Document Detail


Trinucleotide repeat disorders.
MedLine Citation:
PMID:  17417937     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
The discovery that expansion of unstable repeats can cause a variety of neurological disorders has changed the landscape of disease-oriented research for several forms of mental retardation, Huntington disease, inherited ataxias, and muscular dystrophy. The dynamic nature of these mutations provided an explanation for the variable phenotype expressivity within a family. Beyond diagnosis and genetic counseling, the benefits from studying these disorders have been noted in both neurobiology and cell biology. Examples include insight about the role of translational control in synaptic plasticity, the role of RNA processing in the integrity of muscle and neuronal function, the importance of Fe-S-containing enzymes for cellular energy, and the dramatic effects of altering protein conformations on neuronal function and survival. It is exciting that within a span of 15 years, pathogenesis studies of this class of disorders are beginning to reveal pathways that are potential therapeutic targets.
Authors:
Harry T Orr; Huda Y Zoghbi
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Publication Detail:
Type:  Journal Article; Research Support, N.I.H., Extramural; Research Support, Non-U.S. Gov't; Review    
Journal Detail:
Title:  Annual review of neuroscience     Volume:  30     ISSN:  0147-006X     ISO Abbreviation:  Annu. Rev. Neurosci.     Publication Date:  2007  
Date Detail:
Created Date:  2007-06-29     Completed Date:  2007-09-21     Revised Date:  2007-12-03    
Medline Journal Info:
Nlm Unique ID:  7804039     Medline TA:  Annu Rev Neurosci     Country:  United States    
Other Details:
Languages:  eng     Pagination:  575-621     Citation Subset:  IM    
Affiliation:
Institute of Human Genetics, Department of Laboratory Medicine and Pathology, University of Minnesota, Minneapolis, Minnesota 55455, USA. orrxx002@umn.edu
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MeSH Terms
Descriptor/Qualifier:
Animals
Brain / metabolism*,  physiopathology
Cerebellar Ataxia / genetics,  metabolism,  physiopathology
Fragile X Syndrome / genetics,  metabolism,  physiopathology
Genetic Predisposition to Disease / genetics*
Heredodegenerative Disorders, Nervous System / genetics*,  metabolism,  physiopathology
Humans
Huntington Disease / genetics,  metabolism,  physiopathology
Mutation / genetics
Peptides / genetics,  metabolism
Trinucleotide Repeat Expansion / genetics*
Grant Support
ID/Acronym/Agency:
HD024064/HD/NICHD NIH HHS; NS022920/NS/NINDS NIH HHS; NS0456667/NS/NINDS NIH HHS; NS27699/NS/NINDS NIH HHS
Chemical
Reg. No./Substance:
0/Peptides; 26700-71-0/polyglutamine

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


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