Literature DB >> 2243121

Uroporphyrinogen decarboxylase: a splice site mutation causes the deletion of exon 6 in multiple families with porphyria cutanea tarda.

J R Garey1, L M Harrison, K F Franklin, K M Metcalf, E S Radisky, J P Kushner.   

Abstract

Uroporphyrinogen decarboxylase (URO-D) is a cytosolic heme-biosynthetic enzyme that converts uroporphyrinogen to coproporphyrinogen. Defects at the uroporphyrinogen decarboxylase locus cause the human genetic disease familial porphyria cutanea tarda. A splice site mutation has been found in a pedigree with familial porphyria cutanea tarda that causes exon 6 to be deleted from the mRNA. The intron/exon junctions on either side of exon 6 fall between codons, so the resulting protein is shorter than the normal protein, missing only the amino acids coded by exon 6. The shortened protein lacks catalytic activity, is rapidly degraded when exposed to human lymphocyte lysates, and is not detectable by Western blot analysis in lymphocyte lysates derived from affected individuals. The mutation was detected in five of 22 unrelated familial porphyria cutanea tarda pedigrees tested, so it appears to be common. This is the first splice site mutation to be found at the URO-D locus, and the first mutation that causes familial porphyria cutanea tarda to be found in more than one pedigree.

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Year:  1990        PMID: 2243121      PMCID: PMC296884          DOI: 10.1172/JCI114856

Source DB:  PubMed          Journal:  J Clin Invest        ISSN: 0021-9738            Impact factor:   14.808


  34 in total

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Journal:  Nature       Date:  1970-08-15       Impact factor: 49.962

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3.  Scanning from an independently specified branch point defines the 3' splice site of mammalian introns.

Authors:  C W Smith; E B Porro; J G Patton; B Nadal-Ginard
Journal:  Nature       Date:  1989-11-16       Impact factor: 49.962

4.  "Western blotting": electrophoretic transfer of proteins from sodium dodecyl sulfate--polyacrylamide gels to unmodified nitrocellulose and radiographic detection with antibody and radioiodinated protein A.

Authors:  W N Burnette
Journal:  Anal Biochem       Date:  1981-04       Impact factor: 3.365

5.  Purification and properties of uroporphyrinogen decarboxylase from human erythrocytes. A single enzyme catalyzing the four sequential decarboxylations of uroporphyrinogens I and III.

Authors:  H de Verneuil; S Sassa; A Kappas
Journal:  J Biol Chem       Date:  1983-02-25       Impact factor: 5.157

6.  Assays of the heme biosynthetic enzymes. Preface.

Authors:  D F Bishop; R J Desnick
Journal:  Enzyme       Date:  1982

7.  Uroporphyrinogen decarboxylase. A method for measuring enzyme activity.

Authors:  J G Straka; J P Kushner; M A Pryor
Journal:  Enzyme       Date:  1982

8.  An inherited enzymatic defect in porphyria cutanea tarda: decreased uroporphyrinogen decarboxylase activity.

Authors:  J P Kushner; A J Barbuto; G R Lee
Journal:  J Clin Invest       Date:  1976-11       Impact factor: 14.808

9.  DNA sequencing with chain-terminating inhibitors.

Authors:  F Sanger; S Nicklen; A R Coulson
Journal:  Proc Natl Acad Sci U S A       Date:  1977-12       Impact factor: 11.205

10.  Investigations of rat liver uroporphyrinogen decarboxylase. Comparisons of porphyrinogens I and III as substrates and the inhibition by porphyrins.

Authors:  A G Smith; J E Francis
Journal:  Biochem J       Date:  1981-04-01       Impact factor: 3.857

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  15 in total

1.  Spectrin Rouen (beta 220-218), a novel shortened beta-chain variant in a kindred with hereditary elliptocytosis. Characterization of the molecular defect as exon skipping due to a splice site mutation.

Authors:  M Garbarz; W T Tse; P G Gallagher; C Picat; M C Lecomte; F Galibert; D Dhermy; B G Forget
Journal:  J Clin Invest       Date:  1991-07       Impact factor: 14.808

2.  Allogeneic corneoscleral limbus tissue transplantation for treatment of the necrosis in porphyria eye disease.

Authors:  Feng Yan; Yan Lu; Jie Yin; Feng Jiang; Zhen-Ping Huang
Journal:  Int J Ophthalmol       Date:  2014-08-18       Impact factor: 1.779

Review 3.  Molecular genetics of disorders of haem biosynthesis.

Authors:  G H Elder
Journal:  J Clin Pathol       Date:  1993-11       Impact factor: 3.411

Review 4.  [Hepatic porphyrias and alcohol].

Authors:  M O Doss; A Kühnel; U Gross; I Sieg
Journal:  Med Klin (Munich)       Date:  1999-06-15

5.  Characterization of a new mutation (R292G) and a deletion at the human uroporphyrinogen decarboxylase locus in two patients with hepatoerythropoietic porphyria.

Authors:  H de Verneuil; F Bourgeois; F de Rooij; P D Siersema; J H Wilson; B Grandchamp; Y Nordmann
Journal:  Hum Genet       Date:  1992-07       Impact factor: 4.132

Review 6.  Uroporphyrinogen decarboxylase.

Authors:  G H Elder; A G Roberts
Journal:  J Bioenerg Biomembr       Date:  1995-04       Impact factor: 2.945

7.  Familial porphyria cutanea tarda: characterization of seven novel uroporphyrinogen decarboxylase mutations and frequency of common hemochromatosis alleles.

Authors:  M Mendez; L Sorkin; M V Rossetti; K H Astrin; A M del C Batlle; V E Parera; G Aizencang; R J Desnick
Journal:  Am J Hum Genet       Date:  1998-11       Impact factor: 11.025

8.  Uroporphyrinogen decarboxylase: complete human gene sequence and molecular study of three families with hepatoerythropoietic porphyria.

Authors:  M J Moran-Jimenez; C Ged; M Romana; R Enriquez De Salamanca; A Taïeb; G Topi; L D'Alessandro; H de Verneuil
Journal:  Am J Hum Genet       Date:  1996-04       Impact factor: 11.025

9.  Exon skipping due to a mutation in a donor splice site in the WT-1 gene is associated with Wilms' tumor and severe genital malformations.

Authors:  S Schneider; G Wildhardt; R Ludwig; B Royer-Pokora
Journal:  Hum Genet       Date:  1993-07       Impact factor: 4.132

10.  Identification of amino acid changes affecting yeast uroporphyrinogen decarboxylase activity by sequence analysis of hem12 mutant alleles.

Authors:  A Chelstowska; T Zoladek; J Garey; J Kushner; J Rytka; R Labbe-Bois
Journal:  Biochem J       Date:  1992-12-15       Impact factor: 3.857

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