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CFAP157 is a murine downstream effector of FOXJ1 that is specifically required for flagellum morphogenesis and sperm motility.
Development 143, 4736-4748 (2016)
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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Publication type
Article: Journal article
Document type
Scientific Article
Keywords
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
Language
english
Publication Year
2016
HGF-reported in Year
2016
ISSN (print) / ISBN
0950-1991
e-ISSN
1477-9129
Quellenangaben
Volume: 143,
Issue: 24,
Pages: 4736-4748
Publisher
Company of Biologists
Publishing Place
Cambridge
Reviewing status
Peer reviewed
Institute(s)
Institute of Molecular Immunology (IMI)
POF-Topic(s)
30504 - Mechanisms of Genetic and Environmental Influences on Health and Disease
Research field(s)
Immune Response and Infection
PSP Element(s)
G-501793-001
WOS ID
WOS:000393454600017
Scopus ID
85006164643
Erfassungsdatum
2016-12-31