An optimized SpCas9 high-fidelity variant for direct protein delivery.

CRISPR-Cas HDR RNP genome editing hematopoietic stem cells high-fidelity Cas9 protein delivery specificity

Journal

Molecular therapy : the journal of the American Society of Gene Therapy
ISSN: 1525-0024
Titre abrégé: Mol Ther
Pays: United States
ID NLM: 100890581

Informations de publication

Date de publication:
05 07 2023
Historique:
received: 08 12 2022
revised: 08 02 2023
accepted: 07 03 2023
pmc-release: 05 07 2024
medline: 10 7 2023
pubmed: 12 3 2023
entrez: 11 3 2023
Statut: ppublish

Résumé

Electroporation of the Cas9 ribonucleoprotein (RNP) complex offers the advantage of preventing off-target cleavages and potential immune responses produced by long-term expression of the nuclease. Nevertheless, the majority of engineered high-fidelity Streptococcus pyogenes Cas9 (SpCas9) variants are less active than the wild-type enzyme and are not compatible with RNP delivery. Building on our previous studies on evoCas9, we developed a high-fidelity SpCas9 variant suitable for RNP delivery. The editing efficacy and precision of the recombinant high-fidelity Cas9 (rCas9HF), characterized by the K526D substitution, was compared with the R691A mutant (HiFi Cas9), which is currently the only available high-fidelity Cas9 that can be used as an RNP. The comparative analysis was extended to gene substitution experiments where the two high fidelities were used in combination with a DNA donor template, generating different ratios of non-homologous end joining (NHEJ) versus homology-directed repair (HDR) for precise editing. The analyses revealed a heterogeneous efficacy and precision indicating different targeting capabilities between the two variants throughout the genome. The development of rCas9HF, characterized by an editing profile diverse from the currently used HiFi Cas9 in RNP electroporation, increases the genome editing solutions for the highest precision and efficient applications.

Identifiants

pubmed: 36905119
pii: S1525-0016(23)00128-4
doi: 10.1016/j.ymthe.2023.03.007
pmc: PMC10362380
pii:
doi:

Substances chimiques

CRISPR-Associated Protein 9 EC 3.1.-

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

2257-2265

Informations de copyright

Copyright © 2023 The Authors. Published by Elsevier Inc. All rights reserved.

Déclaration de conflit d'intérêts

Declaration of interests A. Casini and A. Cereseto are founders of and hold shares in Alia Therapeutics, a genome editing company. A. Casini is an employee of Alia Therapeutics, and A. Cereseto is a consultant for Alia Therapeutics. A patent application covering the technology disclosed in this manuscript has been filed, and A. Casini and A. Cereseto are listed as inventors.

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Auteurs

Eleonora Pedrazzoli (E)

Department CIBIO, Laboratory of Molecular Virology, University of Trento, Via Sommarive 9, 38123 Trento, Italy.

Andrea Bianchi (A)

Department CIBIO, Laboratory of Molecular Virology, University of Trento, Via Sommarive 9, 38123 Trento, Italy.

Alessandro Umbach (A)

Department CIBIO, Laboratory of Molecular Virology, University of Trento, Via Sommarive 9, 38123 Trento, Italy.

Simone Amistadi (S)

Department CIBIO, Laboratory of Molecular Virology, University of Trento, Via Sommarive 9, 38123 Trento, Italy.

Mégane Brusson (M)

Imagine Institute, Laboratory of Chromatin and Gene Regulation During Development, Université de Paris, INSERM UMR 1163, Paris, France.

Giacomo Frati (G)

Imagine Institute, Laboratory of Chromatin and Gene Regulation During Development, Université de Paris, INSERM UMR 1163, Paris, France.

Matteo Ciciani (M)

Department CIBIO, Laboratory of Molecular Virology, University of Trento, Via Sommarive 9, 38123 Trento, Italy.

Kalina Aleksandra Badowska (KA)

Alia Therapeutics, 38123 Trento, Italy.

Daniele Arosio (D)

Biophysics Institute, National Research Council of Italy, 38123 Trento, Italy.

Annarita Miccio (A)

Imagine Institute, Laboratory of Chromatin and Gene Regulation During Development, Université de Paris, INSERM UMR 1163, Paris, France.

Anna Cereseto (A)

Department CIBIO, Laboratory of Molecular Virology, University of Trento, Via Sommarive 9, 38123 Trento, Italy. Electronic address: anna.cereseto@unitn.it.

Antonio Casini (A)

Alia Therapeutics, 38123 Trento, Italy. Electronic address: antonio.casini@aliatx.com.

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Classifications MeSH