Literature DB >> 492326

Suppression of the nonsense mutation in homozygous beta 0 thalassaemia.

J C Chang, G F Temple, R F Trecartin, Y W Kan.   

Abstract

The common form of beta thalassaemia associated with elevated haemoglobin A2 levels can be broadly classified as beta + or beta 0 type according to the presence or absence of beta-globin chain synthesis in the homozygous state. The molecular pathology of each type is heterogeneous. Apart from a subgroup of Indo-Pakistani patients, the beta-globin structural gene is intact in the majority of patients with beta 0 thalassaemia. The amount of beta-globin mRNA present in the reticulocytes of these patients varies: in some it is absent or barely detectable; in others, a substantial amount is present, but it is nonfunctional. We recently demonstrated that the molecular lesion in a Chinese patient with nonfunctional beta-globin mRNA was due to the mutation of the normal lysine codon AAG at amino acid 17 to the amber terminator codon UAG, which prematurely terminates the beta-globin chain. In the present study we demonstrate the first example of a nonsense mutation in humans which can be suppressed in vitro by the suppressor tRNA, as has been found in other eukaryotic cells and viruses.

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Year:  1979        PMID: 492326     DOI: 10.1038/281602a0

Source DB:  PubMed          Journal:  Nature        ISSN: 0028-0836            Impact factor:   49.962


  16 in total

1.  New amber mutation in a beta-thalassemic gene with nonmeasurable levels of mutant messenger RNA in vivo.

Authors:  G F Atweh; H E Brickner; X X Zhu; H H Kazazian; B G Forget
Journal:  J Clin Invest       Date:  1988-08       Impact factor: 14.808

2.  beta-Thalassemia present in cis to a new beta-chain structural variant, Hb Vicksburg [beta 75 (E19)Leu leads to 0].

Authors:  J G Adams; M H Steinberg; M V Newman; W T Morrison; E J Benz; R Iyer
Journal:  Proc Natl Acad Sci U S A       Date:  1981-01       Impact factor: 11.205

3.  Mutations in C2ORF71 cause autosomal-recessive retinitis pigmentosa.

Authors:  Rob W J Collin; Christine Safieh; Karin W Littink; Stavit A Shalev; Hanna J Garzozi; Leah Rizel; Anan H Abbasi; Frans P M Cremers; Anneke I den Hollander; B Jeroen Klevering; Tamar Ben-Yosef
Journal:  Am J Hum Genet       Date:  2010-04-15       Impact factor: 11.025

Review 4.  mRNA stability in mammalian cells.

Authors:  J Ross
Journal:  Microbiol Rev       Date:  1995-09

5.  Characterization of am404, an amber mutation in the simian virus 40 T antigen gene.

Authors:  D R Rawlins; P Collis; N Muzyczka
Journal:  J Virol       Date:  1983-07       Impact factor: 5.103

Review 6.  The thalassemias: molecular mechanisms of human genetic disease.

Authors:  R A Spritz; B G Forget
Journal:  Am J Hum Genet       Date:  1983-05       Impact factor: 11.025

7.  In vivo aminoacylation of human and Xenopus suppressor tRNAs constructed by site-specific mutagenesis.

Authors:  Y S Ho; Y W Kan
Journal:  Proc Natl Acad Sci U S A       Date:  1987-04       Impact factor: 11.205

Review 8.  Nonsense-mediated mRNA decay in humans at a glance.

Authors:  Tatsuaki Kurosaki; Lynne E Maquat
Journal:  J Cell Sci       Date:  2016-01-19       Impact factor: 5.285

Review 9.  The molecular basis of disorders of human hemoglobin synthesis.

Authors:  F Ramirez; J G Mears; A Bank
Journal:  Mol Cell Biochem       Date:  1980-08-16       Impact factor: 3.396

10.  beta zero thalassemia in Sardinia is caused by a nonsense mutation.

Authors:  R F Trecartin; S A Liebhaber; J C Chang; K Y Lee; Y W Kan; M Furbetta; A Angius; A Cao
Journal:  J Clin Invest       Date:  1981-10       Impact factor: 14.808

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