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Enhancing site-specific DNA integration by a Cas9 nuclease fused with a DNA donor-binding domain

Shufeng Ma, Xinlong Wang, Yongfei Hu, Jie Lv, Chengfang Liu, Kaitong Liao, Xiaohua Guo, Dong Wang, Ying Lin, Zhili Rong

2020Nucleic Acids Research36 citationsDOIOpen Access PDF

Abstract

The CRISPR/Cas system is widely used for genome editing. However, robust and targeted insertion of a DNA segment remains a challenge. Here, we present a fusion nuclease (Cas9-N57) to enhance site-specific DNA integration via a fused DNA binding domain of Sleeping Beauty transposase to tether the DNA segment to the Cas9/sgRNA complex. The insertion was unidirectional and specific, and DNA fragments up to 12 kb in length were successfully integrated. As a test of the system, Cas9-N57 mediated the insertion of a CD19-specific chimeric antigen receptor (CD19-CAR) cassette into the AAVS1 locus in human T cells, and induced intrahepatic cholangiocarcinoma in mice by simultaneously mediating the insertion of oncogenic KrasG12D into the Rosa26 locus and disrupting Trp53 and Pten. Moreover, the nuclease-N57 fusion proteins based on AsCpf1 (AsCas12a) and CjCas9 exhibited similar activity. These findings demonstrate that CRISPR-associated nuclease-N57 protein fusion is a powerful tool for targeted DNA insertion and holds great potential for gene therapy applications.

Topics & Concepts

BiologyNucleaseDNACas9Binding siteGeneticsComputational biologyMolecular biologyCRISPRGeneCRISPR and Genetic EngineeringAdvanced biosensing and bioanalysis techniquesRNA Interference and Gene Delivery