Home LiteratureArticle Details
PMID: 6308558 Published · ppublish English Journal Article Research Support, Non-U.S. Gov't Research Support, U.S. Gov't, P.H.S.

ATA box transcription mutation in beta-thalassemia.

Nucleic acids research ·Vol. 11 ·No. 14 ·1983-07-25 ·Pages 4727-34

Orkin SH, Sexton JP, Cheng TC, Goff SC, Giardina PJ, Lee JI, Kazazian HH

Abstract

DNA sequence analysis of a cloned beta-globin gene from a Chinese patient with beta-thalassemia revealed a single nucleotide substitution (A leads to G) within the ATA box homology and 28 base pairs upstream from the cap site. The patient was homozygous for this particular allele based on restriction mapping at nine different polymorphic sites in the beta-globin gene cluster. Upon transient expression in HeLa cells this gene directed the production of 3-5-fold less beta-globin mRNA than the normal beta-gene. In RNA isolated from the patient's erythroid cells beta-RNA was 10-fold less abundant relative to alpha-RNA than normal, indicating close approximation of the heterologous cell expression results and the in vivo state. These findings support the validity of such transient expression assays for analysis of phenotypes associated with naturally occurring mutant genes and establish the functional significance of nucleotide substitutions at position -28 for human beta-globin gene transcription.

MeSH Terms
Base Composition Base Sequence Cloning, Molecular DNA Restriction Enzymes Erythrocytes/metabolism Genes Globins/genetics Humans Mutation Thalassemia/genetics Transcription, Genetic
Chemicals
Globins DNA Restriction Enzymes
Authors & Affiliations
7 authors, click to expand affiliations / ORCID
Orkin S H
Sexton J P
Cheng T C
Goff S C
Giardina P J
Lee J I
Kazazian H H
References (23)
23 references, click to expand
  1. Mapping of RNA by a modification of the Berk-Sharp procedure: the 5' termini of 15 S beta-globin mRNA precursor and mature 10 s beta-globin mRNA have identical map coordinates.
    Nucleic Acids Res. 1979 Nov 10;7(5):1175-93 PMID: 390497
  2. Sequencing end-labeled DNA with base-specific chemical cleavages.
    Methods Enzymol. 1980;65(1):499-560 PMID: 6246368
  3. Transcription maps of polyoma virus-specific RNA: analysis by two-dimensional nuclease S1 gel mapping.
    Methods Enzymol. 1980;65(1):718-49 PMID: 6154876
  4. The structure and evolution of the human beta-globin gene family.
    Cell. 1980 Oct;21(3):653-68 PMID: 6985477
  5. Specific in vitro transcription of conalbumin gene is drastically decreased by single-point mutation in T-A-T-A box homology sequence.
    Proc Natl Acad Sci U S A. 1980 Dec;77(12):7024-8 PMID: 6938951
  6. A T to A base substitution and small deletions in the conalbumin TATA box drastically decrease specific in vitro transcription.
    Nucleic Acids Res. 1981 Apr 24;9(8):1813-24 PMID: 6264394
  7. Organization and expression of eucaryotic split genes coding for proteins.
    Annu Rev Biochem. 1981;50:349-83 PMID: 6791577
  8. Nonrandom association of polymorphic restriction sites in the beta-globin gene cluster.
    Proc Natl Acad Sci U S A. 1982 Jan;79(1):137-41 PMID: 6275383
  9. Beta + thalassemia: aberrant splicing results from a single point mutation in an intron.
    Cell. 1981 Dec;27(2 Pt 1):289-98 PMID: 6895866
  10. Expression of a beta-globin gene is enhanced by remote SV40 DNA sequences.
    Cell. 1981 Dec;27(2 Pt 1):299-308 PMID: 6277502
  11. Linkage of beta-thalassaemia mutations and beta-globin gene polymorphisms with DNA polymorphisms in human beta-globin gene cluster.
    Nature. 1982 Apr 15;296(5858):627-31 PMID: 6280057
  12. Abnormally spliced messenger RNA in erythroid cells from patients with beta+ thalassemia and monkey cells expressing a cloned beta+-thalassemic gene.
    Cell. 1982 Mar;28(3):585-93 PMID: 6280877
  13. beta-Thalassemia in a Kurdish Jew. Single base changes in the T-A-T-A box.
    J Biol Chem. 1982 Jun 10;257(11):5994-6 PMID: 7076659
  14. Transcriptional control signals of a eukaryotic protein-coding gene.
    Science. 1982 Jul 23;217(4557):316-24 PMID: 6283634
  15. Point mutagenesis of the ovalbumin gene promoter sequence and its effect on in vitro transcription.
    J Biol Chem. 1982 Sep 25;257(18):11070-7 PMID: 6286680
  16. Evidence for multiple origins of the beta E-globin gene in Southeast Asia.
    Proc Natl Acad Sci U S A. 1982 Nov;79(21):6608-11 PMID: 6292908
  17. Abnormal RNA processing due to the exon mutation of beta E-globin gene.
    Nature. 1982 Dec 23;300(5894):768-9 PMID: 7177196
  18. Abnormal RNA splicing causes one form of alpha thalassemia.
    Cell. 1982 Jul;29(3):895-902 PMID: 7151175
  19. A single-base change at a splice site in a beta 0-thalassemic gene causes abnormal RNA splicing.
    Cell. 1982 Jul;29(3):903-11 PMID: 7151176
  20. Formation of stable preinitiation complexes between eukaryotic class B transcription factors and promoter sequences.
    Nature. 1983 Feb 24;301(5902):680-6 PMID: 6828151
  21. Abnormal splice in a mutant human beta-globin gene not at the site of a mutation.
    Proc Natl Acad Sci U S A. 1983 Mar;80(5):1184-8 PMID: 6298782
  22. Three regions upstream from the cap site are required for efficient and accurate transcription of the rabbit beta-globin gene in mouse 3T6 cells.
    Cell. 1983 Mar;32(3):695-706 PMID: 6299573
  23. Specific transcription and RNA splicing defects in five cloned beta-thalassaemia genes.
    Nature. 1983 Apr 14;302(5909):591-6 PMID: 6188062
Article Info
Journal
Nucleic acids research
Abbr.
Nucleic Acids Res
ISSN
0305-1048
Published
1983-07-25
Pages
4727-34
Language
English
Region
England
NLM ID
0411011
PMCID
PMC326082
Subset
IM
Analysis Services
Analysis Services

Contact

No. 2 Wenbo Road, Zhangqiu District, Jinan, Shandong

Qilu Normal University · Genelibs Bioinformatics Lab

750 Shunhua Rd, Jinan

2F, Bldg F, University Science Park

Tel: 0531-88819269

WeChat Official Account

Follow our WeChat subscription account for real-time updates and the latest in medical and biological research.


Business Email

E-mail: [email protected]