Lethal Netherton syndrome due to homozygous p.Arg371X mutation in SPINK5

Pediatr Dermatol. 2013 Jul-Aug;30(4):e65-7. doi: 10.1111/pde.12076. Epub 2013 Jan 17.

Abstract

Here we report a lethal case of Netherton syndrome presenting with neurologic complications, hypernatremic dehydration, failure to thrive, and episodes of sepsis. Molecular analysis of the serine protease inhibitor Kazal-type 5 gene identified a homozygous mutation (c.1111C>T, p.Arg371X). This case highlights the importance of early diagnosis to start appropriate care in a timely fashion and prevent disease complications.

Publication types

  • Case Reports

MeSH terms

  • Fatal Outcome
  • Homozygote
  • Humans
  • Infant
  • Male
  • Netherton Syndrome / genetics*
  • Netherton Syndrome / pathology
  • Netherton Syndrome / physiopathology
  • Point Mutation*
  • Proteinase Inhibitory Proteins, Secretory / genetics*
  • Serine Peptidase Inhibitor Kazal-Type 5

Substances

  • Proteinase Inhibitory Proteins, Secretory
  • SPINK5 protein, human
  • Serine Peptidase Inhibitor Kazal-Type 5