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PMID: 6583702 Published · ppublish English Case Reports Journal Article Research Support, Non-U.S. Gov't Research Support, U.S. Gov't, P.H.S.

beta-Thalassemia in American Blacks: novel mutations in the "TATA" box and an acceptor splice site.

Antonarakis SE, Irkin SH, Cheng TC, Scott AF, Sexton JP, Trusko SP, Charache S, Kazazian HH

Abstract

beta-Thalassemia genes, although often mild in their effects, are common among American Blacks. We have begun a systematic molecular analysis of beta-thalassemia mutations in this group. DNA polymorphisms in the beta-globin gene cluster were examined among 22 beta-thalassemia chromosomes. Six different haplotypes were observed. beta-globin genes of two of these were cloned, and their phenotypes were examined both in heterologous cells upon transient expression and in vivo. The gene found in the most common haplotype (9 of 22 chromosomes) contained a single base substitution (A----G) at position -29 within the highly conserved proximal promoter element (the "TATA" box). This mutant gene directed beta-globin RNA at 25% of normal levels both in heterologous cells and in vivo. It was associated with a mild beta +-thalassemia phenotype. A different gene, isolated from an apparently rare haplotype (1 of 22 chromosomes), had a single base substitution (A----G) within the acceptor splice site of the second intervening sequence. This mutation abolished normal RNA splicing so that the only RNA made from the gene in vitro was an alternatively spliced RNA, which could not encode beta-globin. The mild deficit in beta-globin production attributable to the -29 A----G mutant allele most likely accounts for the frequently mild nature of beta-thalassemia among American Blacks.

MeSH Terms
Adolescent Adult Base Sequence Blacks Female Genes Globins/genetics HeLa Cells/physiology Humans Mutation Nucleic Acid Hybridization Polymorphism, Genetic Thalassemia/genetics Transcription, Genetic United States
Chemicals
Globins
Authors & Affiliations
8 authors, click to expand affiliations / ORCID
Antonarakis S E
Irkin S H
Cheng T C
Scott A F
Sexton J P
Trusko S P
Charache S
Kazazian H H
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Article Info
Journal
Proceedings of the National Academy of Sciences of the United States of America
Abbr.
Proc Natl Acad Sci U S A
ISSN
0027-8424
Published
1984-02-00
Pages
1154-8
Language
English
Region
United States
NLM ID
7505876
PMCID
PMC344784
Subset
IM
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