In the nervous system, cilia dysfunction perturbs the circulationof the cerebrospinal fluid, thus affecting neurogenesis and brainhomeostasis. A role for planar cell polarity (PCP) signaling in the orientation of cilia (rotational polarity) and ciliogenesis is established. However, whether and how PCP regulates cilia positioning in the apical domain (translational polarity) in radial progenitors and ependymal cells remain unclear. By analysis of a large panel of mutant mice, we show that two PCP signals are operating in ciliated cells. The first one, controlled by Celsr2, Celsr3, Fzd3 andVangl2, organize multicilia in individual cells (single cell polarity), whereas the second, governed by Celsr1, Fzd3 and Vangl2, coordinate polarity between cells in both radial progenitors and ependymal cells (tissue polarity). Loss of each of these signals is associated with specific defects in the cytoskeleton. Our data reveal unreported functions of PCP and provide an integrated view of planar polarization of the brain ciliated cells.
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Bibliographic reference
Boutin, Camille ; Labedan, Paul ; Dimidschstein, Jordane ; Richard, Fabrice ; Cremer, Harold ; et. al. A dual role for planar cell polarity genes in ciliated cells. In: Proceedings of the National academy of sciences of the United States of America, Vol. 111, no. 30, p. E3129-E3138 (2014)