Document Detail


Two different entities of spontaneous ovarian hyperstimulation in a woman with FSH receptor mutation.
MedLine Citation:
PMID:  20378412     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Spontaneous ovarian hyperstimulation syndrome (OHSS) is an extremely rare event. Normally OHSS is seen in the context of IVF. In 2003 a mutation of the FSH receptor (FSHR D567N) was identified for the first time as a cause of spontaneous OHSS. In most FSHR mutations, a hypersensitivity to human chorionic gonadotrophin (HCG) or thyroid-stimulating hormone (TSH) is described. This clinical case presents for the first time two occurrences of spontaneous OHSS in a single woman with a FSHR mutation and two different entities. Pathophysiology of both pregnancies was completely different. During the first pregnancy, elevated HCG and androgen concentrations led to spontaneous OHSS and finally to miscarriage. The second pregnancy with spontaneous OHSS was dominated by a latent hypothyroidism and normal HCG concentrations and ended in a delivery of a healthy female newborn. Due to the unusual courses of the pregnancies, the study looked for a mutation in the FSHR and surprisingly identified the same mutation previously described. This report confirms for the first time the in-vitro findings in a single clinical case that TSH as well as HCG leads to spontaneous OHSS in patients with FSHR D567N mutation. Hypothyroidism has to be treated or ruled out.
Authors:
M Dieterich; M Bolz; T Reimer; S Costagliola; B Gerber
Publication Detail:
Type:  Case Reports; Journal Article     Date:  2010-02-26
Journal Detail:
Title:  Reproductive biomedicine online     Volume:  20     ISSN:  1472-6491     ISO Abbreviation:  Reprod. Biomed. Online     Publication Date:  2010 Jun 
Date Detail:
Created Date:  2010-06-01     Completed Date:  2010-12-23     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  101122473     Medline TA:  Reprod Biomed Online     Country:  Netherlands    
Other Details:
Languages:  eng     Pagination:  751-8     Citation Subset:  IM    
Copyright Information:
2010 Reproductive Healthcare Ltd. Published by Elsevier Ltd. All rights reserved.
Affiliation:
Department of Obstetrics and Gynecology, University of Rostock, Suedring 81, 18055 Rostock, Germany. max.dieterich@uni-rostock.de
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MeSH Terms
Descriptor/Qualifier:
Adult
Female
Humans
Mutation*
Ovarian Hyperstimulation Syndrome / classification*,  genetics
Pregnancy
Receptors, FSH / genetics*
Chemical
Reg. No./Substance:
0/Receptors, FSH

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