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Weidemann, M.* ; Schuster-Gossler, K.* ; Stauber, M.* ; Wrede, C.* ; Hegermann, J.* ; Ott, T.* ; Boldt, K.* ; Beyer, T.* ; Serth, K.* ; Kremmer, E. ; Blum, M.* ; Ueffing, M.* ; Gossler, A.*

CFAP157 is a murine downstream effector of FOXJ1 that is specifically required for flagellum morphogenesis and sperm motility.

Development 143, 4736-4748 (2016)
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Open Access Green möglich sobald Postprint bei der ZB eingereicht worden ist.
Motile cilia move extracellular fluids or mediate cellular motility. 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.
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Publikationstyp Artikel: Journalartikel
Dokumenttyp Wissenschaftlicher Artikel
Schlagwörter Axoneme ; Cfap157 ; Ciliogenesis ; Male Infertility ; Spermiogenesis; Left-right Asymmetry; Primary Ciliary Dyskinesia; Fibrous Sheath Formation; Symmetry Breakage; Gene-expression; Ependymal Cells; Male-mice; Protein; Ciliogenesis; Cap350
Sprache englisch
Veröffentlichungsjahr 2016
HGF-Berichtsjahr 2016
ISSN (print) / ISBN 0950-1991
e-ISSN 1477-9129
Zeitschrift Development / Company of Biologists
Quellenangaben Band: 143, Heft: 24, Seiten: 4736-4748 Artikelnummer: , Supplement: ,
Verlag Company of Biologists
Verlagsort Cambridge
Begutachtungsstatus Peer reviewed
Institut(e) Institute of Molecular Immunology (IMI)
POF Topic(s) 30504 - Mechanisms of Genetic and Environmental Influences on Health and Disease
Forschungsfeld(er) Immune Response and Infection
PSP-Element(e) G-501793-001
DOI 10.1242/dev.139626
WOS ID WOS:000393454600017
Scopus ID 85006164643
Erfassungsdatum 2016-12-31
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