Literature DB >> 1634232

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

H de Verneuil1, F Bourgeois, F de Rooij, P D Siersema, J H Wilson, B Grandchamp, Y Nordmann.   

Abstract

A deficiency in the activity of uroporphyrinogen decarboxylase (UROD), the fifth enzyme of the haem biosynthetic pathway, is found in familial porphyria cutanea tarda (F-PCT) and hepatoerythropoietic porphyria (HEP). A new mutation (R292G) and a deletion have been found in a pedigree with two HEP patients (two sisters). The R292G mutation was not detected in 13 unrelated affected patients with F-PCT, so it appears to be uncommon. The possibility that the arginine 292 may participate at the active site of the enzyme is discussed. A summary of the 7 mutations/deletions found in the UROD gene with their frequency is presented.

Entities:  

Mesh:

Substances:

Year:  1992        PMID: 1634232     DOI: 10.1007/bf00219182

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


  26 in total

1.  Identification of lysine at the active site of human 5-aminolaevulinate dehydratase.

Authors:  P N Gibbs; P M Jordan
Journal:  Biochem J       Date:  1986-06-01       Impact factor: 3.857

2.  Some kinetic properties of human red cell uroporphyrinogen decarboxylase.

Authors:  H de Verneuil; B Grandchamp; Y Nordmann
Journal:  Biochim Biophys Acta       Date:  1980-01-11

3.  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

4.  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

5.  Hepatoerythropoietic porphyria: a new uroporphyrinogen decarboxylase defect or homozygous porphyria cutanea tarda?

Authors:  G H Elder; S G Smith; C Herrero; M Lecha; J M Mascaro; A M Muniesa; D B Czarnecki; J Brenan; V Poulos; R E DE Salamanca
Journal:  Lancet       Date:  1981-04-25       Impact factor: 79.321

6.  Identification of a new mutation responsible for hepatoerythropoietic porphyria.

Authors:  M Romana; B Grandchamp; A Dubart; S Amselem; C Chabret; Y Nordmann; M Goossens; P H Romeo
Journal:  Eur J Clin Invest       Date:  1991-04       Impact factor: 4.686

7.  Purification and characterization of bovine hepatic uroporphyrinogen decarboxylase.

Authors:  J G Straka; J P Kushner
Journal:  Biochemistry       Date:  1983-09-27       Impact factor: 3.162

8.  Uroporphyrinogen decarboxylase deficiency in hepatoerythropoietic porphyria: further evidence for genetic heterogeneity.

Authors:  F Kószó; G H Elder; A Roberts; N Simon
Journal:  Br J Dermatol       Date:  1990-03       Impact factor: 9.302

9.  Hepatoerythropoietic porphyria: clinical, biochemical, and enzymatic studies in a three-generation family lineage.

Authors:  A C Toback; S Sassa; M B Poh-Fitzpatrick; J Schechter; E Zaider; L C Harber; A Kappas
Journal:  N Engl J Med       Date:  1987-03-12       Impact factor: 91.245

10.  Uroporphyrinogen decarboxylase. Purification, properties, and inhibition by polychlorinated biphenyl isomers.

Authors:  S Kawanishi; Y Seki; S Sano
Journal:  J Biol Chem       Date:  1983-04-10       Impact factor: 5.157

View more
  10 in total

1.  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

Review 2.  Molecular genetics of disorders of haem biosynthesis.

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

Review 3.  Erythropoietic and hepatic porphyrias.

Authors:  U Gross; G F Hoffmann; M O Doss
Journal:  J Inherit Metab Dis       Date:  2000-11       Impact factor: 4.982

4.  Mouse uroporphyrinogen decarboxylase: cDNA cloning, expression, and mapping.

Authors:  C Wu; W Xu; C A Kozak; R J Desnick
Journal:  Mamm Genome       Date:  1996-05       Impact factor: 2.957

Review 5.  Uroporphyrinogen decarboxylase.

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

6.  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

7.  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

8.  Isolation and characterization of extragenic mutations affecting the expression of the uroporphyrinogen decarboxylase gene (HEM12) in Sacharomyces cerevisiae.

Authors:  T Zoładek; A Chełstowska; R Labbe-Bois; J Rytka
Journal:  Mol Gen Genet       Date:  1995-05-20

9.  Hepatoerythropoietic porphyria precipitated by viral hepatitis.

Authors:  R J Hift; P N Meissner; G Todd
Journal:  Gut       Date:  1993-11       Impact factor: 23.059

10.  A mouse model of familial porphyria cutanea tarda.

Authors:  J D Phillips; L K Jackson; M Bunting; M R Franklin; K R Thomas; J E Levy; N C Andrews; J P Kushner
Journal:  Proc Natl Acad Sci U S A       Date:  2001-01-02       Impact factor: 11.205

  10 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.