Document Detail


Trinucleotide repeat amplification and hypermethylation of a CpG island in FRAXE mental retardation.
MedLine Citation:
PMID:  8334699     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
We have cloned the fragile site FRAXE and demonstrate that individuals with this fragile site possess amplifications of a GCC repeat adjacent to a CpG island in Xq28 of the human X chromosome. Normal individuals have 6-25 copies of the GCC repeat, whereas mentally retarded, FRAXE-positive individuals have > 200 copies and also have methylation at the CpG island. This situation is similar to that seen at the FRAXA locus and is another example in which a trinucleotide repeat expansion is associated with a human genetic disorder. In contrast with the fragile X syndrome, the GCC repeat can expand or contract and is equally unstable when passed through the male or female line. These results also have implications for the understanding of chromosome fragility.
Authors:
S J Knight; A V Flannery; M C Hirst; L Campbell; Z Christodoulou; S R Phelps; J Pointon; H R Middleton-Price; A Barnicoat; M E Pembrey
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Publication Detail:
Type:  Comparative Study; Journal Article; Research Support, Non-U.S. Gov't    
Journal Detail:
Title:  Cell     Volume:  74     ISSN:  0092-8674     ISO Abbreviation:  Cell     Publication Date:  1993 Jul 
Date Detail:
Created Date:  1993-08-20     Completed Date:  1993-08-20     Revised Date:  2009-09-29    
Medline Journal Info:
Nlm Unique ID:  0413066     Medline TA:  Cell     Country:  UNITED STATES    
Other Details:
Languages:  eng     Pagination:  127-34     Citation Subset:  IM    
Affiliation:
Institute of Molecular Medicine, John Radcliffe Hospital, Headington, Oxford, England.
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MeSH Terms
Descriptor/Qualifier:
Base Sequence
Fragile X Syndrome / genetics*
Gene Amplification*
Humans
Mental Retardation / genetics*
Methylation
Molecular Sequence Data
Pedigree
Repetitive Sequences, Nucleic Acid
X Chromosome*
Grant Support
ID/Acronym/Agency:
//Wellcome Trust

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


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