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PMID: 17284601 Published · ppublish English Journal Article Research Support, N.I.H., Extramural Research Support, Non-U.S. Gov't

Telomere dysfunction as a cause of genomic instability in Werner syndrome.

Crabbe L, Jauch A, Naeger CM, Holtgreve-Grez H, Karlseder J

Abstract

Werner syndrome (WS) is a rare human premature aging disease caused by mutations in the gene encoding the RecQ helicase WRN. In addition to the aging features, this disorder is marked by genomic instability, associated with an elevated incidence of cancer. Several lines of evidence suggest that telomere dysfunction is associated with the aging phenotype of the syndrome; however, the origin of the genomic instability observed in WS cells and the reason for the high incidence of cancer in WS have not been established. We previously proposed that WRN helicase activity was necessary to prevent dramatic telomere loss during DNA replication. Here we demonstrate that replication-associated telomere loss is responsible for the chromosome fusions found in WS fibroblasts. Moreover, using metaphase analysis we show that telomere elongation by telomerase can significantly reduce the appearance of new chromosomal aberrations in cells lacking WRN, similar to complementation of WS cells with WRN. Our results suggest that the genome instability in WS cells depends directly on telomere dysfunction, linking chromosome end maintenance to chromosomal aberrations in this disease.

MeSH Terms
Chromosome Aberrations DNA Replication Exodeoxyribonucleases Fibroblasts/metabolism,pathology Genomic Instability Humans Metaphase RecQ Helicases/deficiency Telomerase/metabolism Telomere Werner Syndrome/etiology,genetics,pathology Werner Syndrome Helicase
Chemicals
Telomerase Exodeoxyribonucleases RecQ Helicases WRN protein, human Werner Syndrome Helicase
Authors & Affiliations
5 authors, click to expand affiliations / ORCID
Crabbe Laure
Regulatory Biology Department, The Salk Institute for Biological Studies, 10010 North Torrey Pines Road, San Diego, CA 92037, USA.
Jauch Anna
Naeger Colleen M
Holtgreve-Grez Heidi
Karlseder Jan
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Article Info
Journal
Proceedings of the National Academy of Sciences of the United States of America
Abbr.
Proc Natl Acad Sci U S A
ISSN
0027-8424
Published
2007-02-13
Epub
2007-00-06
Pages
2205-10
Language
English
Region
United States
NLM ID
7505876
PMCID
PMC1794219
Subset
IM
Grants
NIA NIH HHS · R01 AG025837 · United States
PHS HHS · R01 069525 · United States
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