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PMID: 8132745 Published · ppublish English Journal Article Research Support, Non-U.S. Gov't Research Support, U.S. Gov't, P.H.S.

Supravalvular aortic stenosis associated with a deletion disrupting the elastin gene.

The Journal of clinical investigation ·Vol. 93 ·No. 3 ·1994-03-00 ·Pages 1071-7

Ewart AK, Jin W, Atkinson D, Morris CA, Keating MT

Abstract

Supravalvular aortic stenosis (SVAS) is an inherited vascular disease that can cause heart failure and death. SVAS can be inherited as an autosomal dominant trait or as part of a developmental disorder, Williams syndrome (WS). In recent studies we presented evidence suggesting that a translocation disrupting the elastin gene caused SVAS in one family while deletions involving the entire elastin locus caused WS. In this study, pulsed-field, PCR, and Southern analyses showed that a 100-kb deletion of the 3' end of the elastin gene cosegregated with the disease in another SVAS family. DNA sequence analysis localized the breakpoint between elastin exons 27 and 28, the same region disrupted by the SVAS-associated translocation. These data indicate that mutations in the elastin gene cause SVAS and suggest that elastin exons 28-36 may encode critical domains for vascular development.

MeSH Terms
Aortic Stenosis, Subvalvular/genetics Base Sequence Elastin/genetics Electrophoresis, Gel, Pulsed-Field Female Gene Deletion Humans Male Molecular Sequence Data Mutation
Chemicals
Elastin
Authors & Affiliations
5 authors, click to expand affiliations / ORCID
Ewart A K
Department of Human Genetics, University of Utah, Salt Lake City 84112.
Jin W
Atkinson D
Morris C A
Keating M T
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Article Info
Journal
The Journal of clinical investigation
Abbr.
J Clin Invest
ISSN
0021-9738
Published
1994-03-00
Pages
1071-7
Language
English
Region
United States
NLM ID
7802877
PMCID
PMC294040
Subset
IM
Grants
NCRR NIH HHS · MO1 RR-00064 · United States
NHLBI NIH HHS · R01 HL-4807 · United States
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GENBANK
L23859
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