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

摘要: Motile cilia move extracellular fluids or mediate cellular motility. Their function is essential for embryonic development, adult tissue homeostasis and reproduction throughout vertebrates. FOXJ1 a key transcription factor the formation of motile but its downstream genetic programme only partially understood. Here, we characterise novel target, Cfap157, that specifically expressed in ciliated tissues mouse Xenopus FOXJ1-dependent manner. CFAP157 protein localises to basal bodies interacts with tubulin centrosomal CEP350. Cfap157 knockout mice appear normal homozygous males are infertile. Spermatozoa display impaired motility phenotype: Cfap157-deficient sperm exhibit axonemal loops, supernumerary profiles ectopic accessory structures, excess cytoplasm clustered mitochondria midpiece regions, defective axonemes along flagella. Our study thus demonstrates an sperm-specific suggests this effector part mechanism acts during spermiogenesis suppress ensures correct ultrastructure.