WRN exonuclease structure and molecular mechanism imply an editing role in DNA end processing

Nat Struct Mol Biol. 2006 May;13(5):414-22. doi: 10.1038/nsmb1088. Epub 2006 Apr 23.

Abstract

WRN is unique among the five human RecQ DNA helicases in having a functional exonuclease domain (WRN-exo) and being defective in the premature aging and cancer-related disorder Werner syndrome. Here, we characterize WRN-exo crystal structures, biochemical activity and participation in DNA end joining. Metal-ion complex structures, active site mutations and activity assays reveal a nuclease mechanism mediated by two metal ions. The DNA end-binding Ku70/80 complex specifically stimulates WRN-exo activity, and structure-based mutational inactivation of WRN-exo alters DNA end joining in human cells. We furthermore establish structural and biochemical similarities of WRN-exo to DnaQ-family replicative proofreading exonucleases, describing WRN-specific adaptations consistent with double-stranded DNA specificity and functionally important conformational changes. These results indicate WRN-exo is a human DnaQ family member and support DnaQ-like proofreading activities stimulated by Ku70/80, with implications for WRN functions in age-related pathologies and maintenance of genomic integrity.

Publication types

  • Research Support, N.I.H., Extramural

MeSH terms

  • Animals
  • Binding Sites
  • Conserved Sequence
  • Crystallography, X-Ray
  • DNA / genetics*
  • DNA / metabolism*
  • DNA Helicases / chemistry*
  • DNA Helicases / classification
  • DNA Helicases / genetics
  • DNA Helicases / metabolism*
  • Exodeoxyribonucleases
  • Humans
  • Metals / chemistry
  • Metals / metabolism
  • Models, Molecular
  • Molecular Sequence Data
  • Protein Conformation
  • Protein Folding
  • RecQ Helicases
  • Sequence Alignment
  • Sequence Homology, Amino Acid
  • Substrate Specificity
  • Werner Syndrome Helicase

Substances

  • Metals
  • DNA
  • Exodeoxyribonucleases
  • DNA Helicases
  • RecQ Helicases
  • WRN protein, human
  • Werner Syndrome Helicase

Associated data

  • PDB/2FBT
  • PDB/2FBV
  • PDB/2FBX
  • PDB/2FBY
  • PDB/2FC0