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CFAP43 modulates ciliary beating in mouse and Xenopus
- Source :
- Dev. Biol. 459, 109-125 (2020)
- Publication Year :
- 2019
-
Abstract
- 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.
- Subjects :
- 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
Subjects
Details
- ISSN :
- 1095564X
- Volume :
- 459
- Issue :
- 2
- Database :
- OpenAIRE
- Journal :
- Developmental biology
- Accession number :
- edsair.doi.dedup.....a1c7a4c80346c32d1883971a2879b62d