Literature DB >> 2220818

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

A I McClatchey1, D L Kaufman, E L Berson, A J Tobin, V E Shih, J F Gusella, V Ramesh.   

Abstract

Gyrate atrophy (GA), a recessive eye disease involving progressive vision loss due to chorioretinal degeneration, is associated with the deficiency of the mitochondrial enzyme ornithine aminotransferase (OAT), with consequent hyperornithinemia. We and others have reported a number of missense mutations at the OAT locus which result in GA. Here we report a GA patient of Danish/Swedish ancestry in whom one OAT allele produces an mRNA that is missing a single 96-bp exon relative to the normal mRNA. Polymerase-chain-reaction amplification and sequencing revealed a 9-bp deletion covering the splice acceptor region of exon 5, resulting in the absence of exon 5 sequences from the mRNA with no disruption to the reading frame. This mutation, which was not present in 15 other independent GA patients, adds to the array of allelic heterogeneity observed in GA and represents the first example of a splicing mutation associated with this disorder.

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Year:  1990        PMID: 2220818      PMCID: PMC1683684     

Source DB:  PubMed          Journal:  Am J Hum Genet        ISSN: 0002-9297            Impact factor:   11.025


  24 in total

1.  An initiator codon mutation in ornithine-delta-aminotransferase causing gyrate atrophy of the choroid and retina.

Authors:  G A Mitchell; L C Brody; J Looney; G Steel; M Suchanek; C Dowling; V Der Kaloustian; M Kaiser-Kupfer; D Valle
Journal:  J Clin Invest       Date:  1988-02       Impact factor: 14.808

2.  Specific synthesis of DNA in vitro via a polymerase-catalyzed chain reaction.

Authors:  K B Mullis; F A Faloona
Journal:  Methods Enzymol       Date:  1987       Impact factor: 1.600

3.  Gyrate atrophy of the choroid and retina: assignment of the ornithine aminotransferase structural gene to human chromosome 10 and mouse chromosome 7.

Authors:  J J O'Donnell; K Vannas-Sulonen; T B Shows; D R Cox
Journal:  Am J Hum Genet       Date:  1988-12       Impact factor: 11.025

4.  GT to AT transition at a splice donor site causes skipping of the preceding exon in phenylketonuria.

Authors:  J Marvit; A G DiLella; K Brayton; F D Ledley; K J Robson; S L Woo
Journal:  Nucleic Acids Res       Date:  1987-07-24       Impact factor: 16.971

5.  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

6.  Human ornithine-delta-aminotransferase. cDNA cloning and analysis of the structural gene.

Authors:  G A Mitchell; J E Looney; L C Brody; G Steel; M Suchanek; J F Engelhardt; H F Willard; D Valle
Journal:  J Biol Chem       Date:  1988-10-05       Impact factor: 5.157

7.  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

8.  Single base mutation in the pro alpha 2(I) collagen gene that causes efficient splicing of RNA from exon 27 to exon 29 and synthesis of a shortened but in-frame pro alpha 2(I) chain.

Authors:  G Tromp; D J Prockop
Journal:  Proc Natl Acad Sci U S A       Date:  1988-07       Impact factor: 11.205

9.  The ornithine aminotransferase (OAT) locus: analysis of RFLPs in gyrate atrophy.

Authors:  V Ramesh; L A Benoit; P Crawford; P T Harvey; T B Shows; V E Shih; J F Gusella
Journal:  Am J Hum Genet       Date:  1988-02       Impact factor: 11.025

10.  Point mutation affecting processing of the ornithine aminotransferase precursor protein in gyrate atrophy.

Authors:  G Inana; C Chambers; Y Hotta; L Inouye; D Filpula; S Pulford; T Shiono
Journal:  J Biol Chem       Date:  1989-10-15       Impact factor: 5.157

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  6 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.  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

3.  Mapping of ornithine aminotransferase gene sequences to mouse chromosomes 7, X, and 3.

Authors:  V Ramesh; S V Cheng; C A Kozak; B J Herron; V E Shih; B A Taylor; J F Gusella
Journal:  Mamm Genome       Date:  1992       Impact factor: 2.957

4.  Differential expression of human nicotinic acetylcholine receptor alpha subunit variants in muscle and non-muscle tissues.

Authors:  S Talib; T B Okarma; J S Lebkowski
Journal:  Nucleic Acids Res       Date:  1993-01-25       Impact factor: 16.971

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

Authors:  Y Mashima; R G Weleber; N G Kennaway; G Inana
Journal:  Hum Genet       Date:  1992-11       Impact factor: 4.132

6.  Pyridoxine-responsive gyrate atrophy of the choroid and retina: clinical and biochemical correlates of the mutation A226V.

Authors:  J Michaud; G N Thompson; L C Brody; G Steel; C Obie; G Fontaine; K Schappert; C G Keith; D Valle; G A Mitchell
Journal:  Am J Hum Genet       Date:  1995-03       Impact factor: 11.025

  6 in total

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