biostudies-literatureZur Lage PMedical Research CouncilWellcome TrustBiotechnology and Biological Sciences Research Council24https://www.ebi.ac.uk/biostudies/studies/S-EPMC6367277biostudies-literatureUnknown10The motile cilium/flagellum is an ancient eukaryotic organelle. The molecular machinery of ciliary motility comprises a variety of cilium-specific dynein motor complexes along with other complexes that regulate their activity. Assembling the motors requires the function of dedicated "assembly factors" and transport processes. In humans, mutation of any one of at least 40 different genes encoding components of the motility apparatus causes Primary Ciliary Dyskinesia (PCD), a disease of defective ciliary motility. Recently, <i>Drosophila</i> has emerged as a model for motile cilia biology and motile ciliopathies. This is somewhat surprising as most <i>Drosophila</i> cells lack cilia, and motile cilia are confined to just two specialized cell types: the sperm flagellum with a 9+2 axoneme and Frontiers in geneticsSurvey of the Ciliary Motility Machinery of <i>Drosophila</i> Sperm and Ciliated Mechanosensory Neurons Reveals Unexpected Cell-Type Specific Variations: A Model for Motile Ciliopathies.PMC6367277MR/K018558/1BB/M008533/1BB/S000801/1Zur Lage PNewton FGJarman APfalseSurvey of the Ciliary Motility Machinery of <i>Drosophila</i> Sperm and Ciliated Mechanosensory Neurons Reveals Unexpected Cell-Type Specific Variations: A Model for Motile Ciliopathies.The motile cilium/flagellum is an ancient eukaryotic organelle. The molecular machinery of ciliary motility comprises a variety of cilium-specific dynein motor complexes along with other complexes that regulate their activity. Assembling the motors requires the function of dedicated "assembly factors" and transport processes. In humans, mutation of any one of at least 40 different genes encoding components of the motility apparatus causes Primary Ciliary Dyskinesia (PCD), a disease of defective ciliary motility. Recently, <i>Drosophila</i> has emerged as a model for motile cilia biology and motile ciliopathies. This is somewhat surprising as most <i>Drosophila</i> cells lack cilia, and motile cilia are confined to just two specialized cell types: the sperm flagellum with a 9+2 axoneme and 2019-01-01T00:00:00Z20192026-04-07T19:44:59.497Z2019-03-26T22:55:36ZS-EPMC63672773077464810.3389/fgene.2019.00024