Document Detail


CDKN1C mutations in HELLP/preeclamptic mothers of Beckwith-Wiedemann Syndrome (BWS) patients.
MedLine Citation:
PMID:  19386358     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Preeclampsia is the development of new-onset hypertension with proteinuria after 20 weeks of gestation. HELLP syndrome (haemolysis, elevated liver enzymes, and low platelet count) is a severe form of preeclampsia with high rates of neonatal and maternal morbidity. In recent years, loss of function of cdkn1c (a tight-binding inhibitor of G1 cyclin/cyclin-dependent kinase complexes and a negative regulator of cell proliferation) has been observed in several mouse models of preeclampsia. In this paper, we report on three women with HELLP/preeclampsia who had children with Beckwith Wiedemann syndrome, a complex genetic disorder characterised, among other findings, by overgrowth, omphalocele and macroglossia. All three children displayed mutations in CDKN1C predicted to generate truncated proteins. Two of the mutations were maternally inherited while the third was de novo. This finding suggests a fetal contribution to the maternal disease. To the best of our knowledge this is the first report of CDKN1C mutations in children born to women with preeclampsia/HELLP syndrome, thus suggesting the involvement of an imprinted gene in the pathophysiology of preeclampsia.
Authors:
V Romanelli; A Belinchón; A Campos-Barros; K E Heath; S García-Miñaur; V Martínez-Glez; R Palomo; G Mercado; R Gracia; P Lapunzina
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Publication Detail:
Type:  Case Reports; Journal Article     Date:  2009-04-21
Journal Detail:
Title:  Placenta     Volume:  30     ISSN:  1532-3102     ISO Abbreviation:  Placenta     Publication Date:  2009 Jun 
Date Detail:
Created Date:  2009-05-29     Completed Date:  2009-09-30     Revised Date:  2009-12-02    
Medline Journal Info:
Nlm Unique ID:  8006349     Medline TA:  Placenta     Country:  England    
Other Details:
Languages:  eng     Pagination:  551-4     Citation Subset:  IM    
Affiliation:
INGEMM, Instituto de Genética Médica y Molecular, Hospital Universitario La Paz, Universidad Autónoma de Madrid, Spain.
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MeSH Terms
Descriptor/Qualifier:
Base Sequence
Beckwith-Wiedemann Syndrome / complications,  genetics*
Cyclin-Dependent Kinase Inhibitor p57 / genetics*
DNA Mutational Analysis
Female
Genetic Predisposition to Disease
HELLP Syndrome / etiology,  genetics*
Humans
Infant, Newborn
Mutation / physiology
Pre-Eclampsia / etiology,  genetics*
Pregnancy
Chemical
Reg. No./Substance:
0/CDKN1C protein, human; 0/Cyclin-Dependent Kinase Inhibitor p57

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