Danio rerio breed:ZDR Genome sequencing. Danio rerio breed:ZDR

The rapid feasibility of CRISPR-Cas9 gene editing for therapeutic applications has perpetuated the need to assess potentially deleterious off-target editing events. Here we show the importance of controlling for genetic drift using CRISPR-Cas9 edited zebrafish from the same founding parents across two generations during unbiased variant detection. We did not observe an increase in the number of systematic, transmissible variants in the CRISPR-Cas9 edited individuals in the second generation, arguing that in vivo editing is unlikely to precipitate an inflation of likely deleterious point mutations.