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1. The highly conserved FOXJ1 target CFAP161 is dispensable for motile ciliary function in mouse and Xenopus

Author

Anja Beckers, Franziska Fuhl, Tim Ott, Karsten Boldt, Magdalena Maria Brislinger, Peter Walentek, Karin Schuster-Gossler, Jan Hegermann, Leonie Alten, Elisabeth Kremmer, Adina Przykopanski, Katrin Serth, Marius Ueffing, Martin Blum, and Achim Gossler

Subjects
Medicine, Science
Abstract

Abstract Cilia are protrusions of the cell surface and composed of hundreds of proteins many of which are evolutionary and functionally well conserved. In cells assembling motile cilia the expression of numerous ciliary components is under the control of the transcription factor FOXJ1. Here, we analyse the evolutionary conserved FOXJ1 target CFAP161 in Xenopus and mouse. In both species Cfap161 expression correlates with the presence of motile cilia and depends on FOXJ1. Tagged CFAP161 localises to the basal bodies of multiciliated cells of the Xenopus larval epidermis, and in mice CFAP161 protein localises to the axoneme. Surprisingly, disruption of the Cfap161 gene in both species did not lead to motile cilia-related phenotypes, which contrasts with the conserved expression in cells carrying motile cilia and high sequence conservation. In mice mutation of Cfap161 stabilised the mutant mRNA making genetic compensation triggered by mRNA decay unlikely. However, genes related to microtubules and cilia, microtubule motor activity and inner dyneins were dysregulated, which might buffer the Cfap161 mutation.

Published
2021
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2. The highly conserved FOXJ1 target CFAP161 is dispensable for motile ciliary function in mouse and Xenopus

Author

Leonie Alten, Jan Hegermann, Peter Walentek, Katrin Serth, Tim Ott, Marius Ueffing, Franziska Fuhl, Magdalena Brislinger, Martin Blum, Adina Przykopanski, Elisabeth Kremmer, Karsten Boldt, Anja Beckers, Achim Gossler, and Karin Schuster-Gossler

Subjects
Axoneme, Male, Cell biology, Science, Dynein, Xenopus, Biology, Xenopus Proteins, medicine.disease_cause, Microtubules, Article, Mice, Xenopus laevis, Microtubule, medicine, Basal body, Animals, Cilia, Mutation, Multidisciplinary, Ciliogenesis, Cilium, Model vertebrates, Forkhead Transcription Factors, biology.organism_classification, Basal Bodies, Epidermal Cells, Motile cilium, Medicine, Female, Epidermis
Abstract

Cilia are protrusions of the cell surface and composed of hundreds of proteins many of which are evolutionary and functionally well conserved. In cells assembling motile cilia the expression of numerous ciliary components is under the control of the transcription factor FOXJ1. Here, we analyse the evolutionary conserved FOXJ1 target CFAP161 in Xenopus and mouse. In both species Cfap161 expression correlates with the presence of motile cilia and depends on FOXJ1. Tagged CFAP161 localises to the basal bodies of multiciliated cells of the Xenopus larval epidermis, and in mice CFAP161 protein localises to the axoneme. Surprisingly, disruption of the Cfap161 gene in both species did not lead to motile cilia-related phenotypes, which contrasts with the conserved expression in cells carrying motile cilia and high sequence conservation. In mice mutation of Cfap161 stabilised the mutant mRNA making genetic compensation triggered by mRNA decay unlikely. However, genes related to microtubules and cilia, microtubule motor activity and inner dyneins were dysregulated, which might buffer the Cfap161 mutation.

Published
2021

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