{"doi":"10.1091/mbc.e13-10-0616","title":"<i>hemingway</i>is required for sperm flagella assembly and ciliary motility in<i>Drosophila</i>","abstract":"<jats:p>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.</jats:p>","journal":"Molecular Biology of the Cell","year":2014,"id":686609,"datarank":0.49983067652628066,"base_score":3.332204510175204,"endowment":3.332204510175204,"self_citation_contribution":0.49983067652628066,"citation_network_contribution":0.0,"self_endowment_contribution":0.49983067652628066,"citer_contribution":0.0,"corpus_percentile":null,"corpus_rank":null,"citation_count":27,"citer_count":0,"citers_with_citation_signal":0,"citers_with_endowment":0,"datacite_reuse_total":0,"is_dataset":false,"is_dataset_confidence":null,"is_data_producer":false,"deposit_databanks":null,"is_oa":false,"file_count":0,"downloads":0,"has_version_chain":false,"published_date":null,"fair_score":null,"fair_percentile":null,"algorithm_id":"datarank_citation_only_1hop_v6","ranking_scope":"data_only","authors":[{"id":1793762,"name":"David Piepenbrock","orcid":null,"position":1,"is_corresponding":false},{"id":1610205,"name":"Joëlle Thomas","orcid":null,"position":2,"is_corresponding":false},{"id":1793763,"name":"Jennifer Vieillard","orcid":null,"position":3,"is_corresponding":false},{"id":1793764,"name":"Jean-Luc Duteyrat","orcid":null,"position":4,"is_corresponding":false},{"id":1759659,"name":"Elisabeth Cortier","orcid":null,"position":5,"is_corresponding":false},{"id":781700,"name":"Anne Laurençon","orcid":"0000-0003-1791-1882","position":6,"is_corresponding":false},{"id":1413422,"name":"Martin C. 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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.</jats:p>","is_dataset_classified":null,"base_score":3.332204510175204,"endowment":3.332204510175204,"datacite_reuse_total":0,"file_count":0,"downloads":0,"views":0,"has_version_chain":false,"is_dataset":false,"is_oa":false,"pmid":"24554765","pmcid":"PMC3982993","openalex_id":"https://openalex.org/W2018594621","authors":[],"funders":[{"funder_name":"Deutsche Forschungsgemeinschaft","grant_id":"154113120/SFB 889","title":"Cellular Mechanisms of Sensory Processing"}],"total_grants":1,"fwci":1.2794,"citation_percentile":0.80926267,"influential_citations":0,"citation_trend":[{"year":2015,"count":5},{"year":2017,"count":1},{"year":2018,"count":2},{"year":2019,"count":3},{"year":2020,"count":3},{"year":2021,"count":2},{"year":2022,"count":2},{"year":2023,"count":4},{"year":2025,"count":4},{"year":2026,"count":1}],"oa_status":"closed","license":"CC BY NC SA","oa_locations":[{"url":"https://doi.org/10.1091/mbc.e13-10-0616","host_type":"journal"},{"url":"https://pubmed.ncbi.nlm.nih.gov/24554765","host_type":"repository"},{"url":"https://www.ncbi.nlm.nih.gov/pmc/articles/3982993","host_type":"repository"},{"url":"https://hal.science/hal-03080657","host_type":"repository"},{"url":"https://europepmc.org/articles/PMC3982993","host_type":"Europe_PMC"},{"url":"https://europepmc.org/articles/PMC3982993?pdf=render","host_type":"Europe_PMC"},{"url":"http://dx.doi.org/10.1091/mbc.E13-10-0616","host_type":""},{"url":"https://dx.doi.org/10.1091/mbc.e13-10-0616","host_type":""},{"url":"https://resolver.sub.uni-goettingen.de/purl?gro-2/33684","host_type":""},{"url":"https://hal.science/hal-03080657v1","host_type":""},{"url":"https://hal.science/hal-03080657v1/document","host_type":""},{"url":"https://doi.org/https://doi.org/10.1091/mbc.E13-10-0616","host_type":""}],"fields_of_study":["Genetic and Kidney Cyst Diseases","Protist diversity and phylogeny","Microtubule and mitosis dynamics","0301 basic medicine","03 medical and health sciences","0303 health sciences"],"mesh_terms":["Regulatory Factor X Transcription Factors","Amino Acid Sequence","Animals","Binding Sites","Cilia","Ciliary Motility Disorders","DNA-Binding Proteins","Drosophila melanogaster","Infertility, Male","Male","Molecular Sequence Data","Promoter Regions, Genetic","Sperm Tail","Spermatogenesis","Transcription Factors","Sequence Alignment","Protein Structure, Tertiary","Drosophila Proteins","Animals, Genetically Modified","Hearing Loss","Gene Knockout Techniques"],"keywords":["Cilium","Axoneme","Flagellum","Motile cilium","Biology","Intraflagellar transport","Motility","Cell biology","Dynein","Sperm motility","Primary ciliary dyskinesia","Sperm","Drosophila melanogaster","Genetics","Microtubule","Gene","Male","570","[SDV]Life Sciences [q-bio]","Molecular Sequence Data","Regulatory Factor X Transcription Factors","Animals, Genetically Modified","Gene Knockout Techniques","Animals","Drosophila Proteins","Amino Acid Sequence","Cilia","Hearing Loss","Promoter Regions, Genetic","Spermatogenesis","Infertility, Male","Binding Sites","Articles","Protein Structure, Tertiary","[SDV] Life Sciences [q-bio]","DNA-Binding Proteins","Sperm Tail","Sequence Alignment","Ciliary Motility Disorders"],"sdg_mappings":[{"sdg_number":0,"sdg_label":"Life below water"}],"linked_datasets":[],"clinical_trials":[],"software_tools":[],"database_accessions":[],"source":"live","citation_network_status":"fetched"},"created_at":"2026-08-18T19:58:19.075027Z","pmid":null,"pmcid":null,"fwci":null,"citation_percentile":null,"influential_citations":0,"oa_status":null,"license":null,"views":0,"total_file_size_bytes":0,"version_count":0,"fair_f":null,"fair_a":null,"fair_i":null,"fair_r":null,"fair_zscore":null,"fair_rationale":null,"fair_model":null,"fair_agent_version":null,"fair_fulltext_source":null,"fair_has_llm":null,"fair_computed_at":null,"clinical_trials":[],"software_tools":[],"db_accessions":[],"linked_datasets":[],"topics":[]}