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Identification et caractérisation de nouvelles protéines de la zone de transition des cils et des flagelles

Abstract : Cilia and flagella are highly conserved organelles among eukaryotes species. They are composed of a microtubular cytoskeleton and play essential functions during development and in numerous physiological processes. As a result, in humans, cilia dysfunction leads to a wide range of pathologies, called ciliopathies.At the ciliary base, the transition zone (TZ), a complex structure, is required for proper cilia assembly and regulates the traffic of ciliary components in and out cilia. Defects in TZ proteins lead to severe ciliopathies. The TZ is composed of 3 protein complexes, MKS, NPHP et CEP290 that closely interact. Additional proteins, like CBY, conserved between mammals and Drosophila, have been described at the TZ but their precise role and relationships with the other TZ complexes are unknown. Two modes of cilia assembly have been described: compartmentalized and cytosolic ciliogenesis. Whereas the function of the TZ in compartmentalized ciliogenesis is well documented, its role in cytosolic ciliogenesis remains poorly characterized. During my PhD, I characterized new TZ proteins conserved in mammals and Drosophila and analyzed their function during cilia assembly in Drosophila. First, I performed a proteomic screen in murine IMCD3 cells and characterized the CBY module composed of CBY, FAM92A1 and DZIP1L. This complex is conserved in Drosophila and locates at the TZ. Moreover, I showed that this module is necessary for TZ assembly and centriolar docking to the plasma membrane and hence required for cilia and flagella assembly. In absence of these proteins, Drosophila show severe ciliogenesis defects both in sperm cells and in sensory neurons.In conclusion, this work brings new insights into the understanding of TZ assembly and of the mechanisms, that control ciliogenesis
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Jean-André Lapart. Identification et caractérisation de nouvelles protéines de la zone de transition des cils et des flagelles. Biologie cellulaire. Université de Lyon, 2017. Français. ⟨NNT : 2017LYSE1108⟩. ⟨tel-02003362⟩

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