BLD10/CEP135 Is a Microtubule-Associated Protein that Controls the Formation of the Flagellum Central Microtubule Pair

Carvalho-Santos, ZitaMachado, PedroAlvarez-Martins, InêsGouveia, Susana M.Jana, Swadhin C.Duarte, PauloAmado, TiagoBranco, PedroFreitas, Micael C.Silva, Sara T.N.Antony, ClaudeBandeiras, Tiago M.Bettencourt-Dias, Mónica2018-02-092018-02-092012-08-14Zita Carvalho-Santos, Pedro Machado, Inês Alvarez-Martins, Susana M. Gouveia, Swadhin C. Jana, Paulo Duarte, Tiago Amado, Pedro Branco, Micael C. Freitas, Sara T.N. Silva, Claude Antony, Tiago M. Bandeiras, Mónica Bettencourt-Dias, BLD10/CEP135 Is a Microtubule-Associated Protein that Controls the Formation of the Flagellum Central Microtubule Pair, Developmental Cell, Volume 23, Issue 2, 2012, Pages 412-424, ISSN 1534-5807, https://doi.org/10.1016/j.devcel.2012.06.001. (http://www.sciencedirect.com/science/article/pii/S1534580712002511)http://hdl.handle.net/10400.7/835The deposited article is a post-print version and has been submitted to peer review.The deposited article is a pre-print versionThis deposit is composed by the main article and the supplementary materials are present in the publisher's page in the following link: https://ars.els-cdn.com/content/image/1-s2.0-S1534580712002511-mmc1.pdfCilia and flagella are involved in a variety of processes and human diseases, including ciliopathies and sterility. Their motility is often controlled by a central microtubule (MT) pair localized within the ciliary MT-based skeleton, the axoneme. We characterized the formation of the motility apparatus in detail in Drosophila spermatogenesis. We show that assembly of the central MT pair starts prior to the meiotic divisions, with nucleation of a singlet MT within the basal body of a small cilium, and that the second MT of the pair only assembles much later, upon flagella formation. BLD10/CEP135, a conserved player in centriole and flagella biogenesis, can bind and stabilize MTs and is required for the early steps of central MT pair formation. This work describes a genetically tractable system to study motile cilia formation and provides an explanation for BLD10/CEP135's role in assembling highly stable MT-based structures, such as motile axonemes and centrioles.engAnimalsDrosophila ProteinsDrosophila melanogasterFlagellaMaleMicroscopy, Electron, TransmissionMicrotubule-Associated ProteinsMicrotubulesProtein BindingSpermatogenesisCiliaBLD10/CEP135 Is a Microtubule-Associated Protein that Controls the Formation of the Flagellum Central Microtubule Pairjournal article10.1016/j.devcel.2012.06.001