Molecular defect in human erythropoietic protoporphyria with fatal liver failure

Hum Genet. 1993 May;91(4):303-6. doi: 10.1007/BF00217346.

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.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Base Sequence
  • Cell Line, Transformed
  • DNA Mutational Analysis
  • Ferrochelatase / genetics*
  • Gene Deletion
  • Genes, Dominant
  • Humans
  • Liver Failure, Acute / genetics*
  • Lymphocytes
  • Male
  • Molecular Sequence Data
  • Oligonucleotide Probes
  • Pedigree
  • Point Mutation
  • Polymerase Chain Reaction
  • Porphyria, Hepatoerythropoietic / enzymology
  • Porphyria, Hepatoerythropoietic / genetics*

Substances

  • Oligonucleotide Probes
  • Ferrochelatase