PUBLICATION
Centrosomal protein FOR20 is essential for cilia-dependent development in zebrafish embryos
- Authors
- Xie, S., Jin, J., Xu, Z., Huang, Y., Zhang, W., Zhao, L., Lo, L.J., Peng, J., Liu, W., Wang, F., Shu, Q., Zhou, T.
- ID
- ZDB-PUB-181127-82
- Date
- 2018
- Source
- FASEB journal : official publication of the Federation of American Societies for Experimental Biology 33(3): 3613-3622 (Journal)
- Registered Authors
- Keywords
- Kupffer’s vesicle, ciliogenesis, ciliopathies, morphants
- MeSH Terms
-
- Animals
- Body Patterning/genetics
- Centrosome/physiology*
- Cilia/genetics*
- Embryonic Development/genetics
- Gene Expression Regulation, Developmental/genetics
- Gene Knockdown Techniques/methods
- Morpholinos/genetics
- Mutation/genetics
- RNA, Messenger/genetics
- Zebrafish/genetics*
- Zebrafish Proteins/genetics*
- PubMed
- 30475641 Full text @ FASEB J.
Citation
Xie, S., Jin, J., Xu, Z., Huang, Y., Zhang, W., Zhao, L., Lo, L.J., Peng, J., Liu, W., Wang, F., Shu, Q., Zhou, T. (2018) Centrosomal protein FOR20 is essential for cilia-dependent development in zebrafish embryos. FASEB journal : official publication of the Federation of American Societies for Experimental Biology. 33(3):3613-3622.
Abstract
Centrosomal proteins play critical roles in ciliogenesis. Mutations in many centrosomal proteins have been documented to contribute to developmental defects and cilia-related diseases. Centrosomal protein fibroblast growth factor receptor 1 oncogene partner-related protein of 20 kDa (FOR20) is crucial for ciliogenesis in mammalian cells and the unicellular eukaryote Paramecium; however, the biologic significance of FOR20 in vertebrate development remains unclear. We cloned the zebrafish homolog of the for20 gene and found that for20 mRNA is enriched in ciliated tissues during early zebrafish development. Knockdown of for20 by morpholino oligonucleotides in zebrafish results in multiple ciliary phenotypes, including curved body, hydrocephaly, pericardial edema, kidney cysts, and left-right asymmetry defects. for20 morphants show reduced number and length of cilia in Kupffer's vesicle and pronephric ducts. High-speed video microscopy reveals that cilia in most for20 morphants are consistently paralyzed or beat arrhythmically. To confirm the ciliary phenotypes of for20 morphants, we used the CRISPR/Cas9 system to disrupt for20 gene in zebrafish. for20 mutants exhibit multiple ciliary phenotypes resembling the defects in for20 morphants. All of these phenotypes in for20 morphants and mutants are significantly reversed by exogenous expression of for20 mRNA. Taken together, these data suggest that FOR20 is required for cilia-mediated processes during zebrafish embryogenesis.-Xie, S., Jin, J., Xu, Z., Huang, Y., Zhang, W., Zhao, L., Lo, L. J., Peng, J., Liu, W., Wang, F., Shu, Q., Zhou, T. Centrosomal protein FOR20 is essential for cilia-dependent development in zebrafish embryos.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping