CFAP43 modulates ciliary beating in mouse and Xenopus
| Autor: | Michaela Mai, Ev Rachev, Lena Tveriakhina, Martin Blum, Franziska Fuhl, Tim Ott, Achim Gossler, Jan Hegermann, Karsten Boldt, Karin Schuster-Gossler, Anja Beckers, Elisabeth Kremmer, Marius Ueffing |
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| Rok vydání: | 2019 |
| Předmět: |
Male
Mucociliary clearance Xenopus Biology Xenopus Proteins 03 medical and health sciences Mice Xenopus laevis 0302 clinical medicine Ciliogenesis Animals Cilia Spermatogenesis Molecular Biology Infertility Male 030304 developmental biology Mice Knockout 0303 health sciences Epidermis (botany) Cilium Morphant Forkhead Transcription Factors Cell Biology respiratory system biology.organism_classification Mucus Spermatozoa Cfap43 Hydrocephalus Mucociliary Clearance Male Infertility Xenopus Laevis Mouse Cell biology Trachea Cytoskeletal Proteins Epidermal Cells Sperm Tail Motile cilium 030217 neurology & neurosurgery Developmental Biology |
| Zdroj: | Dev. Biol. 459, 109-125 (2020) |
| ISSN: | 1095-564X |
| Popis: | Malfunctions of motile cilia cause a variety of developmental defects and diseases in humans and animal model organisms. Defects include impaired mucociliary clearance of the airways, sperm immotility, hydrocephalus and organ laterality. Here, we characterize the evolutionary conserved Cfap43 gene by loss-of-function experiments in the mouse and the frog Xenopus laevis. Cfap43 is expressed in tissues carrying motile cilia and acts as a target gene of the transcription factor FOXJ1, which is essential for the induction of motile ciliogenesis. We show that CFAP43, a protein of unknown biochemical function, localizes to the ciliary axoneme. CFAP43 is involved in the regulation of the beating frequency of tracheal cilia and loss of CFAP43 causes severe mucus accumulation in the nasal cavity. Likewise, morphant and crispant frog embryos revealed impaired function of motile cilia of the larval epidermis, a model for airway mucociliary epithelia. CFAP43 participates in the formation of flagellar axonemes during spermatogenesis as mice mutant for Cfap43 display male infertility, consistent with observations in male sterile patients. In addition, mice mutant for Cfap43 display early onset hydrocephalus. Together, these results confirm the role of CFAP43 in the male reproductive tract and pinpoint additional functions in airway epithelia mucus clearance and brain development. |
| Databáze: | OpenAIRE |
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