[Epidermolysis bullosa simplex: genotype-phenotype correlation in Danish patients]

Ugeskr Laeger. 2000 Mar 27;162(13):1873-6.
[Article in Danish]

Abstract

Epidermolysis bullosa simplex (EBS) is a group of autosomal dominant inherited skin disorders caused by mutations in the keratin genes K5 or K14. We examined five Danish families with EBS-Weber-Cockayne (WC) or EBS-Koebner (K) and two sporadic cases of EBS-Dowling-Meara (DM) in order to investigate the mutational spectrum and evaluate the genotype-phenotype correlation in Danish patients. Three new K14 mutations, one new and one previously described K5 mutation were identified by DNA sequence analysis. The positions of the EBS-DM mutations were consistent with previous studies, whereas the EBS-WC and EBS-K mutations were found in regions of the keratin genes not typically associated with this type of EBS mutations. In conclusion, we found a strict genotype-phenotype correlation. Furthermore, we found that the position of the mutation in the keratin gene is not the only determinant for severity of the disease; the nature of the amino acid substitution should also be considered when predicting the severity of the EBS disorder.

Publication types

  • English Abstract
  • Research Support, Non-U.S. Gov't

MeSH terms

  • DNA Mutational Analysis
  • Denmark
  • Epidermolysis Bullosa Simplex / genetics*
  • Female
  • Genotype
  • Humans
  • Keratins / genetics
  • Male
  • Pedigree
  • Phenotype
  • Polymerase Chain Reaction

Substances

  • Keratins