Document Detail


Animal model for fragile X syndrome.
MedLine Citation:
PMID:  9562525     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
The fragile X syndrome, one of the most common forms of inherited mental retardation, is caused by an expansion of a polymorphic CGG repeat upstream of the coding region in the FMR1 gene. The expansion blocks expression of the FMR1 gene due to methylation of the FMR1 promoter. Functional studies on the FMR1 protein have shown that the protein can bind RNA and might be involved in transport of RNAs from the nucleus to the cytoplasm. A role of FMR1 protein on translation of certain mRNAs has been suggested. An animal model for fragile X syndrome exists and these mice show some behavioural difficulties mimicking the human fragile X syndrome phenotype. This review presents what is known about the protein and what is learned from the animal model for fragile X syndrome.
Authors:
B A Oostra; A T Hoogeveen
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Publication Detail:
Type:  Journal Article; Research Support, Non-U.S. Gov't; Review    
Journal Detail:
Title:  Annals of medicine     Volume:  29     ISSN:  0785-3890     ISO Abbreviation:  Ann. Med.     Publication Date:  1997 Dec 
Date Detail:
Created Date:  1998-06-09     Completed Date:  1998-06-09     Revised Date:  2008-11-21    
Medline Journal Info:
Nlm Unique ID:  8906388     Medline TA:  Ann Med     Country:  ENGLAND    
Other Details:
Languages:  eng     Pagination:  563-7     Citation Subset:  IM    
Affiliation:
MGC Department of Clinical Genetics, Erasmus University, Rotterdam, The Netherlands. oostra@kgen.fgg.eur.nl
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MeSH Terms
Descriptor/Qualifier:
Animals
Carrier Proteins / genetics
Codon / genetics
Cytosine
Disease Models, Animal*
Fragile X Mental Retardation Protein
Fragile X Syndrome / genetics*
Gene Expression Regulation
Guanine
Humans
Mental Retardation / genetics
Methylation
Mice
Mice, Knockout
Mice, Transgenic
Nerve Tissue Proteins / genetics
Phenotype
Polymorphism, Genetic / genetics
Promoter Regions, Genetic / genetics
Protein Biosynthesis
RNA, Messenger / genetics
RNA-Binding Proteins / genetics
Repetitive Sequences, Nucleic Acid / genetics
Chemical
Reg. No./Substance:
0/Carrier Proteins; 0/Codon; 0/FMR1 protein, human; 0/Fmr1 protein, mouse; 0/Nerve Tissue Proteins; 0/RNA, Messenger; 0/RNA-Binding Proteins; 139135-51-6/Fragile X Mental Retardation Protein; 71-30-7/Cytosine; 73-40-5/Guanine

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


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