Synergic multiciliary beating relies on cilia generatedfrom basal bodies, with which basal feet are regularlyassociated through molecular mechanisms that remainunknown. Here we show that the coordinated multiciliaryaction is disturbed in Odf2 mutant mice, resulting inprimary ciliary dyskinesia and a characteristic coughing/sneezing-like phenotype. Odf2-deficiency depleted basalfeet from basal bodies to perturb the planar cell polarity(PCP) of basal bodies, as shown by ultra-high voltageelectron microscopic tomography (UHVEMT) of wildand Odf2 mutant tracheas. The apical microtubular lattice,which is organized by the keystone positioning ofbasal feet/basal bodies, was lost in Odf2-mutant animals,irrespective of normal localization of Vangl1, the PCPcore protein. These findings demonstrate that Odf2 isrequired for the formation of basal feet. Odf2-based basalfeet play a critical role in the PCP-based arrangement ofthe microtubular lattice and basal bodies, therebyenabling coordinated multiciliary beating.
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