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Mutation frequency is not increased in CRISPR–Cas9-edited mice

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Fig. 1: Parent–progeny study.

Data Availability

The data are available at SRA under project number PRJNA470569.


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This study was funded through the IRPs of NIDDK and NHLBI. Research support was received from the NIH Director’s Challenge Innovation Award program (to L.H.). We also thank the team of the NIH HPC Biowulf cluster, especially W. Resch, for their advice, as well as their continued maintenance of hardware and software. We acknowledge the Genomics Platform at The Broad Institute for their support and services provided for the generation of the WGS data.

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Authors and Affiliations



M.W., H.E.S., C.L., C.W. and L.H. designed the study. C.L. and C.W. conducted mouse experiments. M.W. and H.E.S. carried out computational analyses. M.W., H.E.S. and L.H. wrote the manuscript.

Corresponding authors

Correspondence to Michaela Willi or Lothar Hennighausen.

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Competing interests

The authors declare no competing interests.

Integrated supplementary information

Supplementary Figure 1 Equivalence test.

(a) The equivalence test for the de novo SNPs is significant, which verifies that the number of de novo SNPs is equivalent. (CRISPR and control group: n = 6; δ = +/- 1.35) (b) The equivalence test for the de novo indels is inconclusive, as the 90% CI reaches over zero and the lower boundary (CRISPR and control group: n = 6; δ = +/- 0.82).

Supplementary information

Supplementary Text and Figures

Supplementary Figure 1, Supplementary Methods, Supplementary Notes 1–4 and Supplementary Tables 1–7

Reporting Summary

Supplementary Data

De novo mutations in CRISPR–Cas9-edited and control mice

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Willi, M., Smith, H.E., Wang, C. et al. Mutation frequency is not increased in CRISPR–Cas9-edited mice. Nat Methods 15, 756–758 (2018).

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