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Dzip1 and Fam92 form a ciliary transition zone complex with cell type specific roles in Drosophila

Abstract : Cilia and flagella are conserved eukaryotic organelles essential for cellular signaling and motility. Cilia dysfunctions cause life-threatening ciliopathies, many of which are due to defects in the transition zone (TZ), a complex structure of the ciliary base. Therefore, understanding TZ assembly, which relies on ordered interactions of multiprotein modules, is of critical importance. Here, we show that Drosophila Dzip1 and Fam92 form a functional module which constrains the conserved core TZ protein, Cep290, to the ciliary base. We identify cell type specific roles of this functional module in two different tissues. While it is required for TZ assembly in all Drosophila ciliated cells, it also regulates basal-body growth and docking to the plasma membrane during spermatogenesis. We therefore demonstrate a novel regulatory role for Dzip1 and Fam92 in mediating membrane/basal-body interactions and show that these interactions exhibit cell type specific functions in basal-body maturation and TZ organization.
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https://hal.umontpellier.fr/hal-02408620
Contributeur : Alain Mangé <>
Soumis le : vendredi 13 décembre 2019 - 09:56:32
Dernière modification le : vendredi 15 mai 2020 - 12:22:10
Archivage à long terme le : : samedi 14 mars 2020 - 13:51:28

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10.7554_elife.49307.pdf
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Jean-André Lapart, Marco Gottardo, Elisabeth Cortier, Jean-Luc Duteyrat, Céline Augière, et al.. Dzip1 and Fam92 form a ciliary transition zone complex with cell type specific roles in Drosophila. eLife, eLife Sciences Publication, 2019, 8, ⟨10.7554/eLife.49307⟩. ⟨hal-02408620⟩

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