Document Detail


The fragile X syndrome.
MedLine Citation:
PMID:  9678703     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
The fragile X syndrome is characterised by mental retardation, behavioural features, and physical features, such as a long face with large protruding ears and macro-orchidism. In 1991, after identification of the fragile X mental retardation (FMR1) gene, the cytogenetic marker (a fragile site at Xq27.3) became replaced by molecular diagnosis. The fragile X syndrome was one of the first examples of a "novel" class of disorders caused by a trinucleotide repeat expansion. In the normal population, the CGG repeat varies from six to 54 units. Affected subjects have expanded CGG repeats (>200) in the first exon of the FMR1 gene (the full mutation). Phenotypically normal carriers of the fragile X syndrome have a repeat in the 43 to 200 range (the premutation). The cloning of the FMR1 gene led to the characterisation of its protein product FMRP, encouraged further clinical studies, and opened up the possibility of more accurate family studies and fragile X screening programmes.
Authors:
B B de Vries; D J Halley; B A Oostra; M F Niermeijer
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Publication Detail:
Type:  Journal Article; Research Support, Non-U.S. Gov't; Review    
Journal Detail:
Title:  Journal of medical genetics     Volume:  35     ISSN:  0022-2593     ISO Abbreviation:  J. Med. Genet.     Publication Date:  1998 Jul 
Date Detail:
Created Date:  1998-10-01     Completed Date:  1998-10-01     Revised Date:  2011-08-01    
Medline Journal Info:
Nlm Unique ID:  2985087R     Medline TA:  J Med Genet     Country:  ENGLAND    
Other Details:
Languages:  eng     Pagination:  579-89     Citation Subset:  IM    
Affiliation:
Department of Clinical Genetics, University Hospital Dijkzigt and Erasmus University, Rotterdam, The Netherlands.
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MeSH Terms
Descriptor/Qualifier:
Chromosome Fragile Sites
Chromosome Fragility
DNA Mutational Analysis
Female
Fragile X Mental Retardation Protein
Fragile X Syndrome / diagnosis,  epidemiology,  genetics*
Genetic Testing
Humans
Male
Mental Retardation / genetics
Minisatellite Repeats
Nerve Tissue Proteins / genetics
Phenotype
RNA-Binding Proteins*
X Chromosome / genetics
Chemical
Reg. No./Substance:
0/FMR1 protein, human; 0/Nerve Tissue Proteins; 0/RNA-Binding Proteins; 139135-51-6/Fragile X Mental Retardation Protein
Comments/Corrections
Comment In:
J Med Genet. 1999 Feb;36(2):171-2   [PMID:  10051022 ]

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