The progress of CRISPR/Cas9-mediated gene editing in generating mouse/zebrafish models of human skeletal diseases

Publication date: Available online 13 June 2019Source: Computational and Structural Biotechnology JournalAuthor(s): Nan Wu, Bowen Liu, Huakang Du, Sen Zhao, Yaqi Li, Xi Cheng, Shengru Wang, Jiachen Lin, Junde Zhou, Guixing Qiu, Jianguo Zhang, Zhihong WuAbstractGenetic factors play a substantial role in the etiology of skeletal diseases, which involve 1) defects in skeletal development, including intramembranous ossification defects and endochondral ossification defects; 2) defects in skeletal metabolism, including late bone growth and bone remodeling; 3) defects in early developmental processes related to skeletal diseases, such as neural crest cell (NCC) and cilia defects; 4) disturbance of the cellular signaling pathways which potentially affect bone growth. Efficient and high-throughput genetic methods have enabled the exploration and verification of disease-causing genes and variants. Animal models including mouse and zebrafish have been extensively used in functional mechanism studies of causal genes and variants. The conventional approaches of generating mutant animal models include spontaneous mutagenesis, random integration, and targeted integration via mouse embryonic stem cells. These approaches are costly and time-consuming. Recent development and application of gene-editing tools, especially the CRISPR/Cas9 system, has significantly accelerated the process of gene-editing in diverse organisms. Here we review both mice and zebrafish models of human skeletal disease...
Source: Computational and Structural Biotechnology Journal - Category: Biotechnology Source Type: research