Document Detail


Advances in understanding the molecular basis of FXTAS.
MedLine Citation:
PMID:  20430935     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Fragile X-associated tremor/ataxia syndrome (FXTAS) is an adult-onset neurodegenerative disorder among carriers of premutation expansions (55-200 CGG repeats) of the fragile X mental retardation 1 (FMR1) gene. The clinical features of FXTAS, as well as other forms of clinical involvement in carriers without FXTAS, are thought to arise from a toxic gain of function of transcriptionally active FMR1 containing expanded CGG repeats. Although the precise mechanisms involved in rCGG toxicity are unknown, here we discuss the latest advances and models that contribute to the understanding of the molecular basis of FXTAS, and the emerging view of FXTAS as the end-stage of a process that begins in early development.
Authors:
Dolores Garcia-Arocena; Paul J Hagerman
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Publication Detail:
Type:  Journal Article; Research Support, N.I.H., Extramural; Review     Date:  2010-04-29
Journal Detail:
Title:  Human molecular genetics     Volume:  19     ISSN:  1460-2083     ISO Abbreviation:  Hum. Mol. Genet.     Publication Date:  2010 Apr 
Date Detail:
Created Date:  2010-05-20     Completed Date:  2010-08-24     Revised Date:  2011-07-28    
Medline Journal Info:
Nlm Unique ID:  9208958     Medline TA:  Hum Mol Genet     Country:  England    
Other Details:
Languages:  eng     Pagination:  R83-9     Citation Subset:  IM    
Affiliation:
Department of Biochemistry and Molecular Medicine, University of California, Davis, CA, USA.
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MeSH Terms
Descriptor/Qualifier:
Fragile X Mental Retardation Protein / genetics
Fragile X Syndrome / genetics*
Humans
RNA / genetics
Tremor / genetics*
Grant Support
ID/Acronym/Agency:
R01 HD040661/HD/NICHD NIH HHS; R01 HD040661-08/HD/NICHD NIH HHS; R01 HD040661-09/HD/NICHD NIH HHS; RL1 AG032119/AG/NIA NIH HHS; UL1DE19583/DE/NIDCR NIH HHS
Chemical
Reg. No./Substance:
0/FMR1 protein, human; 139135-51-6/Fragile X Mental Retardation Protein; 63231-63-0/RNA
Comments/Corrections

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