PUBLICATION
The Generation of Zebrafish Mariner Model Using the CRISPR/Cas9 System
- Authors
- Zou, B., Desmidt, A.A., Mittal, R., Yan, D., Richmond, M., Tekin, M., Liu, X., Lu, Z.
- ID
- ZDB-PUB-190702-7
- Date
- 2019
- Source
- Anatomical record (Hoboken, N.J. : 2007) 303(3): 556-562 (Journal)
- Registered Authors
- Keywords
- CRISPR/Cas9, hearing loss, mariner, myo7aa, zebrafish
- MeSH Terms
-
- Phenotype*
- Zebrafish/genetics
- Clustered Regularly Interspaced Short Palindromic Repeats
- Disease Models, Animal
- Myosins/genetics
- CRISPR-Cas Systems*
- Gene Editing/methods*
- Animals
- Zebrafish Proteins/genetics*
- Deafness/genetics*
- Behavior, Animal/physiology
- PubMed
- 31260171 Full text @ Anat. Rec. (Hoboken)
Citation
Zou, B., Desmidt, A.A., Mittal, R., Yan, D., Richmond, M., Tekin, M., Liu, X., Lu, Z. (2019) The Generation of Zebrafish Mariner Model Using the CRISPR/Cas9 System. Anatomical record (Hoboken, N.J. : 2007). 303(3):556-562.
Abstract
Targeted genome editing mediated by clustered, regularly interspaced, short palindromic repeat (CRISPR)/CRISPR-associated nuclease 9 (Cas9) technology has emerged as a powerful tool for gene function studies and has great potential for gene therapy. Although CRISPR/Cas9 has been widely used in many research fields, only a few successful zebrafish models have been established using this technology in hearing research. In this study, we successfully created zebrafish mariner mutants by targeting the motor head domain of Myo7aa using CRISPR/Cas9. The CRISPR/Cas9-generated mutants showed unbalanced swimming behavior and disorganized sterocilia of inner ear hair cells, which resemble the phenotype of the zebrafish mariner mutants. In addition, we found that CRISPR/Cas9-generated mutants have reduced number of stereociliary bundles of inner-ear hair cells and have significant hearing loss. Furthermore, phenotypic analysis was performed on F0 larvae within the first week post fertilization, which dramatically shortens data collection period. Therefore, results of this study showed that CRISPR/Cas9 is a quick and effective method to generate zebrafish mutants as a model for studying human genetic deafness . This article is protected by copyright. All rights reserved.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping