Imaging, University of Cagliari, Cagliari, Italy.
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22
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Abstract
The Ovarian Hyperstimulation Syndrome (OHSS) represents one of the biggest nightmares of all physicians involved in Assisted Reproductive Technologies (ART). Every year, several hundreds of women are hospitalized and to date several deaths have been reported. The pivotal event in the development of OHSS is the disruption of capillary integrity that results in leakage of intravascular fluid and proteins into third space. On the molecular level, human chorionic godadotropin (HCG) either exogenous or endogenous, functions as the triggering point for the production of vascular endothelial growth factor (VEGF) that is the main mediator to increase permeability on the vascular bed. Spontaneous OHSS has also been reported, either due to inappropriate activation of a mutant FSH receptor or due to very high levels of HCG during pregnancy. The available evidence on the several preventive and therapeutic approaches with special attention to level 1 evidence when available is also presented. OHSS is a self-resolving condition and the main role of the physician is to correct and maintain the intravascular volume, to support renal function and respiration and prevent thrombotic events. An algorithm on the management of OHSS on an outpatient basis and in the hospital is based on the previous mentioned principles.
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Affiliation(s)
- Nikos F Vlahos
- Second Department of Obstetrics and Gynicology, Aretaieion Hospital, National Kapodestrian University of Athens, School of Medicine, Greece.
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23
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Oztekin O, Soylu F, Tatli O. Spontaneous Ovarian Hyperstimulation Syndrome in a Normal Singleton Pregnancy. Taiwan J Obstet Gynecol 2006; 45:272-5. [PMID: 17175480 DOI: 10.1016/s1028-4559(09)60241-2] [Citation(s) in RCA: 18] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/30/2022] Open
Abstract
OBJECTIVE It is known that most cases of ovarian hyperstimulation syndrome (OHSS) are associated with the therapies for ovulation induction. However, OHSS may rarely be associated with a spontaneous ovulatory cycle, usually in the case of multiple gestations, hypothyroidism or polycystic ovary syndrome. CASE REPORT A case of OHSS in a woman who became pregnant naturally and who had no underlying disease is presented here. The patient was managed expectantly with no complications. CONCLUSION Although spontaneous ovarian hyperstimulation is a rare entity, it is important to differentiate it from other causes of ovarian enlargement. Occasionally, life-threatening situations may occur, but it is usually a self-limiting process.
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Affiliation(s)
- Ozer Oztekin
- Department of Obstetrics and Gynecology, Faculty of Medicine, Pamukkale University, Denizli, Turkey.
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24
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Ozden S, Gürbüz B, Yalti S, Ergül B, Ozturkmen M. Ovarian hyperstimulation associated with a spontaneous pregnancy. J OBSTET GYNAECOL 2005; 25:394-5. [PMID: 16091334 DOI: 10.1080/01443610500150577] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/25/2022]
Affiliation(s)
- S Ozden
- Zeynep Kamil Women and children Education and Research Hospital, Istanbul, Turkey
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25
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Shimon I, Rubinek T, Bar-Hava I, Nass D, Hadani M, Amsterdam A, Harel G. Ovarian hyperstimulation without elevated serum estradiol associated with pure follicle-stimulating hormone-secreting pituitary adenoma. J Clin Endocrinol Metab 2001; 86:3635-40. [PMID: 11502789 DOI: 10.1210/jcem.86.8.7766] [Citation(s) in RCA: 42] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/19/2022]
Abstract
We report a unique case of a 28-yr-old woman with a gonadotroph adenoma secreting FSH, presented with ovarian hyperstimulation, without elevation of serum estradiol. She presented with abdominal pain and large ovaries (both 10 cm in diameter) with multiple follicular cysts shortly after discontinuing oral contraceptive pills. She had a supranormal PRL level of 71 microg/liter (normal, <20), FSH of 8.4-9.2 IU/liter (normal for follicular phase, 2.4-10), LH of 0.01 IU/liter (normal, 1.6-9.3), estradiol of 108 pmol/liter (normal for follicular phase, 80-790), and free alpha-subunit level of 0.11 microg/liter (normal, <1.8). A nuclear magnetic resonance study revealed invasive pituitary macroadenoma, 30 mm in diameter. Dopamine agonist (cabergoline) treatment normalized serum PRL but had no affect on FSH levels. A transsphenoidal surgery was performed, and most of the adenoma was resected. One month after surgery the patient resumed menstruation, and the hormonal profile included serum FSH of 6.3 IU/liter, LH of 2.1 IU/liter, estradiol of 156 pmol/liter, and PRL of 10 microg/liter. The excised adenoma tissue exhibited intense immunostaining for FSH and secreted this hormone to culture medium. Stimulation with TRH (both in vivo preoperatively and in vitro study of the excised tumor) had no effect on FSH secretion from the adenoma. Estradiol did not suppress FSH release from cultured adenoma cells. Patient serum samples showed significant FSH bioactivity when tested in a human granulosa cell line. This case is remarkable because the ovarian hyperstimulation related to the FSH-secreting adenoma was not associated with high levels of serum estradiol, probably due to insufficient LH production by the normal pituitary. Thus, it supports the two-cell, two-gonadotropin theory, that both FSH and LH are necessary for normal ovarian estrogen production.
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Affiliation(s)
- I Shimon
- Institutes of Endocrinology, Sheba Medical Center, Tel-Hashomer 52621, Israel.
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26
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Chae HD, Park EJ, Kim SH, Kim CH, Kang BM, Chang YS. Ovarian hyperstimulation syndrome complicating a spontaneous singleton pregnancy: a case report. J Assist Reprod Genet 2001; 18:120-3. [PMID: 11285979 PMCID: PMC3455559 DOI: 10.1023/a:1026543027300] [Citation(s) in RCA: 31] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/12/2022] Open
Abstract
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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Affiliation(s)
- H D Chae
- Division of Reproductive Endocrinology and Infertility, Department of Obstetrics and Gynecology, College of Medicine, University of Ulsan, Asan Medical Center, 388-1, Poongnap-Dong, Songpa-Gu, Seoul, 138-736, Korea
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27
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Akerman FM, Lei Z, Rao CV, Nakajima ST. A case of spontaneous ovarian hyperstimulation syndrome with a potential mutation in the hCG/LH receptor gene. Fertil Steril 2000; 74:403-4. [PMID: 10927070 DOI: 10.1016/s0015-0282(00)00628-2] [Citation(s) in RCA: 17] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/30/2022]
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28
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Välimäki MJ, Tiitinen A, Alfthan H, Paetau A, Poranen A, Sane T, Stenman UH. Ovarian hyperstimulation caused by gonadotroph adenoma secreting follicle-stimulating hormone in 28-year-old woman. J Clin Endocrinol Metab 1999; 84:4204-8. [PMID: 10566673 DOI: 10.1210/jcem.84.11.6138] [Citation(s) in RCA: 37] [Impact Index Per Article: 1.4] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/08/2023]
Abstract
Ovarian hyperstimulation caused by a gonadotroph adenoma in premenopausal women has been described only twice before this report. A 28-yr-old woman presented with menstrual disturbances and pelvic pains that began after stopping the use of contraceptive pills. Transvaginal ultrasound revealed enlarged ovaries with multiple cysts. The patient had elevated serum estradiol (up to 2900 pmol/L; normal, 80-300 pmol/L in the follicular phase) and inhibin (6.4 kU/L; normal, 0.5-2.5 kU/L) levels. Serum LH was appropriately suppressed (0.6 IU/L), but serum FSH varied from 4.9-8.1 IU/L. Both gonadotropins as well as the free alpha-subunit showed a paradoxical response to the stimulus by TRH. A nuclear magnetic resonance study unraveled a pituitary tumor, 12-14 mm in diameter, extending up to the suprasellar cistern. After pituitary surgery, all hormone values normalized, and the patient resumed regular ovulatory cycles. In immunostaining, 20-30% of the cells of the tumor stained positively for FSHbeta. We conclude that a gonadotropin-producing adenoma must be considered in the differential diagnosis of a patient presenting with large multicystic ovaries and high estradiol levels in the absence of exogenous gonadotropins.
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Affiliation(s)
- M J Välimäki
- Department of Medicine, Helsinki University Central Hospital, Finland.
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29
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Todros T, Carmazzi CM, Bontempo S, Gaglioti P, Donvito V, Massobrio M. Spontaneous ovarian hyperstimulation syndrome and deep vein thrombosis in pregnancy: case report. Hum Reprod 1999; 14:2245-8. [PMID: 10469688 DOI: 10.1093/humrep/14.9.2245] [Citation(s) in RCA: 27] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/14/2022] Open
Abstract
This report describes a case of spontaneous ovarian hyperstimulation syndrome (OHSS) occurring in a pregnant woman carrying the factor V Leiden mutation. Even though prophylactic treatment for thrombo-embolism was adopted by administering low molecular weight heparin, the pregnancy was complicated by thromboses of the left subclavian, axillary, humeral and internal jugular veins during the second trimester of gestation. The pregnancy was managed conservatively and a healthy newborn was delivered at term. In order to avoid unnecessary laparotomy, we emphasize the importance of careful diagnosis in order to differentiate spontaneous OHSS from ovarian carcinoma, as well as the necessity to look for the presence of coagulation disorders in women affected by OHSS.
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Affiliation(s)
- T Todros
- Department of Gynecology and Obstetrics, University of Turin, Italy
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30
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Enskog A, Henriksson M, Unander M, Nilsson L, Brännström M. Prospective study of the clinical and laboratory parameters of patients in whom ovarian hyperstimulation syndrome developed during controlled ovarian hyperstimulation for in vitro fertilization. Fertil Steril 1999; 71:808-14. [PMID: 10231037 DOI: 10.1016/s0015-0282(99)00090-4] [Citation(s) in RCA: 121] [Impact Index Per Article: 4.7] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/15/2022]
Abstract
OBJECTIVE To compare patient characteristics and clinical and laboratory parameters in patients in whom ovarian hyperstimulation syndrome (OHSS) develops with those in whom it does not develop. DESIGN Prospective cohort study. SETTING Reproductive medicine unit at a university medical center. PATIENT(S) All patients undergoing IVF (n = 428) who received controlled ovarian hyperstimulation during a 6-month period. INTERVENTION(S) Prospective data collection. MAIN OUTCOME MEASURE(S) Patient characteristics (age, body mass index, medical history, smoking habits) and clinical and laboratory data obtained during controlled ovarian hyperstimulation were evaluated in patients who had severe OHSS, any degree of OHSS, or a significant risk of OHSS and compared with the remaining populations. RESULT(S) Severe OHSS developed in 18 patients (4.2%) and mild or moderate OHSS developed in 7.3%. As a group, all the patients with OHSS were significantly younger, received lower doses of gonadotropins, had ovaries containing a higher number of total and large follicles, had a higher number of retrieved oocytes, and had a higher pregnancy rate than the patients without OHSS. The patients with severe OHSS also had an increased prevalence of allergy (56% versus 21%) and were more likely to ultimately give birth. CONCLUSION(S) The observed differences may be useful in elucidating the pathophysiology of OHSS and identifying patients who are at increased risk for OHSS.
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Affiliation(s)
- A Enskog
- Department of Anesthesia and Intensive Care, Göteborg University, Sweden
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31
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SPONTANEOUS OVARIAN HYPERSTIMULATION AND PRIMARY HYPOTHYROIDISM WITH A NATURALLY CONCEIVED PREGNANCY. Obstet Gynecol 1999. [DOI: 10.1097/00006250-199905001-00005] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/26/2022]
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Abstract
Hyperreactio luteinalis is a rare benign condition characterized by bilateral ovarian enlargement associated with pregnancies where high concentrations of maternal serum human chorionic gonadotrophins are present. This condition may mimic the ovarian hyperstimulation syndrome. We report a case of a 34 year old woman with a history of chronic renal failure on haemodialysis who presented at 10 weeks' gestational age with hyperreactio luteinalis which was treated conservatively. Because of chronic renal failure, the presentation and course of the disease was different from that which has been previously reported.
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Affiliation(s)
- M al-Ramahi
- Department of Obstetrics and Gynecology, University of Ottawa, Ottawa Hospital, Ontario, Canada
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33
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34
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Karabacak O, Girgin MO, Yucesoy G. Is it polycystic ovary syndrome causing the hyperstimulation again? Am J Obstet Gynecol 1998; 179:838. [PMID: 9758010 DOI: 10.1016/s0002-9378(98)70106-1] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/21/2022]
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35
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Edi-Osagie EC, Hopkins RE. Recurrent idiopathic ovarian hyperstimulation syndrome in pregnancy. BRITISH JOURNAL OF OBSTETRICS AND GYNAECOLOGY 1997; 104:952-4. [PMID: 9255091 DOI: 10.1111/j.1471-0528.1997.tb14359.x] [Citation(s) in RCA: 9] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 02/05/2023]
Affiliation(s)
- E C Edi-Osagie
- Department of Obstetrics and Gynaecology, Bolton General Hospital, Farnworth, UK
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36
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Abstract
Only two cases of severe spontaneous ovarian hyperstimulation syndrome with pregnancy have been reported. We report the third one and the only one that was successfully managed conservatively throughout pregnancy.
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Affiliation(s)
- S K Abu-Louz
- Department of Obstetrics and Gynecology, Faculty of Medicine, Ain Shams University, Cairo, Egypt
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37
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Zalel Y, Orvieto R, Ben-Rafael Z, Homburg R, Fisher O, Insler V. Recurrent spontaneous ovarian hyperstimulation syndrome associated with polycystic ovary syndrome. Gynecol Endocrinol 1995; 9:313-5. [PMID: 8629460 DOI: 10.3109/09513599509160465] [Citation(s) in RCA: 38] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/01/2023] Open
Abstract
Ovarian hyperstimulation syndrome (OHSS) is the most serious potentially life-threatening iatrogenic complication of ovulation induction. Presented here is the first reported case of recurrent severe OHSS which developed spontaneously in a women with polycystic ovary syndrome, diagnosed early in her second pregnancy, and necessitated intensive fluid and colloid therapy.
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Affiliation(s)
- Y Zalel
- Department of Obstetrics and Gynecology, Kaplan Hospital, Rehovot, Israel
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38
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Abstract
Women with hyperandrogenic disorders represent a unique group among those with infertility due to anovulation. Although antiestrogens are effective in restoring ovulation in most women, it remains unclear whether these treatments restore fecundability per ovulatory cycle and the ability to maintain pregnancy in these individuals. Moreover, antiestrogens are ineffective in restoring ovulation in some hyperandrogenic anovulatory women, whose condition poses unique and vexing challenges for the infertility therapist. Gonadotropin treatment in antiestrogen-resistant women often leads to ovarian hyperstimulation syndrome, which has been addressed by modification of dosing schedules (e.g., low-dose administration), pretreatment with gonadotropin-releasing hormone (GnRH) analogs, and elimination of luteinizing hormone from the administered gonadotropins. Surgical reduction in ovarian volume has met with some success, although there may be a risk of inducing surgical adhesions of the adnexa. The second major reproductive adversity facing these patients is their elevated risk of endometrial cancer. Unopposed estrogen exposure probably contributes to this risk, but hyperandrogenicity and hyperinsulinism may act independently or in concert with estrogen to amplify the risk in these women. While the risks and strategies for preventive care in these women need to be better defined, reproductive health specialists are urged to continue using presently accepted measures, including education, to maintain these women's reproductive health.
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Affiliation(s)
- M Gibson
- Robert C. Byrd Health Sciences Center, West Virginia University, Morgantown 26506-9186
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