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Spacer Extension Reconciles Specificity and Activity in High-Fidelity Cas9 Genome Editing

Lu, Z.; Wei, R.; Zheng, R.; Shin, Y.-C.; Zhang, Q.; Li, J.; Wang, X.; Wei, Y.; Liu, B.; Chen, Y.; Zhang, H.; Chen, H.; Ma, L.

2025-01-18 genomics
10.1101/2025.01.16.633458 bioRxiv
Show abstract

AbstractEngineering high-fidelity CRISPR enzymes often leads to reduced cleavage activity, creating a significant hurdle in balancing nuclease specificity and efficiency for clinical applications. Here, we demonstrate that extending the spacer to 21 or 22 nucleotides restores the impaired cleavage activity of SuperFi-Cas9, an engineered high-fidelity Cas9 variant with seven mutations at the PAM-distal region. Structural and mutational analyses reveal that the spacer extension strengthens additional interactions at the PAM-distal end, stabilizing the nuclease- sgRNA-DNA complex, which appears to be disturbed due to the seven mutations. This approach not only provides a high-fidelity Cas9 with uncompromised efficiency but also introduces a novel strategy to enhance CRISPR complex stability. Our findings offer a promising avenue for precise and efficient genome editing, crucial for advancing CRISPR technologies toward clinical translation.

Published in Nature Structural & Molecular Biology (predicted rank #21) · training set

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