Document Detail


Folate metabolism in cells from fragile X syndrome patients and carriers.
MedLine Citation:
PMID:  6585141     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
The in vitro folate sensitivity of the fragile site at Xq27 and the claims of a beneficial response of patients given folic acid prompted us to examine the folate metabolism in cells cultured from fragile X syndrome patients and carriers. Using Epstein-Barr virus we established permanent lymphoblastoid lines from 4 fragile X syndrome males and 3 carriers from 7 families. All these lines expressed the fragile site when 0.1 microM 5-fluorodeoxyuridine (FUdR) was added to the cultures 24 hr prior to harvest; thus, the lines seemed suitable for seeking an intrinsic defect. Fragile X syndrome patient and carrier lines and normal control cell lines did not differ in regard to folate requirement for growth, the ability to use homocysteine in place of methionine, the ability to utilize reduced folates as the sole folate source, or methotrexate sensitivity. These results suggest that no intrinsic defect in folate metabolism is present in fragile X syndrome cells.
Authors:
J C Wang; R W Erbe
Publication Detail:
Type:  Comparative Study; Journal Article; Research Support, U.S. Gov't, P.H.S.    
Journal Detail:
Title:  American journal of medical genetics     Volume:  17     ISSN:  0148-7299     ISO Abbreviation:  Am. J. Med. Genet.     Publication Date:  1984 Jan 
Date Detail:
Created Date:  1984-05-11     Completed Date:  1984-05-11     Revised Date:  2007-11-14    
Medline Journal Info:
Nlm Unique ID:  7708900     Medline TA:  Am J Med Genet     Country:  UNITED STATES    
Other Details:
Languages:  eng     Pagination:  303-10     Citation Subset:  IM    
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MeSH Terms
Descriptor/Qualifier:
Adolescent
Adult
Aged
Cell Line
Child
Clone Cells
Culture Media
Drug Resistance
Female
Fibroblasts / metabolism
Folic Acid / metabolism*
Fragile X Syndrome / genetics,  metabolism*
Genetic Markers
Heterozygote*
Homocysteine / pharmacology
Humans
Lymphocytes / metabolism
Male
Methotrexate / pharmacology
Middle Aged
Sex Chromosome Aberrations / metabolism*
Grant Support
ID/Acronym/Agency:
5 T32 GM07748/GM/NIGMS NIH HHS; HD06356/HD/NICHD NIH HHS
Chemical
Reg. No./Substance:
0/Culture Media; 0/Genetic Markers; 454-28-4/Homocysteine; 59-05-2/Methotrexate; 59-30-3/Folic Acid

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


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