The Progress of CRISPR/Cas9-Mediated Gene Editing in Generating Mouse/Zebrafish Models of Human Skeletal Diseases
| Autor: | Xi Cheng, Deciphering Disorders Involving Scoliosis, Nan Wu, Junde Zhou, Jiachen Lin, Zhihong Wu, COmorbidities (Disco) study, Shengru Wang, Yaqi Li, Sen Zhao, Jianguo Zhang, Huakang Du, Bowen Liu, Guixing Qiu |
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| Jazyk: | angličtina |
| Rok vydání: | 2019 |
| Předmět: |
Bone growth
0303 health sciences Cas9 lcsh:Biotechnology Biophysics Mutagenesis (molecular biology technique) Computational biology Review Article Biology biology.organism_classification Biochemistry Computer Science Applications 03 medical and health sciences 0302 clinical medicine Genome editing Structural Biology 030220 oncology & carcinogenesis lcsh:TP248.13-248.65 Intramembranous ossification Genetics CRISPR Endochondral ossification Zebrafish 030304 developmental biology Biotechnology |
| Zdroj: | Computational and Structural Biotechnology Journal, Vol 17, Iss, Pp 954-962 (2019) Computational and Structural Biotechnology Journal |
| ISSN: | 2001-0370 |
| Popis: | Genetic factors play a substantial role in the etiology of skeletal diseases, which involve 1) defects in skeletal development, including intramembranous ossification and endochondral ossification; 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 functions; 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 diseases generated by CRISPR/Cas9 system, and their contributions to deciphering the underpins of disease mechanisms. Graphical Abstract Unlabelled Image |
| Databáze: | OpenAIRE |
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