Identification of novel variants of XPA and POLH/XPV genes in xeroderma pigmentosum patients in Vietnam

Per Med. 2024;21(6):341-351. doi: 10.1080/17410541.2024.2393073. Epub 2024 Dec 10.

Abstract

Xeroderma pigmentosum (XP) disorder is recognized as a genetic condition inherited by autosomal recessive fashion. XP results from a defective DNA repair mechanism that significantly increases skin cancer risk. Fifteen Vietnamese patients were investigated with typical clinical manifestations of XP. Eight XP genes (XPA to XPG and POLH/XPV) were sequenced using peripheral blood samples. Overall, three novel variants on the XPA and XPV genes were detected in members of two families. One novel missense variant c.388A>G (p.R130G) of XPA was found in three patients with XP group A, two novel variants: c.680G>A (p.C227Y) and c.1652dupC (p.Gln553Profs*8) of XPV in one patient with XP group F/G. Our study contributes to the recognition of new mutations in XP patients which have not been reported in Human Gene Mutation Database (HGMD).

Keywords: DNA repair; NER; POLH; XPA; genetic variants; nucleotide excision repair; rare autosomal recessive; skin cancer; translesion synthesis; xeroderma pigmentosum.

Plain language summary

[Box: see text].

MeSH terms

  • Adolescent
  • Adult
  • Child
  • Child, Preschool
  • DNA Repair / genetics
  • DNA-Binding Proteins / genetics
  • Female
  • Humans
  • Male
  • Mutation / genetics
  • Pedigree
  • Vietnam
  • Xeroderma Pigmentosum Group A Protein* / genetics
  • Xeroderma Pigmentosum* / genetics
  • Young Adult

Substances

  • Xeroderma Pigmentosum Group A Protein
  • XPA protein, human
  • DNA-Binding Proteins