Document Detail


STXBP1 mutations cause not only Ohtahara syndrome but also West syndrome--result of Japanese cohort study.
MedLine Citation:
PMID:  21204804     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
We performed STXBP1 mutation analyses in 86 patients with various types of epilepsies, including 10 patients with OS, 43 with West syndrome, 2 with Lennox-Gastaut syndrome, 12 with symptomatic generalized epilepsy, 14 with symptomatic partial epilepsy, and 5 with other undetermined types of epilepsy. In all patients, the etiology was unknown, but ARX and CDKL5 mutations were negative in all cases. All coding exons of STXBP1 were analyzed by direct-sequencing. Two de novo nucleotide alterations of STXBP1 were identified in two patients with Ohtahara and West syndrome, respectively. No de novo or deleterious mutations in STXBP1 were found in the remaining 84 patients with various types of symptomatic epilepsies. This is the first case report showing that STXBP1 mutations caused West syndrome from the onset of epilepsy. STXBP1 analysis should be considered as an etiology of symptomatic West syndrome without explainable cause.
Authors:
Motoko Otsuka; Hirokazu Oguni; Jao-Shwann Liang; Hiroko Ikeda; Katsumi Imai; Kyoko Hirasawa; Kaoru Imai; Emiko Tachikawa; Keiko Shimojima; Makiko Osawa; Toshiyuki Yamamoto
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Publication Detail:
Type:  Journal Article     Date:  2010-11-03
Journal Detail:
Title:  Epilepsia     Volume:  51     ISSN:  1528-1167     ISO Abbreviation:  Epilepsia     Publication Date:  2010 Dec 
Date Detail:
Created Date:  2011-01-05     Completed Date:  2011-02-14     Revised Date:  2011-03-21    
Medline Journal Info:
Nlm Unique ID:  2983306R     Medline TA:  Epilepsia     Country:  United States    
Other Details:
Languages:  eng     Pagination:  2449-52     Citation Subset:  IM    
Copyright Information:
Wiley Periodicals, Inc. © 2010 International League Against Epilepsy.
Affiliation:
Department of Pediatrics, Tokyo Women's Medical University, Shinjuku-ku, Tokyo, Japan.
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MeSH Terms
Descriptor/Qualifier:
Asian Continental Ancestry Group / genetics
Child
Cohort Studies
Female
Haploinsufficiency / genetics
Humans
Infant
Male
Mental Retardation / diagnosis,  genetics
Munc18 Proteins / genetics*
Mutation / genetics*
Mutation, Missense / genetics
Spasms, Infantile / diagnosis,  genetics*
Chemical
Reg. No./Substance:
0/Munc18 Proteins; 0/STXBP1 protein, human

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


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