Molecular epidemiology of erythropoietic protoporphyria in the U.K

Br J Dermatol. 2010 Mar;162(3):642-6. doi: 10.1111/j.1365-2133.2010.09631.x. Epub 2010 Jan 22.

Abstract

Background: Erythropoietic protoporphyria (EPP) is a cutaneous porphyria caused by mutations in the ferrochelatase (FECH) or, less frequently, the delta-aminolaevulinate synthase 2 (ALAS2) gene. Predictive genetic counselling requires accurate molecular diagnosis and knowledge of patterns of inheritance.

Objectives: To investigate the molecular epidemiology of EPP in the U.K.

Methods: DNA samples from 191 unrelated patients resident in the U.K. were analysed for mutations in the FECH and ALAS2 genes and for the FECH IVS3-48 dimorphism.

Results: Mutations were identified in 179 (94%) patients. Most (169; 94%) had a FECH mutation on one allele and were classified as having pseudodominant EPP (psdEPP); seven (4%) patients had FECH mutations on both alleles (autosomal recessive EPP) and three (2%) patients had ALAS2 mutations (X-linked dominant protoporphyria). The FECH IVS3-48C allele was strongly associated with psdEPP and with the absence of mutations at the FECH or ALAS2 loci. Fifty-six FECH mutations were identified, 19 being previously unreported. Missense mutations were predominant in autosomal recessive EPP (82%) but not in psdEPP (32%). One mutation (c.314 + 2T>G) was present in 41 (24%) of EPP families, most of whom appeared to be descended from a common ancestor resident in the north of England.

Conclusions: These data define the prevalence and molecular epidemiology of each type of EPP in the U.K.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • 5-Aminolevulinate Synthetase / genetics*
  • 5-Aminolevulinate Synthetase / metabolism
  • Cross-Sectional Studies
  • DNA Mutational Analysis / methods
  • Ferrochelatase / genetics*
  • Ferrochelatase / metabolism
  • Genetic Predisposition to Disease / genetics
  • Humans
  • Mutation / genetics*
  • Pedigree
  • Prevalence
  • Protoporphyria, Erythropoietic / epidemiology
  • Protoporphyria, Erythropoietic / genetics*
  • Sequence Analysis, DNA
  • Statistics as Topic
  • United Kingdom / epidemiology

Substances

  • 5-Aminolevulinate Synthetase
  • Ferrochelatase