Genome editing with the HDR-enhancing DNA-PKcs inhibitor AZD7648 causes large-scale genomic alterations.

Cullot, Grégoire; Aird, Eric J; Schlapansky, Moritz F; Yeh, Charles D; van de Venn, Lilly; Vykhlyantseva, Iryna; Kreutzer, Susanne; Mailänder, Dominic et al. · Nat Biotechnol · 2025

basic_science · Level V

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Abstract

The DNA-PKcs inhibitor AZD7648 enhances CRISPR-Cas9-directed homology-directed repair efficiencies, with potential for clinical utility, but its possible on-target consequences are unknown. We found that genome editing with AZD7648 causes frequent kilobase-scale and megabase-scale deletions, chromosome arm loss and translocations. These large-scale chromosomal alterations evade detection through typical genome editing assays, prompting caution in deploying AZD7648 and reinforcing the need to investigate multiple types of potential editing outcomes.

Medical subject headings