Analysis of cilia structure and function in zebrafish

Leventea, Eleni; Hazime, Khodor S.; Zhao, Chengtian; Malicki, Jarema

doi:10.1016/bs.mcb.2016.04.016

Cited by 28 publications

(29 citation statements)

References 184 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…Although the rescue of ift54 mutant phenotype in this study was incomplete, most ift54 functions were restored by the conditional transgene used, and in contrast to ift54 tp49 homozygotes, which are extremely abnormal by 5 dpf and die shortly thereafter, some transgenic ift54 tp49 homozygotes survive more than a year. The Zebrafish nose features non‐motile cilia that project from multiciliated dendritic knobs of olfactory sensory neurons and motile cilia that differentiate from multiciliated cells at the rim of the olfactory pit . The poor rescue of the nasal cilia suggests that multiciliated cells may be especially difficult to rescue, as both type of cilia were poorly rescued in our transgenic approach.…”

Section: Discussionmentioning

confidence: 97%

“…The Zebrafish nose features non-motile cilia that project from multiciliated dendritic knobs of olfactory sensory neurons and motile cilia that differentiate from multiciliated cells at the rim of the olfactory pit. 44,45 The poor rescue of the nasal cilia suggests that multiciliated cells may be especially difficult to rescue, as both type of cilia were poorly rescued in our transgenic approach. Similarly, the scoliosis that we observe in rescued ift54 mutant animals may due to defects in multiciliated ependymal cells, since defects in ependymal cell cilia have been shown to cause scoliosis.…”

Section: Discussionmentioning

confidence: 99%

See 1 more Smart Citation

The role of endothelial cilia in postembryonic vascular development

Elworthy

Savage

Wilkinson

et al. 2019

Developmental Dynamics

View full text Add to dashboard Cite

Abstrct 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 hemorrhage 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 postembryonic, 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 wild‐type ift54 flanked by recombinase sites, then used a Tg(kdrl:cre)s898 transgene to induce endothelial‐specific inactivation of ift54 at postembryonic 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, or formation of vessels after tail fin resection in adult animals. Conclusions Endothelial cilia are not essential for development and remodeling of the vasculature in juvenile and adult Zebrafish when inactivated after embryogenesis.

show abstract

Section: Discussionmentioning

confidence: 97%

Section: Discussionmentioning

confidence: 99%

The role of endothelial cilia in postembryonic vascular development

Elworthy

Savage

Wilkinson

et al. 2019

Developmental Dynamics

View full text Add to dashboard Cite

show abstract

“…Interestingly, due to the presence of both multiciliated cells and mucus-secreting goblet cells, the embryo's skin, to some extent, resembles the epithelium lining the mammalian respiratory tracts [58]. Equally importantly, in the case of both models, the microscopic methods are well developed, and gene manipulation and genome editing are possible [44,[59][60][61][62][63][64][65].…”

Section: Vertebrate Modelsmentioning

confidence: 99%

Rare Human Diseases: Model Organisms in Deciphering the Molecular Basis of Primary Ciliary Dyskinesia

Poprzeczko

Bicka

Farahat

et al. 2019

Cells

View full text Add to dashboard Cite

Primary ciliary dyskinesia (PCD) is a recessive heterogeneous disorder of motile cilia, affecting one per 15,000-30,000 individuals; however, the frequency of this disorder is likely underestimated. Even though more than 40 genes are currently associated with PCD, in the case of approximately 30% of patients, the genetic cause of the manifested PCD symptoms remains unknown. Because motile cilia are highly evolutionarily conserved organelles at both the proteomic and ultrastructural levels, analyses in the unicellular and multicellular model organisms can help not only to identify new proteins essential for cilia motility (and thus identify new putative PCD-causative genes), but also to elucidate the function of the proteins encoded by known PCD-causative genes. Consequently, studies involving model organisms can help us to understand the molecular mechanism(s) behind the phenotypic changes observed in the motile cilia of PCD affected patients. Here, we summarize the current state of the art in the genetics and biology of PCD and emphasize the impact of the studies conducted using model organisms on existing knowledge.

show abstract

“…Transmission electron microscopy of kinocilia from zebrafish inner ear hair cells reveals a 9+2 axoneme (9 doublet microtubules surrounding a central pair of single microtubules) (Leventea et al, 2016), as described for the supernumerary ear kinocilia in homozygous mib ta52b mutants (Yu et al, 2011). Although based on very few examples in zebrafish, these studies corroborate findings for utricular and lateral line kinocilia in another teleost fish, the burbot (Lota lota) (Flock, 1964;Flock and Duvall, 1965;Flock and Wersäll, 1962).…”

Section: Ultrastructure Of Cilia In the Zebrafish Earmentioning

confidence: 99%

Cilia in the developing zebrafish ear

Whitfield

2019

Phil. Trans. R. Soc. B

View full text Add to dashboard Cite

The inner ear, which mediates the senses of hearing and balance, derives from a simple ectodermal vesicle in the vertebrate embryo. In the zebrafish, the otic placode and vesicle express a whole suite of genes required for ciliogenesis and ciliary motility. Every cell of the otic epithelium is ciliated at early stages; at least three different ciliary subtypes can be distinguished on the basis of length, motility, genetic requirements and function. In the early otic vesicle, most cilia are short and immotile. Long, immotile kinocilia on the first sensory hair cells tether the otoliths, biomineralized aggregates of calcium carbonate and protein. Small numbers of motile cilia at the poles of the otic vesicle contribute to the accuracy of otolith tethering, but neither the presence of cilia nor ciliary motility is absolutely required for this process. Instead, otolith tethering is dependent on the presence of hair cells and the function of the glycoprotein Otogelin. Otic cilia or ciliary proteins also mediate sensitivity to ototoxins and coordinate responses to extracellular signals. Other studies are beginning to unravel the role of ciliary proteins in cellular compartments other than the kinocilium, where they are important for the integrity and survival of the sensory hair cell. This article is part of the Theo Murphy meeting issue ‘Unity and diversity of cilia in locomotion and transport’.

show abstract

Analysis of cilia structure and function in zebrafish

Cited by 28 publications

References 184 publications

The role of endothelial cilia in postembryonic vascular development

The role of endothelial cilia in postembryonic vascular development

Rare Human Diseases: Model Organisms in Deciphering the Molecular Basis of Primary Ciliary Dyskinesia

Cilia in the developing zebrafish ear

Contact Info

Product

Resources

About