Identification of a novel mutation in 3beta-hydroxysteroid-Delta8-Delta7-isomerase in a case of Conradi-Hünermann-Happle syndrome

Exp Dermatol. 2001 Aug;10(4):286-9. doi: 10.1034/j.1600-0625.2001.100409.x.

Abstract

The X-linked dominant Conradi-Hünermann-Happle (CDPX2, MIM 302960) syndrome belongs to the rare, heterogeneous group of diseases called chondrodysplasia punctata. The disease has been connected recently with deficiency of 3beta-hydroxysteroid-Delta8-Delta7-isomerase (also called emopamil-binding protein, EBP), catalysing an intermediate step in the conversion of lanosterol to cholesterol (1, 2). We report a case of CDPX2 with a new missense mutation (C-->G 439) in exon 4, leading to a R147G aminoacid substitution in the EBP.

Publication types

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

MeSH terms

  • Adolescent
  • Amino Acid Substitution
  • Base Sequence / genetics
  • Chondrodysplasia Punctata / genetics*
  • Electrophoresis, Polyacrylamide Gel
  • Female
  • Heteroduplex Analysis
  • Heterozygote
  • Humans
  • Mutation, Missense / genetics*
  • Pedigree
  • Polymerase Chain Reaction
  • Reference Values
  • Steroid Isomerases / genetics*

Substances

  • Steroid Isomerases
  • delta(8)-delta(7)-sterol isomerase