Journal
DEVELOPMENTAL DYNAMICS
Volume 239, Issue 8, Pages 2190-2197Publisher
WILEY
DOI: 10.1002/dvdy.22355
Keywords
cilia; asymmetry; intraflagellar transport; axonemal dynein; situs inversus
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Funding
- NIGMS
- NIGMS [044228]
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In the alga Chlamydomonas reinhardtii, Oda16 functions during ciliary assembly as an adaptor for intra-flagellar transport of outer arm dynein. Oda16 orthologs only occur in genomes of organisms that use motile cilia; however, such cilia play multiple roles during vertebrate development and the contribution of Oda16 to their assembly remains unexplored. We demonstrate that the zebrafish Oda16 ortholog (Wdr69) is expressed in organs with motile cilia and retains a role in dynein assembly. Antisense morpholino knockdown of Wdr69 disrupts ciliary motility and results in multiple phenotypes associated with vertebrate ciliopathies. Affected cilia included those in Kupffer's vesicle, where Wdr69 plays a role in generation of asymmetric fluid flow and establishment of organ laterality, and otic vesicles, where Wdr69 is needed to develop normal numbers of otoliths. Analysis of cilium ultrastructure revealed loss of outer dynein arms in morphant embryos. These results support a remarkable level of functional conservation for Oda16/Wdr69. Developmental Dynamics 239:2190-2197, 2010. (C) 2010 Wiley-Liss, Inc.
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