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Rachev, E.* ; Schuster-Gossler, K.* ; Fuhl, F.* ; Ott, T.* ; Tveriakhina, L.* ; Beckers, A.* ; Hegermann, J.* ; Boldt, K.* ; Mai, M.* ; Kremmer, E. ; Ueffing, M.* ; Blum, M.* ; Gossler, A.*

CFAP43 modulates ciliary beating in mouse and Xenopus.

Dev. Biol. 459, 109-125 (2020)
Verlagsversion Postprint DOI
Open Access Green
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.
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Publikationstyp Artikel: Journalartikel
Dokumenttyp Wissenschaftlicher Artikel
Schlagwörter Cfap43 ; Ciliogenesis ; Cilia ; Hydrocephalus ; Mucociliary Clearance ; Male Infertility ; Xenopus Laevis ; Mouse; Ependymal Cells; Hydrocephalus; Ciliogenesis; Sperm; Expression; Flow
Sprache englisch
Veröffentlichungsjahr 2020
Prepublished im Jahr 2019
HGF-Berichtsjahr 2019
ISSN (print) / ISBN 0012-1606
e-ISSN 0012-1606
Zeitschrift Developmental Biology
Quellenangaben Band: 459, Heft: 2, Seiten: 109-125 Artikelnummer: , Supplement: ,
Verlag Elsevier
Verlagsort 525 B St, Ste 1900, San Diego, Ca 92101-4495 Usa
Begutachtungsstatus Peer reviewed
Institut(e) Institute of Molecular Immunology (IMI)
CF Monoclonal Antibodies (CF-MAB)
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
G-501760-001
Förderungen Tistou & Charlotte Kerstan Stiftung
Landesgraduiertenforderung Baden-Wurttemberg
DFG Cluster of Excellence "REBIRTH"
DFG grant
DOI 10.1016/j.ydbio.2019.12.010
WOS ID WOS:000582177200006
Scopus ID 85077383872
Erfassungsdatum 2020-01-14
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