PUBLICATION
The Progress of CRISPR/Cas9-Mediated Gene Editing in Generating Mouse/Zebrafish Models of Human Skeletal Diseases
- Authors
- Wu, N., Liu, B., Du, H., Zhao, S., Li, Y., Cheng, X., Wang, S., Lin, J., Zhou, J., Deciphering Disorders Involving Scoliosis and COmorbidities (DISCO) study, Qiu, G., Wu, Z., Zhang, J.
- ID
- ZDB-PUB-190801-12
- Date
- 2019
- Source
- Computational and structural biotechnology journal 17: 954-962 (Review)
- Registered Authors
- Keywords
- none
- MeSH Terms
- none
- PubMed
- 31360334 Full text @ Comput Struct Biotechnol J
Citation
Wu, N., Liu, B., Du, H., Zhao, S., Li, Y., Cheng, X., Wang, S., Lin, J., Zhou, J., Deciphering Disorders Involving Scoliosis and COmorbidities (DISCO) study, Qiu, G., Wu, Z., Zhang, J. (2019) The Progress of CRISPR/Cas9-Mediated Gene Editing in Generating Mouse/Zebrafish Models of Human Skeletal Diseases. Computational and structural biotechnology journal. 17:954-962.
Abstract
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.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping