Literature DB >> 3775362

Uroporphyrinogen decarboxylase structural mutant (Gly281----Glu) in a case of porphyria.

H de Verneuil, B Grandchamp, C Beaumont, C Picat, Y Nordmann.   

Abstract

Uroporphyrinogen decarboxylase deficiency in man is responsible for familial porphyria cutanea tarda and hepatoerythropoietic porphyria. A recent study of a family with hepatoerythropoietic porphyria showed that the enzyme defect resulted from rapid degradation of the protein in vivo. Cloning and sequencing of a complementary DNA for the mutated gene revealed that the mutation was due to the replacement of a glycine residue by a glutamic acid residue at position 281. This base change leads to a protein that is very rapidly degraded in the presence of cell lysate. Characterization of the mutation will allow comparison of this defect in a homozygous patient with defects in other patients with familial porphyria cutanea tarda.

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Year:  1986        PMID: 3775362     DOI: 10.1126/science.3775362

Source DB:  PubMed          Journal:  Science        ISSN: 0036-8075            Impact factor:   47.728


  23 in total

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2.  Steady-state levels of uroporphyrinogen decarboxylase mRNA in lymphoblastoid cell lines from patients with familial porphyria cutanea tarda and their relatives.

Authors:  J L Hansen; M A Pryor; J B Kennedy; J P Kushner
Journal:  Am J Hum Genet       Date:  1988-06       Impact factor: 11.025

3.  Molecular heterogeneity of acute intermittent porphyria: identification of four additional mutations resulting in the CRIM-negative subtype of the disease.

Authors:  M H Delfau; C Picat; F De Rooij; G Voortman; J C Deybach; Y Nordmann; B Grandchamp
Journal:  Am J Hum Genet       Date:  1991-08       Impact factor: 11.025

4.  Hepatoerythropoietic porphyria misdiagnosed as child abuse: cutaneous, arthritic, and hematologic manifestations in siblings with a novel UROD mutation.

Authors:  Julie L Cantatore-Francis; Jessica Cohen-Pfeffer; Manisha Balwani; Philip Kahn; Herbert M Lazarus; Robert J Desnick; Julie V Schaffer
Journal:  Arch Dermatol       Date:  2010-05

5.  The effects in vivo of mutationally modified uroporphyrinogen decarboxylase in different hem12 mutants of baker's yeast (Saccharomyces cerevisiae).

Authors:  A Kurlandzka; T Zoladek; J Rytka; R Labbe-Bois; D Urban-Grimal
Journal:  Biochem J       Date:  1988-07-01       Impact factor: 3.857

6.  The CpG dinucleotide and human genetic disease.

Authors:  D N Cooper; H Youssoufian
Journal:  Hum Genet       Date:  1988-02       Impact factor: 4.132

Review 7.  Uroporphyrinogen decarboxylase.

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

8.  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

Review 9.  Diagnosis of genetic disease using recombinant DNA. Supplement.

Authors:  D N Cooper; J Schmidtke
Journal:  Hum Genet       Date:  1987-09       Impact factor: 4.132

10.  Structure of the gene for human uroporphyrinogen decarboxylase.

Authors:  M Romana; A Dubart; D Beaupain; C Chabret; M Goossens; P H Romeo
Journal:  Nucleic Acids Res       Date:  1987-09-25       Impact factor: 16.971

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