Centrin2 regulates CP110 removal in primary cilium formation.
Bottom Line: We disrupted CETN2 in human retinal pigmented epithelial cells, and despite having intact centrioles, they were unable to make cilia upon serum starvation, showing abnormal localization of distal appendage proteins and failing to remove the ciliation inhibitor CP110.Knockdown of CP110 rescued ciliation in CETN2-deficient cells.Thus, centrin2 regulates primary ciliogenesis through controlling CP110 levels.
Affiliation: Centre for Chromosome Biology, School of Natural Sciences, National University of Ireland, Galway, Galway, Ireland.Show MeSH
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Mentions: TEM confirmed the integrity of the centrioles in centrin2-deficient cells (Fig. 4 a). This analysis also demonstrated that centrioles docked successfully with ciliary vesicles, a key early step in ciliogenesis that is directed by Cep164 (Fig. 4 a; Sorokin, 1962; Schmidt et al., 2012; Tanos et al., 2013). Despite the abnormal Cep164 distribution that we saw in centrin-deficient cells, this observation indicates that the distal appendages are capable of allowing docking and that the defect in ciliation lies at a later stage in the process.
Affiliation: Centre for Chromosome Biology, School of Natural Sciences, National University of Ireland, Galway, Galway, Ireland.