The role of endothelial cilia in post-embryonic vascular development
- Elworthy, S., Savage, A.M., Wilkinson, R.N., Malicki, J.J., Chico, T.J.A.
- Developmental Dynamics : an official publication of the American Association of Anatomists 248(6): 410-425 (Journal)
- Registered Authors
- Chico, Tim J., Elworthy, Stone, Malicki, Jarema, Wilkinson, Robert
- Zebrafish, angiogenesis, cilia, elipsa, vascular development, vascular mural cells
- MeSH Terms
- Endothelium, Vascular*
- 30980582 Full text @ Dev. Dyn.
Elworthy, S., Savage, A.M., Wilkinson, R.N., Malicki, J.J., Chico, T.J.A. (2019) The role of endothelial cilia in post-embryonic vascular development. Developmental Dynamics : an official publication of the American Association of Anatomists. 248(6):410-425.
Background Cilia are essential for morphogenesis and maintenance of many tissues. Loss-of-function of cilia in early zebrafish development causes a range of vascular defects including cerebral haemorrhage and reduced arterial vascular mural cell coverage. In contrast, loss of endothelial cilia in mice has little effect on vascular development. We therefore used a conditional rescue approach to induce endothelial cilia ablation after early embryonic development and examined the effect on vascular development and mural cell development in post-embryonic, juvenile and adult zebrafish.
Results ift54(elipsa) mutant zebrafish are unable to form cilia. We rescued cilia formation and ameliorated the phenotype of ift54 mutants using a novel Tg(ubi:loxP-ift54-loxP-myr-mcherry,myl7:EGFP)sh488 transgene expressing wildtype ift54 flanked by recombinase sites, then used a Tg(kdrl:cre)s898 transgene to induce endothelial-specific inactivation of ift54 at post-embryonic ages. Fish without endothelial ift54 function could survive to adulthood and exhibited no vascular defects. Endothelial inactivation of ift54 did not affect development of tagln-positive vascular mural cells around either the aorta or the caudal fin vessels, nor formation of vessels after tailfin resection in adult animals.
Conclusions Endothelial cilia are not essential for development and remodelling of the vasculature in juvenile and adult zebrafish when inactivated after embryogenesis. This article is protected by copyright. All rights reserved.
Genes / Markers
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Engineered Foreign Genes