Literature DB >> 1487247

A single-base change at a splice acceptor site in the ornithine aminotransferase gene causes abnormal RNA splicing in gyrate atrophy.

Y Mashima1, R G Weleber, N G Kennaway, G Inana.   

Abstract

Gyrate atrophy (GA) is an autosomal recessive eye disease involving a progressive loss of vision due to chorioretinal degeneration in which the mitochondrial matrix enzyme ornithine aminotransferase (OAT) is defective. Two sisters with GA are described in this study in whom an A-to-G substitution at the 3' splice acceptor site of intron 4 in one allele of the OAT gene results in a truncated OAT mRNA devoid of exon 5 sequence. The mutation in the other allele was identified to be a mis-sense mutation at codon 318 by denaturing gradient gel electrophoresis and direct sequencing of the polymerase chain reaction (PCR)-amplified DNA. Thus, these GA patients are compound heterozygotes with respect to mutations in the OAT gene that result in inactivation of OAT.

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Year:  1992        PMID: 1487247     DOI: 10.1007/bf00220086

Source DB:  PubMed          Journal:  Hum Genet        ISSN: 0340-6717            Impact factor:   4.132


  16 in total

1.  Identification of RNA splicing errors resulting in human ornithine transcarbamylase deficiency.

Authors:  R P Carstens; W A Fenton; L R Rosenberg
Journal:  Am J Hum Genet       Date:  1991-06       Impact factor: 11.025

2.  Gyrate atrophy of the choroid and retina: characterization of mutant ornithine aminotransferase and mechanism of response to vitamin B6.

Authors:  N G Kennaway; L Stankova; M K Wirtz; R G Weleber
Journal:  Am J Hum Genet       Date:  1989-03       Impact factor: 11.025

3.  Inheritance of ornithine aminotransferase gene, mRNA, and enzyme defect in a family with gyrate atrophy of the choroid and retina.

Authors:  Y Hotta; N G Kennaway; R G Weleber; G Inana
Journal:  Am J Hum Genet       Date:  1989-03       Impact factor: 11.025

4.  Splicing defect at the ornithine aminotransferase (OAT) locus in gyrate atrophy.

Authors:  A I McClatchey; D L Kaufman; E L Berson; A J Tobin; V E Shih; J F Gusella; V Ramesh
Journal:  Am J Hum Genet       Date:  1990-11       Impact factor: 11.025

5.  Characterization of a splicing mutation in group A xeroderma pigmentosum.

Authors:  I Satokata; K Tanaka; N Miura; I Miyamoto; Y Satoh; S Kondo; Y Okada
Journal:  Proc Natl Acad Sci U S A       Date:  1990-12       Impact factor: 11.205

6.  Nonsense-codon mutations of the ornithine aminotransferase gene with decreased levels of mutant mRNA in gyrate atrophy.

Authors:  Y Mashima; A Murakami; R G Weleber; N G Kennaway; L Clarke; T Shiono; G Inana
Journal:  Am J Hum Genet       Date:  1992-07       Impact factor: 11.025

7.  Molecular basis of ornithine aminotransferase deficiency in B-6-responsive and -nonresponsive forms of gyrate atrophy.

Authors:  V Ramesh; A I McClatchey; N Ramesh; L A Benoit; E L Berson; V E Shih; J F Gusella
Journal:  Proc Natl Acad Sci U S A       Date:  1988-06       Impact factor: 11.205

8.  Expression defect of ornithine aminotransferase gene in gyrate atrophy.

Authors:  G Inana; Y Hotta; C Zintz; K Takki; R G Weleber; N G Kennaway; K Nakayasu; A Nakajima; T Shiono
Journal:  Invest Ophthalmol Vis Sci       Date:  1988-07       Impact factor: 4.799

9.  At least two mutant alleles of ornithine delta-aminotransferase cause gyrate atrophy of the choroid and retina in Finns.

Authors:  G A Mitchell; L C Brody; I Sipila; J E Looney; C Wong; J F Engelhardt; A S Patel; G Steel; C Obie; M Kaiser-Kupfer
Journal:  Proc Natl Acad Sci U S A       Date:  1989-01       Impact factor: 11.205

10.  Clinical trial of vitamin B6 for gyrate atrophy of the choroid and retina.

Authors:  R G Weleber; N G Kennaway
Journal:  Ophthalmology       Date:  1981-04       Impact factor: 12.079

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  3 in total

Review 1.  Recent advances in the gene map of inherited eye disorders: primary hereditary diseases of the retina, choroid, and vitreous.

Authors:  P J Rosenfeld; V A McKusick; J S Amberger; T P Dryja
Journal:  J Med Genet       Date:  1994-12       Impact factor: 6.318

2.  Rhodopsin mutations in a Scottish retinitis pigmentosa population, including a novel splice site mutation in intron four.

Authors:  C Bell; C A Converse; H M Hammer; A Osborne; N E Haites
Journal:  Br J Ophthalmol       Date:  1994-12       Impact factor: 4.638

3.  Acceptor splice site mutation in the invariant AG of intron 5 of the protein C gene, causing type I protein C deficiency.

Authors:  J M Soria; J Fontcuberta; M Chillón; M Borrell; X Estivill; N Sala
Journal:  Hum Genet       Date:  1993-11       Impact factor: 4.132

  3 in total

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