Connexin 26 (GJB2) mutation in an Argentinean patient with keratitis-ichthyosis-deafness (KID) syndrome: a case report

BMC Med Genet. 2016 May 4;17(1):37. doi: 10.1186/s12881-016-0298-y.

Abstract

Background: Keratitis-Ichthyosis-Deafness (KID) syndrome is a rare condition characterized by pre-lingual sensorineural deafness with skin hyperkeratinization. The primary cause of the disease is a loss-of-function mutation in the GJB2 gene. Mutations in Argentinean patients have not been described.

Case presentation: We studied a 2 year-old boy with bilateral congenital sensorineural deafness with dry skin over the entire body, hypotrichosis of the scalp, thin and light-blond hair. Analysis of the GJB2 gene nucleotide sequence revealed the substitution of guanine-148 by adenine predicted to result in an Asp50Asn amino acid substitution.

Conclusion: This is the first KID report in a patient from Argentina. This de novo mutation proved to be the cause of keratitis-ichthyosis-deafness syndrome (KID-syndrome) in the patient, and has implications in medical genetic practice.

Keywords: Connexin; Deafness; GJB2; KID syndrome; Mutations; p.Asp50Asn.

Publication types

  • Case Reports

MeSH terms

  • Amino Acid Substitution*
  • Argentina
  • Child, Preschool
  • Connexin 26
  • Connexins / genetics*
  • Genetic Predisposition to Disease
  • Humans
  • Keratitis / genetics*
  • Male
  • Sequence Analysis, DNA / methods*

Substances

  • Connexins
  • GJB2 protein, human
  • Connexin 26

Supplementary concepts

  • Keratitis-Ichthyosis-Deafness Syndrome