Document Detail


FXTAS: a progressive neurologic syndrome associated with Fragile X premutation.
MedLine Citation:
PMID:  16131424     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
The FMR1 gene is involved in two different syndromes: Fragile X syndrome and Fragile X-associated tremor/ataxia syndrome (FXTAS). Fragile X syndrome is a childhood disease and is associated with mental retardation as the main clinical characteristic, whereas FXTAS develops in men and women over 50 years of age. FXTAS represents a new form of inclusion disorder with a high prevalence in the general population. The neurologic phenotype of FXTAS includes intention tremor and ataxia. Associated features are dementia, parkinsonism, neuropathy, and autonomic dysfunction. Elevated FMR1 transcripts have been proposed as the molecular basis of the pathogenic mechanism leading to FXTAS. This review discusses recent developments in the clinical phenotype, prevalence and screening, animal models, and molecular mechanisms of RNA-based pathogenesis in FXTAS.
Authors:
Rob Willemsen; Edwin Mientjes; Ben A Oostra
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Publication Detail:
Type:  Journal Article; Research Support, N.I.H., Extramural; Research Support, Non-U.S. Gov't; Research Support, U.S. Gov't, P.H.S.; Review    
Journal Detail:
Title:  Current neurology and neuroscience reports     Volume:  5     ISSN:  1528-4042     ISO Abbreviation:  Curr Neurol Neurosci Rep     Publication Date:  2005 Sep 
Date Detail:
Created Date:  2005-08-31     Completed Date:  2006-01-20     Revised Date:  2007-11-14    
Medline Journal Info:
Nlm Unique ID:  100931790     Medline TA:  Curr Neurol Neurosci Rep     Country:  United States    
Other Details:
Languages:  eng     Pagination:  405-10     Citation Subset:  IM    
Affiliation:
Department of Clinical Genetics, Erasmus MC, PO Box 1738, 3000 DR, Rotterdam, The Netherlands. r.willemsen@erasmusmc.nl
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MeSH Terms
Descriptor/Qualifier:
Cerebellar Ataxia / etiology,  genetics,  metabolism
Chromosomes, Human, X*
Fragile X Syndrome / complications*,  genetics*,  metabolism
Humans
Magnetic Resonance Imaging / methods
Models, Molecular
Mutation*
Tremor / etiology,  genetics
Ubiquitin / metabolism
Grant Support
ID/Acronym/Agency:
R01 HD38038/HD/NICHD NIH HHS
Chemical
Reg. No./Substance:
0/Ubiquitin

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


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