Clinical profile and mutation analysis of xeroderma pigmentosum in Indian patients

Indian J Dermatol Venereol Leprol. 2015 Jan-Feb;81(1):16-22. doi: 10.4103/0378-6323.148559.

Abstract

Background: Xeroderma pigmentosum (XP) is an autosomal recessive genetic disorder characterized by cutaneous and ocular photosensitivity and an increased risk of developing cutaneous neoplasms. Progressive neurological abnormalities develop in a quarter of XP patients.

Aim: To study the clinical profile and perform a mutation analysis in Indian patients with xeroderma pigmentosum.

Methods: Ten families with 13 patients with XP were referred to our clinic over 2 years. The genes XPA, XPB and XPC were sequentially analyzed till a pathogenic mutation was identified.

Results: Homozygous mutations in the XPA gene were seen in patients with moderate to severe mental retardation (6/10 families) but not in those without neurological features. Two unrelated families with a common family name and belonging to the same community from Maharashtra were found to have an identical mutation in the XPA gene, namely c.335_338delTTATinsCATAAGAAA (p.F112SfsX2). Testing of the XPC gene in two families with four affected children led to the identification of the novel mutations c.1243C>T or p.R415X and c.1677C>A or p.Y559X. In two families, mutations could not be identified in XPA, XPB and XPC genes.

Limitation: The sample size is small.

Conclusion: Indian patients who have neurological abnormalities associated with XP should be screened for mutations in the XPA gene.

MeSH terms

  • Adult
  • Child
  • Child, Preschool
  • DNA Helicases / genetics*
  • DNA Mutational Analysis
  • DNA-Binding Proteins / genetics*
  • Female
  • Homozygote
  • Humans
  • India
  • Intellectual Disability / genetics*
  • Male
  • Xeroderma Pigmentosum / complications
  • Xeroderma Pigmentosum / genetics*
  • Xeroderma Pigmentosum Group A Protein / genetics*

Substances

  • DNA-Binding Proteins
  • XPA protein, human
  • Xeroderma Pigmentosum Group A Protein
  • XPBC-ERCC-3 protein
  • XPC protein, human
  • DNA Helicases