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Their function is essential for embryonic development, adult tissue homeostasis and reproduction throughout vertebrates. FOXJ1 is a key transcription factor for the formation of motile cilia but its downstream genetic programme is only partially understood. Here, we characterise a novel FOXJ1 target, Cfap157, that is specifically expressed in motile ciliated tissues in mouse and Xenopus in a FOXJ1-dependent manner. CFAP157 protein localises to basal bodies and interacts with tubulin and the centrosomal protein CEP350. Cfap157 knockout mice appear normal but homozygous males are infertile. Spermatozoa display impaired motility and a novel phenotype: Cfap157-deficient sperm exhibit axonemal loops, supernumerary axonemal profiles with ectopic accessory structures, excess cytoplasm and clustered mitochondria in the midpiece regions, and defective axonemes along the flagella. Our study thus demonstrates an essential sperm-specific function for CFAP157 and suggests that this novel FOXJ1 effector is part of a mechanism that acts during spermiogenesis to suppress the formation of supernumerary axonemes and ensures a correct ultrastructure.<\/jats:p>","DOI":"10.1242\/dev.139626","type":"journal-article","created":{"date-parts":[[2017,1,11]],"date-time":"2017-01-11T18:48:46Z","timestamp":1484160526000},"page":"4736-4748","source":"Crossref","is-referenced-by-count":34,"title":["CFAP157 is a murine downstream effector of FOXJ1 that is specifically required for flagellum morphogenesis and sperm motility"],"prefix":"10.1242","volume":"143","author":[{"given":"Marina","family":"Weidemann","sequence":"first","affiliation":[{"name":"Institute for Molecular Biology, OE5250, Hannover Medical School 1 , Carl-Neuberg-Str. 1, Hannover 30625 , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Karin","family":"Schuster-Gossler","sequence":"additional","affiliation":[{"name":"Institute for Molecular Biology, OE5250, Hannover Medical School 1 , Carl-Neuberg-Str. 1, Hannover 30625 , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael","family":"Stauber","sequence":"additional","affiliation":[{"name":"Institute for Molecular Biology, OE5250, Hannover Medical School 1 , Carl-Neuberg-Str. 1, Hannover 30625 , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christoph","family":"Wrede","sequence":"additional","affiliation":[{"name":"Institute of Functional and Applied Anatomy, OE8840, Hannover Medical School 2 , Carl-Neuberg-Str. 1, Hannover 30625 , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jan","family":"Hegermann","sequence":"additional","affiliation":[{"name":"Institute of Functional and Applied Anatomy, OE8840, Hannover Medical School 2 , Carl-Neuberg-Str. 1, Hannover 30625 , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tim","family":"Ott","sequence":"additional","affiliation":[{"name":"Institute of Zoology, University of Hohenheim 3 , Garbenstra\u00dfe 30, Stuttgart 70593 , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Karsten","family":"Boldt","sequence":"additional","affiliation":[{"name":"Institute of Ophthalmic Research, Center for Ophthalmology, University of T\u00fcbingen 4 , R\u00f6ntgenweg 11, T\u00fcbingen 72076 , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tina","family":"Beyer","sequence":"additional","affiliation":[{"name":"Institute of Ophthalmic Research, Center for Ophthalmology, University of T\u00fcbingen 4 , R\u00f6ntgenweg 11, T\u00fcbingen 72076 , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Katrin","family":"Serth","sequence":"additional","affiliation":[{"name":"Institute for Molecular Biology, OE5250, Hannover Medical School 1 , Carl-Neuberg-Str. 1, Hannover 30625 , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elisabeth","family":"Kremmer","sequence":"additional","affiliation":[{"name":"Institute of Molecular Immunology, Helmholtz Zentrum M\u00fcnchen, German Research Center for Environmental Health (GmbH), Core Facility Monoclonal Antibodies 5 , Marchioninistr. 25, M\u00fcnchen 81377 , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Martin","family":"Blum","sequence":"additional","affiliation":[{"name":"Institute of Zoology, University of Hohenheim 3 , Garbenstra\u00dfe 30, Stuttgart 70593 , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marius","family":"Ueffing","sequence":"additional","affiliation":[{"name":"Institute of Ophthalmic Research, Center for Ophthalmology, University of T\u00fcbingen 4 , R\u00f6ntgenweg 11, T\u00fcbingen 72076 , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9103-9116","authenticated-orcid":false,"given":"Achim","family":"Gossler","sequence":"additional","affiliation":[{"name":"Institute for Molecular Biology, OE5250, Hannover Medical School 1 , Carl-Neuberg-Str. 1, Hannover 30625 , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","reference":[{"key":"2024061213155906600_DEV139626C1","first-page":"144","article-title":"Male and female infertility problems in the immotile-cilia syndrome","volume":"127","author":"Afzelius","year":"1983","journal-title":"Eur. 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