Novel mutation in the human HPRT1 gene and the Lesch-Nyhan disease

Nucleosides Nucleotides Nucleic Acids. 2017 Nov 2;36(11):704-711. doi: 10.1080/15257770.2017.1395037. Epub 2017 Nov 29.

Abstract

Lesch-Nyhan disease (LND) is a rare X-linked inherited neurogenetic disorder of purine metabolism in which the enzyme, hypoxanthine-guanine phosphoribosyltransferase (HGprt) is defective. The authors report a novel point mutation that led to HGprt-related neurological dysfunction (HND) in a family in which there was a missense mutation in exon 6 of the coding region of the HPRT1 gene: g.34938G>T, c.403G>T, p.D135Y. Molecular diagnosis is consistent with the genetic heterogeneity of the HPRT1 gene responsible for HGprt deficiency. It allows fast, accurate carrier detection and genetic counseling.

Keywords: HND; HPRT1 gene, HGprt enzyme; Lesch-Nyhan disease; amyloid precursor protein; epigenetics; epistasis; mutation.

MeSH terms

  • Base Sequence
  • Child, Preschool
  • Exons / genetics
  • Female
  • Humans
  • Hypoxanthine Phosphoribosyltransferase / genetics*
  • Hypoxanthine Phosphoribosyltransferase / metabolism
  • Lesch-Nyhan Syndrome / enzymology*
  • Lesch-Nyhan Syndrome / genetics*
  • Male
  • Mutation, Missense*
  • Pedigree

Substances

  • Hypoxanthine Phosphoribosyltransferase