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PMID: 17417937 Published · ppublish English Journal Article Research Support, N.I.H., Extramural Research Support, Non-U.S. Gov't Review

Trinucleotide repeat disorders.

Annual review of neuroscience ·Vol. 30 ·2007-00-00 ·Pages 575-621

Orr HT, Zoghbi HY

Abstract

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.

MeSH Terms
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
Chemicals
Peptides polyglutamine
Authors & Affiliations
2 authors, click to expand affiliations / ORCID
Orr Harry T
Institute of Human Genetics, Department of Laboratory Medicine and Pathology, University of Minnesota, Minneapolis, Minnesota 55455, USA. orrxx002@umn.edu
Zoghbi Huda Y
Article Info
Journal
Annual review of neuroscience
Abbr.
Annu Rev Neurosci
ISSN
0147-006X
Published
2007-00-00
Pages
575-621
Language
English
Region
United States
NLM ID
7804039
Subset
IM
Grants
NICHD NIH HHS · HD024064 · United States
NINDS NIH HHS · NS022920 · United States
NINDS NIH HHS · NS0456667 · United States
NINDS NIH HHS · NS27699 · United States
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