Genome instability is a feature of nearly all cancers and can be exploited for therapy. In addition, a growing number of genome maintenance genes have been associated with developmental disorders. Efforts to understand the role of genome instability in these processes will be greatly facilitated by a more comprehensive understanding of their genetic network. We highlight recent genetic screens in model organisms that have assisted in the discovery of novel regulators of genome stability and focus on the contribution of mice as a model organism to understanding the role of genome instability during embryonic development, tumour formation and cancer therapy.
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