Primary cilia play critical roles in development and disease. Their assembly and disassembly are tightly coupled to cell cycle progression. Here, we present data identifying KIF14 as a regulator of cilia formation and Hedgehog (HH) signaling. We show that RNAi depletion of KIF14 specifically leads to defects in ciliogenesis and basal body (BB) biogenesis, as its absence hampers the efficiency of primary cilium formation and the dynamics of primary cilium elongation, and disrupts the localization of the distal appendage proteins SCLT1 and FBF1 and components of the IFT-B complex. We identify deregulated Aurora A activity as a mechanism contributing to the primary cilium and BB formation defects seen after KIF14 depletion. In addition, we show that primary cilia in KIF14-depleted cells are defective in response to HH pathway activation, independently of the effects of Aurora A. In sum, our data point to KIF14 as a critical node connecting cell cycle machinery, effective ciliogenesis, and HH signaling.

Central European Institute of Technology Brno Czech Republic

Department of Histology and Embryology Masaryk University Faculty of Medicine Brno Czech Republic

Laboratory of Hereditary Kidney Diseases Institut National de la Santé et de la Recherche Médicale UMR 1163 Paris University Imagine Institute Paris France

Paris Diderot University Paris France

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A protocol for generation and live-cell imaging analysis of primary cilia reporter cell lines

Primary Cilia Formation Does Not Rely on WNT/β-Catenin Signaling