Document Detail


Tremor/ataxia syndrome and fragile X premutation: diagnostic caveats.
MedLine Citation:
PMID:  17194594     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
The fragile X-associated tremor/ataxia syndrome (FXTAS) is a newly discovered late-onset neurodegenerative disorder caused by a premutation in the FMR1 X-linked gene. We present examples of a discrepancy between obvious brain changes observed on MRI, and minimal clinical neurological manifestations in three older carriers of this premutation. This discrepancy occurred in three of nine carriers ascertained in an unbiased manner. If the systematic follow-up studies of adult carriers confirm this trend, this will have an impact on early diagnosis of neurological involvement and possible prevention. If MRI changes precede clinical manifestation of FXTAS this may explain the low detection rate of fragile X carriers among patients with neurological syndromes associated with tremor/ataxia.
Authors:
D Z Loesch; L Litewka; A Churchyard; E Gould; F Tassone; M Cook
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Publication Detail:
Type:  Case Reports; Journal Article; Research Support, N.I.H., Extramural; Research Support, Non-U.S. Gov't     Date:  2006-12-27
Journal Detail:
Title:  Journal of clinical neuroscience : official journal of the Neurosurgical Society of Australasia     Volume:  14     ISSN:  0967-5868     ISO Abbreviation:  J Clin Neurosci     Publication Date:  2007 Mar 
Date Detail:
Created Date:  2007-01-29     Completed Date:  2007-04-18     Revised Date:  2007-12-03    
Medline Journal Info:
Nlm Unique ID:  9433352     Medline TA:  J Clin Neurosci     Country:  Scotland    
Other Details:
Languages:  eng     Pagination:  245-8     Citation Subset:  IM    
Affiliation:
School of Psychological Science, La Trobe University, Bundoora, Melbourne, Victoria 3086, Australia. d.loesch@latrobe.edu.au
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MeSH Terms
Descriptor/Qualifier:
Aged
Ataxia / genetics,  pathology
Cerebellum / pathology
Fragile X Mental Retardation Protein / genetics*
Fragile X Syndrome / genetics*,  pathology*
Heterozygote
Humans
Magnetic Resonance Imaging*
Male
Middle Aged
Tremor / genetics,  pathology
Grant Support
ID/Acronym/Agency:
HD36071/HD/NICHD NIH HHS
Chemical
Reg. No./Substance:
0/FMR1 protein, human; 139135-51-6/Fragile X Mental Retardation Protein

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


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