Author: ["Antonia Borovina","Simone Superina","Daniel Voskas","Brian Ciruna"]
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Abstract
Several recent studies have proposed that the planar cell polarity pathway (PCP) regulates cilia formation. Zebrafish embryos lacking the core PCP component Vangl2 do not have defects in ciliogenesis, but instead show impairment of primary cilia position and orientation. Cilia are microtubule-based organelles that project into the extracellular space, function in the perception and integration of environmental cues1, and regulate Hedgehog signal transduction2. The emergent association of ciliary defects with diverse and pleiotropic human disorders3,4 has fuelled investigations into the molecular genetic regulation of ciliogenesis. Although recent studies implicate planar cell polarity (PCP) in cilia formation, this conclusion is based on analyses of proteins that are not specific to, or downstream effectors of PCP signal transduction5,6,7,8,9,10. Here we characterize zebrafish embryos devoid of all Vangl2 function11, a core and specific component of the PCP signalling pathway. Using Arl13b–GFP as a live marker of the ciliary axoneme, we demonstrate that Vangl2 is not required for ciliogenesis. Instead, Vangl2 controls the posterior tilting of primary motile cilia lining the neurocoel, Kupffer's vesicle and pronephric duct. Furthermore, we show that Vangl2 is required for asymmetric localization of cilia to the posterior apical membrane of neuroepithelial cells. Our results indicate a broad and essential role for PCP in the asymmetric localization and orientation of motile primary cilia, establishing directional fluid flow implicated in normal embryonic development and disease.Cite this article
Borovina, A., Superina, S., Voskas, D. et al. Vangl2 directs the posterior tilting and asymmetric localization of motile primary cilia. Nat Cell Biol 12, 407–412 (2010). https://doi.org/10.1038/ncb2042 