A novel mutation of keratin 9 in epidermolytic palmoplantar keratoderma combined with knuckle pads

Am J Med Genet A. 2003 Jul 30;120A(3):345-9. doi: 10.1002/ajmg.a.20090.

Abstract

Epidermolytic palmoplantar keratoderma (EPPK) is an autosomal dominantly inherited disease. We studied a family from Shandong, China, having patients suffering from EPPK with a unique symptom-knuckle pads. We noticed that both the hyperkeratosis and knuckle pads in the Chinese family were friction-related. Candidate gene analysis was carried out using linkage analysis and direct sequencing. A novel L160F mutation in keratin 9 was found, and its effects on the secondary structure of keratin 9 were studied. We predict that the L160F mutation is also responsible for the knuckle pads in the family. Our study provides a new clue for the study of the function of keratin 9.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • DNA Mutational Analysis
  • Genetic Linkage
  • Hand Deformities, Congenital / genetics*
  • Hand Deformities, Congenital / physiopathology
  • Humans
  • Keratins / genetics*
  • Keratoderma, Palmoplantar / genetics*
  • Keratoderma, Palmoplantar / physiopathology
  • Mutation
  • Protein Structure, Secondary
  • Sequence Analysis, DNA

Substances

  • Keratins