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Journal ArticleDOI

Single-strand break repair and genetic disease

Keith W. Caldecott
- 01 Aug 2008 - 
- Vol. 9, Iss: 8, pp 619-631
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TLDR
The molecular mechanisms and organization of the DNA-repair pathways that remove single-strand breaks are reviewed and the connection between defects in these pathways and hereditary neurodegenerative disease are discussed.
Abstract
Hereditary defects in the repair of DNA damage are implicated in a variety of diseases, many of which are typified by neurological dysfunction and/or increased genetic instability and cancer. Of the different types of DNA damage that arise in cells, single-strand breaks (SSBs) are the most common, arising at a frequency of tens of thousands per cell per day from direct attack by intracellular metabolites and from spontaneous DNA decay. Here, the molecular mechanisms and organization of the DNA-repair pathways that remove SSBs are reviewed and the connection between defects in these pathways and hereditary neurodegenerative disease are discussed.

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Citations
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Journal ArticleDOI

The DNA-damage response in human biology and disease

TL;DR: The authors' improving understanding of DNA-damage responses is providing new avenues for disease management, and these responses are biologically significant because they prevent diverse human diseases.
Journal ArticleDOI

The DNA Damage Response: Making It Safe to Play with Knives

TL;DR: This review will focus on how the DDR controls DNA repair and the phenotypic consequences of defects in these critical regulatory functions in mammals.
Journal ArticleDOI

Cell cycle, CDKs and cancer: a changing paradigm

TL;DR: Genetic evidence suggests that tumour cells may also require specific interphase CDKs for proliferation, and selective CDK inhibition may provide therapeutic benefit against certain human neoplasias.
Journal ArticleDOI

DNA Damage, Aging, and Cancer

TL;DR: Evidence that cancer and diseases of aging are two sides of the DNAdamage problem is presented, followed by an account of the derailment of genome guardian mechanisms in cancer and of how this cancerspecific phenomenon can be exploited for treatment.
Journal ArticleDOI

Dynamics of DNA damage response proteins at DNA breaks: a focus on protein modifications

TL;DR: How the development of various complementary methodologies has provided valuable insights into the spatiotemporal dynamics of DDR protein assembly/disassembly at sites of DNA strand breaks in eukaryotic cells is outlined.
References
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Journal ArticleDOI

Aprataxin mutations are a rare cause of early onset ataxia in Germany.

TL;DR: A group of 165 early onset ataxia patients were screened for APTX mutations and two non-related patients homozygous for the W293X nonsense mutation were detected, describing several new transcript variants of the APTX gene.
Journal ArticleDOI

E2F1 Regulates the Base Excision Repair Gene XRCC1 and Promotes DNA Repair

TL;DR: In this article, the XRCC1 promoter was cloned into a luciferase reporter to study the upstream mechanisms of the E2F1 transcription factor, which is important for efficient single strand break/base excision repair.

E2F1 Regulates the Base Excision Repair Gene XRCC1 and

TL;DR: The E2F1 transcription factor activates S-phase-promoting genes, mediates apoptosis, and stimulates DNA repair through incompletely understood mechanisms, and new mechanistic insight is provided into the role of the E 2F pathway in maintaining genomic stability.
Journal ArticleDOI

Poly(ADP-ribosylation) reduces the steady-state level of breaks in DNA following treatment of human cells with alkylating agents.

TL;DR: It is suggested that continued synthesis of poly(ADP-ribose) reduces the steady state level of breaks during excision repair of alkylation damage, which is probably mediated by the stimulation of DNA ligase activity.
Journal Article

Role for DNA polymerase beta in response to ionizing radiation

TL;DR: It is shown that mouse embryonic fibroblasts deficient in DNA polymerase beta are considerably more sensitive to ionizing radiation than wild-type cells, but only when confluent, the first evidence of a role for DNA polymerases beta in radiosensitivity in vivo.
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