1. hemingwayis required for sperm flagella assembly and ciliary motility inDrosophila
- Author
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Elisabeth Cortier, Fabien Soulavie, Anne Laurençon, Martin C. Göpfert, David Piepenbrock, Jennifer Vieillard, Joëlle Thomas, Jean-Luc Duteyrat, Bénédicte Durand, Inflammasome NLRP3 – NLRP3 Inflammasome, Centre International de Recherche en Infectiologie (CIRI), École normale supérieure de Lyon (ENS de Lyon)-Université Claude Bernard Lyon 1 (UCBL), Université de Lyon-Université de Lyon-Université Jean Monnet - Saint-Étienne (UJM)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS)-École normale supérieure de Lyon (ENS de Lyon)-Université Claude Bernard Lyon 1 (UCBL), Université de Lyon-Université de Lyon-Université Jean Monnet - Saint-Étienne (UJM)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS), Centre International de Recherche en Infectiologie - UMR (CIRI), École normale supérieure - Lyon (ENS Lyon)-Université Claude Bernard Lyon 1 (UCBL), Université de Lyon-Université de Lyon-Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS)-École normale supérieure - Lyon (ENS Lyon)-Université Claude Bernard Lyon 1 (UCBL), and Université de Lyon-Université de Lyon-Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS)
- Subjects
Male ,Axoneme ,[SDV]Life Sciences [q-bio] ,Molecular Sequence Data ,Motility ,Regulatory Factor X Transcription Factors ,Biology ,Flagellum ,Animals, Genetically Modified ,Gene Knockout Techniques ,03 medical and health sciences ,0302 clinical medicine ,medicine ,Animals ,Drosophila Proteins ,Amino Acid Sequence ,Cilia ,Hearing Loss ,Promoter Regions, Genetic ,Spermatogenesis ,Molecular Biology ,Infertility, Male ,Cytoskeleton ,ComputingMilieux_MISCELLANEOUS ,030304 developmental biology ,Primary ciliary dyskinesia ,Genetics ,0303 health sciences ,Binding Sites ,Cilium ,fungi ,food and beverages ,Articles ,Cell Biology ,respiratory system ,medicine.disease ,Protein Structure, Tertiary ,Cell biology ,DNA-Binding Proteins ,Drosophila melanogaster ,Sperm Tail ,Motile cilium ,Ciliary Motility Disorders ,Sequence Alignment ,030217 neurology & neurosurgery ,Drosophila Protein ,Transcription Factors - Abstract
Motile cilia play important functions in many organisms. In Drosophila, hemingway (hmw) encodes a novel protein conserved in species with motile cilia. hmw-mutant flies are hearing impaired and male sterile. HMW is required for acquisition of motile properties of cilia in the fly sound receiver and stability of the sperm axoneme., Cilia play major functions in physiology and development, and ciliary dysfunctions are responsible for several diseases in humans called ciliopathies. Cilia motility is required for cell and fluid propulsion in organisms. In humans, cilia motility deficiencies lead to primary ciliary dyskinesia, with upper-airways recurrent infections, left–right asymmetry perturbations, and fertility defects. In Drosophila, we identified hemingway (hmw) as a novel component required for motile cilia function. hmw encodes a 604–amino acid protein characterized by a highly conserved coiled-coil domain also found in the human orthologue, KIAA1430. We show that HMW is conserved in species with motile cilia and that, in Drosophila, hmw is expressed in ciliated sensory neurons and spermatozoa. We created hmw-knockout flies and found that they are hearing impaired and male sterile. hmw is implicated in the motility of ciliated auditory sensory neurons and, in the testis, is required for elongation and maintenance of sperm flagella. Because HMW is absent from mature flagella, we propose that HMW is not a structural component of the motile axoneme but is required for proper acquisition of motile properties. This identifies HMW as a novel, evolutionarily conserved component necessary for motile cilium function and flagella assembly.
- Published
- 2014