Literature DB >> 8500787

Molecular defect in human erythropoietic protoporphyria with fatal liver failure.

Y Nakahashi1, H Miyazaki, Y Kadota, Y Naitoh, K Inoue, M Yamamoto, N Hayashi, S Taketani.   

Abstract

We investigated the molecular basis of ferrochelatase in a Japanese patient with erythropoietic protoporphyria (EPP), complicated by fatal liver failure, and defined a novel point mutation in the ferrochelatase gene. cDNAs were synthesized using Epstein-Barr-virus-transformed lymphoblastoid cells from the proband. cDNA clones encoding ferrochelatase in the proband were isolated by amplification using the polymerase chain reaction. There were two sizes of ferrochelatase cDNAs; one was normal in size, the other being smaller. Sequence analysis of the abnormally sized cDNA clones revealed that they lacked exon 9 of the ferrochelatase gene. Genomic DNA analysis demonstrated that the proband had the abnormal allele and that it contained a G to A point mutation at the first position of the donor site of intron 9. An identical mutation was detected in the affected family members of the proband by allele-specific oligonucleotide hybridization analysis. EPP is inherited in an autosomal dominant manner in this family.

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Year:  1993        PMID: 8500787     DOI: 10.1007/BF00217346

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


  15 in total

1.  Erythropoietic protoporphyria. A new porphyria syndrome with solar urticaria due to protoporphyrinaemia.

Authors:  I A MAGNUS; A JARRETT; T A PRANKERD; C RIMINGTON
Journal:  Lancet       Date:  1961-08-26       Impact factor: 79.321

2.  Molecular cloning and sequence analysis of cDNA encoding human ferrochelatase.

Authors:  Y Nakahashi; S Taketani; M Okuda; K Inoue; R Tokunaga
Journal:  Biochem Biophys Res Commun       Date:  1990-12-14       Impact factor: 3.575

3.  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

Review 4.  Splicing of messenger RNA precursors.

Authors:  R A Padgett; P J Grabowski; M M Konarska; S Seiler; P A Sharp
Journal:  Annu Rev Biochem       Date:  1986       Impact factor: 23.643

5.  Ferrochelatase activity in human lymphocytes, as quantified by a new high-performance liquid-chromatographic method.

Authors:  E Rossi; K A Costin; P Garcia-Webb
Journal:  Clin Chem       Date:  1988-12       Impact factor: 8.327

6.  Erythropoietic protoporphyria in the house mouse. A recessive inherited ferrochelatase deficiency with anemia, photosensitivity, and liver disease.

Authors:  S Tutois; X Montagutelli; V Da Silva; H Jouault; P Rouyer-Fessard; K Leroy-Viard; J L Guénet; Y Nordmann; Y Beuzard; J C Deybach
Journal:  J Clin Invest       Date:  1991-11       Impact factor: 14.808

7.  A molecular defect in human protoporphyria.

Authors:  D A Brenner; J M Didier; F Frasier; S R Christensen; G A Evans; H A Dailey
Journal:  Am J Hum Genet       Date:  1992-06       Impact factor: 11.025

8.  Human erythropoietic protoporphyria: two point mutations in the ferrochelatase gene.

Authors:  J Lamoril; S Boulechfar; H de Verneuil; B Grandchamp; Y Nordmann; J C Deybach
Journal:  Biochem Biophys Res Commun       Date:  1991-12-16       Impact factor: 3.575

9.  Fatal liver failure in protoporphyria. Synergism between ethanol excess and the genetic defect.

Authors:  H L Bonkovsky; A R Schned
Journal:  Gastroenterology       Date:  1986-01       Impact factor: 22.682

10.  Hereditary hepatic porphyria due to homozygous delta-aminolevulinic acid dehydratase deficiency: studies in lymphocytes and erythrocytes.

Authors:  S Sassa; H Fujita; M Doss; A Hassoun; L Verstraeten; R Mercelis; A Kappas
Journal:  Eur J Clin Invest       Date:  1991-04       Impact factor: 4.686

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  7 in total

1.  Molecular defects in ferrochelatase in patients with protoporphyria requiring liver transplantation.

Authors:  J Bloomer; C Bruzzone; L Zhu; Y Scarlett; S Magness; D Brenner
Journal:  J Clin Invest       Date:  1998-07-01       Impact factor: 14.808

Review 2.  Erythropoietic protoporphyria.

Authors:  T M Cox
Journal:  J Inherit Metab Dis       Date:  1997-06       Impact factor: 4.982

Review 3.  Theodore Woodward Award. Pathogenesis of biochemical abnormalities in protoporphyria.

Authors:  J R Bloomer; M B Poh-Fitzpatrick
Journal:  Trans Am Clin Climatol Assoc       Date:  2000

Review 4.  Porphyrias: animal models and prospects for cellular and gene therapy.

Authors:  H de Verneuil; C Ged; S Boulechfar; F Moreau-Gaudry
Journal:  J Bioenerg Biomembr       Date:  1995-04       Impact factor: 2.945

Review 5.  The ferrochelatase gene structure and molecular defects associated with erythropoietic protoporphyria.

Authors:  S Taketani; H Fujita
Journal:  J Bioenerg Biomembr       Date:  1995-04       Impact factor: 2.945

6.  Human ferrochelatase: a novel mutation in patients with erythropoietic protoporphyria and an isoform caused by alternative splicing.

Authors:  X Schneider-Yin; B W Schäfer; O Tönz; E I Minder
Journal:  Hum Genet       Date:  1995-04       Impact factor: 4.132

7.  Molecular defects in erythropoietic protoporphyria with terminal liver failure.

Authors:  X Schneider-Yin; B W Schäfer; P Möhr; G Burg; E I Minder
Journal:  Hum Genet       Date:  1994-06       Impact factor: 4.132

  7 in total

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