Literature DB >> 2385250

Mutations affecting RNA splicing in man are detected more frequently in somatic than in germ cells.

A M Rossi1, J C Thijssen, A D Tates, H Vrieling, A T Natarajan, P H Lohman, A A van Zeeland.   

Abstract

The spectrum of DNA sequence alterations in the hypoxanthine-guanine phosphoribosyltransferase (hprt) gene of HPRTase-deficient T-lymphocytes isolated from the blood of healthy male donors was determined and compared with the spectrum found in patients suffering from genetic diseases (Lesch-Nyhan syndrome or gouty arthritis) associated with a mutation in the same gene. Most of the T-cell mutants still produced hprt mRNA which was converted into cDNA and used for DNA sequence analysis after amplification using the polymerase chain reaction (PCR). In 39% of the 31 analyzed T-cell mutants of normal donors 1 or 2 exons were completely or partially deleted from hprt mRNA, probably because of a mutation in a splice acceptor site. Among patients suffering from the Lesch-Nyhan syndrome or gouty arthritis, the class of splice mutations amounts only to 7%. These data suggest that carriers of splice mutations often do not show the characteristics of HPRTase deficiency associated with these genetic diseases, because correctly spliced hprt mRNA is still produced at a low level.

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Year:  1990        PMID: 2385250     DOI: 10.1016/0165-7992(90)90084-w

Source DB:  PubMed          Journal:  Mutat Res        ISSN: 0027-5107            Impact factor:   2.433


  9 in total

1.  Influence of sex, smoking and age on human hprt mutation frequencies and spectra.

Authors:  J Curry; L Karnaoukhova; G C Guenette; B W Glickman
Journal:  Genetics       Date:  1999-07       Impact factor: 4.562

Review 2.  Too many mutants with multiple mutations.

Authors:  John W Drake
Journal:  Crit Rev Biochem Mol Biol       Date:  2007 Jul-Aug       Impact factor: 8.250

3.  Identification of mutations in the alpha-L-iduronidase gene (IDUA) that cause Hurler and Scheie syndromes.

Authors:  H S Scott; T Litjens; P V Nelson; P R Thompson; D A Brooks; J J Hopwood; C P Morris
Journal:  Am J Hum Genet       Date:  1993-11       Impact factor: 11.025

4.  Selection against blood cells deficient in hypoxanthine phosphoribosyltransferase (HPRT) in Lesch-Nyhan heterozygotes occurs at the level of multipotent stem cells.

Authors:  M Hakoda; Y Hirai; M Akiyama; H Yamanaka; C Terai; N Kamatani; S Kashiwazaki
Journal:  Hum Genet       Date:  1995-12       Impact factor: 4.132

5.  Mutations which alter splicing in the human hypoxanthine-guanine phosphoribosyltransferase gene.

Authors:  H Steingrimsdottir; G Rowley; G Dorado; J Cole; A R Lehmann
Journal:  Nucleic Acids Res       Date:  1992-03-25       Impact factor: 16.971

6.  Characterization of in vivo somatic mutations at the hypoxanthine phosphoribosyltransferase gene of a human control population.

Authors:  K Burkhart-Schultz; C B Thomas; C L Thompson; C L Strout; E Brinson; I M Jones
Journal:  Environ Health Perspect       Date:  1993-04-22       Impact factor: 9.031

7.  Mutations induced in the hypoxanthine phosphoribosyl transferase gene by three urban air pollutants: acetaldehyde, benzo[a]pyrene diolepoxide, and ethylene oxide.

Authors:  B Lambert; B Andersson; T Bastlova; S M Hou; D Hellgren; A Kolman
Journal:  Environ Health Perspect       Date:  1994-10       Impact factor: 9.031

8.  Mechanisms for induction of mutations and chromosome alterations.

Authors:  A T Natarajan
Journal:  Environ Health Perspect       Date:  1993-10       Impact factor: 9.031

9.  Somatic cell gene mutations in humans: biomarkers for genotoxicity.

Authors:  R J Albertini; J A Nicklas; J P O'Neill
Journal:  Environ Health Perspect       Date:  1993-10       Impact factor: 9.031

  9 in total

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