Cellular Werner phenotypes in mice expressing a putative dominant-negative human WRN gene

Genetics. 2000 Jan;154(1):357-62. doi: 10.1093/genetics/154.1.357.

Abstract

Mutations at the Werner helicase locus (WRN) are responsible for the Werner syndrome (WS). WS patients prematurely develop an aged appearance and various age-related disorders. We have generated transgenic mice expressing human WRN with a putative dominant-negative mutation (K577M-WRN). Primary tail fibroblast cultures from K577M-WRN mice showed three characteristics of WS cells: hypersensitivity to 4-nitroquinoline-1-oxide (4NQO), reduced replicative potential, and reduced expression of the endogenous WRN protein. These data suggest that K577M-WRN mice may provide a novel mouse model for the WS.

Publication types

  • Research Support, U.S. Gov't, P.H.S.

MeSH terms

  • 4-Nitroquinoline-1-oxide / analogs & derivatives
  • 4-Nitroquinoline-1-oxide / pharmacology
  • Alleles
  • Animals
  • Cell Division
  • DNA Helicases / genetics*
  • Down-Regulation
  • Exodeoxyribonucleases
  • Genes, Dominant*
  • Humans
  • Mice
  • Mice, Transgenic
  • Phenotype
  • Quinolones / pharmacology
  • RecQ Helicases
  • Werner Syndrome / genetics*
  • Werner Syndrome Helicase

Substances

  • 4-nitroquinolone-1-oxide
  • Quinolones
  • 4-Nitroquinoline-1-oxide
  • Exodeoxyribonucleases
  • DNA Helicases
  • RecQ Helicases
  • WRN protein, human
  • Werner Syndrome Helicase