Literature DB >> 3753711

Molecular analysis of uroporphyrinogen decarboxylase deficiency in a family with two cases of hepatoerythropoietic porphyria.

H de Verneuil, B Grandchamp, P H Romeo, N Raich, C Beaumont, M Goossens, H Nicolas, Y Nordmann.   

Abstract

In order to determine the molecular basis of uroporphyrinogen (URO) decarboxylase deficiency responsible for hepatoerythropoietic porphyria (HEP) and familial porphyria cutanea tarda, we used a human URO decarboxylase cDNA to analyze the organization and expression of the URO decarboxylase gene in lymphoblastoid cells from normal individuals and from two patients with HEP. We could detect neither deletions nor rearrangements in the URO decarboxylase gene. Synthesis, processing, and cell-free translation of the specific transcripts appeared to be normal. The half-life of the abnormal protein was 12 times shorter than that of the normal enzyme. The results indicate that the enzyme defect is due to a rapid degradation of the protein in vivo. This study is the first to provide information regarding the molecular mechanism responsible for the URO decarboxylase deficiency in HEP.

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Year:  1986        PMID: 3753711      PMCID: PMC423363          DOI: 10.1172/JCI112321

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


  24 in total

1.  Cleavage of structural proteins during the assembly of the head of bacteriophage T4.

Authors:  U K Laemmli
Journal:  Nature       Date:  1970-08-15       Impact factor: 49.962

2.  A case of biochemically unclassifiable hepatic porphyria.

Authors:  J P Aguadé; A Castells; A Indacochea; J Rodés
Journal:  Br J Dermatol       Date:  1969-04       Impact factor: 9.302

3.  DNA in medicine. Human genetics.

Authors:  R L White
Journal:  Lancet       Date:  1984-12-01       Impact factor: 79.321

4.  An efficient mRNA-dependent translation system from reticulocyte lysates.

Authors:  H R Pelham; R J Jackson
Journal:  Eur J Biochem       Date:  1976-08-01

5.  Familial and sporadic porphyria cutanea: two different diseases.

Authors:  H de Verneuil; G Aitken; Y Nordmann
Journal:  Hum Genet       Date:  1978-10-31       Impact factor: 4.132

6.  Enzymatic and immunological studies of uroporphyrinogen decarboxylase in familial porphyria cutanea tarda and hepatoerythropoietic porphyria.

Authors:  H de Verneuil; C Beaumont; J C Deybach; Y Nordmann; Z Sfar; R Kastally
Journal:  Am J Hum Genet       Date:  1984-05       Impact factor: 11.025

7.  Isolation and identification of a cDNA clone coding for rat uroporphyrinogen decarboxylase.

Authors:  P H Romeo; A Dubart; B Grandchamp; H de Verneuil; J Rosa; Y Nordmann; M Goossens
Journal:  Proc Natl Acad Sci U S A       Date:  1984-06       Impact factor: 11.205

8.  Decreased activity of hepatic uroporphyrinogen decarboxylase in sporadic porphyria cutanea tarda.

Authors:  G H Elder; G B Lee; J A Tovey
Journal:  N Engl J Med       Date:  1978-08-10       Impact factor: 91.245

9.  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

10.  Genetic expression in partial adenosine deaminase deficiency. mRNA levels and protein turnover for the enzyme variants in human B-lymphoblast cell lines.

Authors:  P E Daddona; B L Davidson; J L Perignon; W N Kelley
Journal:  J Biol Chem       Date:  1985-03-25       Impact factor: 5.157

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  5 in total

1.  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

2.  The molecular defect of ferrochelatase in a patient with erythropoietic protoporphyria.

Authors:  Y Nakahashi; H Fujita; S Taketani; N Ishida; A Kappas; S Sassa
Journal:  Proc Natl Acad Sci U S A       Date:  1992-01-01       Impact factor: 11.205

3.  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 4.  Diagnosis of genetic disease using recombinant DNA. Supplement.

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

5.  Immunochemical study of uroporphyrinogen decarboxylase in a patient with mild hepatoerythropoietic porphyria.

Authors:  H Fujita; S Sassa; A C Toback; A Kappas
Journal:  J Clin Invest       Date:  1987-05       Impact factor: 14.808

  5 in total

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