Confirmation of the origin of NISCH syndrome

Hum Mutat. 2006 May;27(5):408-10. doi: 10.1002/humu.20333.

Abstract

Neonatal ichthyosis-sclerosing cholangitis (NISCH) syndrome, a rare autosomal recessive ichthyosis syndrome characterized by scalp hypotrichosis, scarring alopecia, ichthyosis, and sclerosing cholangitis, was described for the first time in 2002. It is caused by a mutation in the gene coding for the tight junction protein claudin-1. Only four patients carrying the same mutation of the CLDN1 gene have been described until now. We report a patient presenting with the clinical characteristics of NISCH syndrome and carrying a novel mutation in the CLDN1 gene.

Publication types

  • Case Reports

MeSH terms

  • Adolescent
  • Cholangitis, Sclerosing / diagnosis
  • Cholangitis, Sclerosing / genetics*
  • Claudin-1
  • Female
  • Frameshift Mutation
  • Humans
  • Ichthyosis / diagnosis
  • Ichthyosis / genetics*
  • Membrane Proteins / genetics*
  • Membrane Proteins / metabolism
  • Skin / cytology
  • Syndrome

Substances

  • CLDN1 protein, human
  • Claudin-1
  • Membrane Proteins

Associated data

  • OMIM/603718
  • OMIM/607626