Document Detail


Aortic hypoplasia and cardiac valvular abnormalities in a boy with fragile X syndrome.
MedLine Citation:
PMID:  2972207     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
An 18-year-old mentally retarded male with the Martin-Bell syndrome was fragile X positive. He died suddenly with viral pneumonia and myocarditis. At autopsy, generalized tubular hypoplasia of the aorta and a mild coarctation were discovered. The base of the mitral and tricuspid valves showed striking aberrations in elastin distribution and structure by light microscopy. Local collagen alterations were also noted. Comparable changes were seen in the skin elastin as well as a severe depletion of acid mucopolysaccharides. These changes suggest a structural disruption underlying the clinical connective tissue problems in some patients with the fragile X syndrome.
Authors:
G Waldstein; R Hagerman
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  American journal of medical genetics     Volume:  30     ISSN:  0148-7299     ISO Abbreviation:  Am. J. Med. Genet.     Publication Date:    1988 May-Jun
Date Detail:
Created Date:  1988-11-08     Completed Date:  1988-11-08     Revised Date:  2004-11-17    
Medline Journal Info:
Nlm Unique ID:  7708900     Medline TA:  Am J Med Genet     Country:  UNITED STATES    
Other Details:
Languages:  eng     Pagination:  83-98     Citation Subset:  IM    
Affiliation:
Department of Pathology, Children's Hospital, Denver, CO 80218.
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MeSH Terms
Descriptor/Qualifier:
Adolescent
Aorta / abnormalities*
Collagen / metabolism
Connective Tissue / metabolism,  pathology
Elastin / metabolism
Fragile X Syndrome / genetics,  metabolism,  pathology*
Glycosaminoglycans / metabolism
Heart Valves / abnormalities*
Humans
Male
Pedigree
Sex Chromosome Aberrations / pathology*
Chemical
Reg. No./Substance:
0/Glycosaminoglycans; 9007-34-5/Collagen; 9007-58-3/Elastin

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


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