High-efficiency transgene integration by homology-directed repair in human primary cells using DNA-PKcs inhibition.


Journal

Nature biotechnology
ISSN: 1546-1696
Titre abrégé: Nat Biotechnol
Pays: United States
ID NLM: 9604648

Informations de publication

Date de publication:
03 Aug 2023
Historique:
received: 29 06 2022
accepted: 28 06 2023
medline: 4 8 2023
pubmed: 4 8 2023
entrez: 3 8 2023
Statut: aheadofprint

Résumé

Therapeutic applications of nuclease-based genome editing would benefit from improved methods for transgene integration via homology-directed repair (HDR). To improve HDR efficiency, we screened six small-molecule inhibitors of DNA-dependent protein kinase catalytic subunit (DNA-PKcs), a key protein in the alternative repair pathway of non-homologous end joining (NHEJ), which generates genomic insertions/deletions (INDELs). From this screen, we identified AZD7648 as the most potent compound. The use of AZD7648 significantly increased HDR (up to 50-fold) and concomitantly decreased INDELs across different genomic loci in various therapeutically relevant primary human cell types. In all cases, the ratio of HDR to INDELs markedly increased, and, in certain situations, INDEL-free high-frequency (>50%) targeted integration was achieved. This approach has the potential to improve the therapeutic efficacy of cell-based therapies and broaden the use of targeted integration as a research tool.

Identifiants

pubmed: 37537500
doi: 10.1038/s41587-023-01888-4
pii: 10.1038/s41587-023-01888-4
doi:

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Informations de copyright

© 2023. The Author(s), under exclusive licence to Springer Nature America, Inc.

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Auteurs

Sridhar Selvaraj (S)

Department of Pediatrics, Stanford University, Stanford, CA, USA.
Institute for Stem Cell Biology and Regenerative Medicine, Stanford University, Stanford, CA, USA.

William N Feist (WN)

Department of Pediatrics, Stanford University, Stanford, CA, USA.
Institute for Stem Cell Biology and Regenerative Medicine, Stanford University, Stanford, CA, USA.

Sebastien Viel (S)

Department of Pediatrics, Stanford University, Stanford, CA, USA.
Institute for Stem Cell Biology and Regenerative Medicine, Stanford University, Stanford, CA, USA.
Immunology Department, Lyon Sud University Hospital, Pierre-Bénite, France.
International Center of Research in Infectiology, Lyon University, INSERM U1111, CNRS UMR 5308, ENS, UCBL, Lyon, France.

Sriram Vaidyanathan (S)

Department of Pediatrics, Stanford University, Stanford, CA, USA.
Institute for Stem Cell Biology and Regenerative Medicine, Stanford University, Stanford, CA, USA.
Center for Gene Therapy, Abigail Wexner Research Institute, Nationwide Children's Hospital, Columbus, OH, USA.
Department of Pediatrics, The Ohio State University, Columbus, OH, USA.

Amanda M Dudek (AM)

Department of Pediatrics, Stanford University, Stanford, CA, USA.
Institute for Stem Cell Biology and Regenerative Medicine, Stanford University, Stanford, CA, USA.

Marc Gastou (M)

Department of Pediatrics, Stanford University, Stanford, CA, USA.
Institute for Stem Cell Biology and Regenerative Medicine, Stanford University, Stanford, CA, USA.

Sarah J Rockwood (SJ)

Department of Pediatrics, Stanford University, Stanford, CA, USA.
Institute for Stem Cell Biology and Regenerative Medicine, Stanford University, Stanford, CA, USA.

Freja K Ekman (FK)

Department of Pediatrics, Stanford University, Stanford, CA, USA.
Institute for Stem Cell Biology and Regenerative Medicine, Stanford University, Stanford, CA, USA.

Aluya R Oseghale (AR)

Department of Pediatrics, Stanford University, Stanford, CA, USA.
Institute for Stem Cell Biology and Regenerative Medicine, Stanford University, Stanford, CA, USA.

Liwen Xu (L)

Department of Pediatrics, Stanford University, Stanford, CA, USA.
Institute for Stem Cell Biology and Regenerative Medicine, Stanford University, Stanford, CA, USA.

Mara Pavel-Dinu (M)

Department of Pediatrics, Stanford University, Stanford, CA, USA.
Institute for Stem Cell Biology and Regenerative Medicine, Stanford University, Stanford, CA, USA.

Sofia E Luna (SE)

Department of Pediatrics, Stanford University, Stanford, CA, USA.
Institute for Stem Cell Biology and Regenerative Medicine, Stanford University, Stanford, CA, USA.

M Kyle Cromer (MK)

Department of Pediatrics, Stanford University, Stanford, CA, USA.
Institute for Stem Cell Biology and Regenerative Medicine, Stanford University, Stanford, CA, USA.
Department of Surgery, University of California, San Francisco, San Francisco, CA, USA.

Ruhi Sayana (R)

Department of Pediatrics, Stanford University, Stanford, CA, USA.
Institute for Stem Cell Biology and Regenerative Medicine, Stanford University, Stanford, CA, USA.

Natalia Gomez-Ospina (N)

Department of Pediatrics, Stanford University, Stanford, CA, USA.
Institute for Stem Cell Biology and Regenerative Medicine, Stanford University, Stanford, CA, USA.

Matthew H Porteus (MH)

Department of Pediatrics, Stanford University, Stanford, CA, USA. mporteus@stanford.edu.
Institute for Stem Cell Biology and Regenerative Medicine, Stanford University, Stanford, CA, USA. mporteus@stanford.edu.

Classifications MeSH