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

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 anal...

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Autores principales:	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, Achim Gossler
Formato:	article
Lenguaje:	EN
Publicado:	Nature Portfolio 2021
Materias:	Medicine R Science Q
Acceso en línea:	https://doaj.org/article/4e99c332b9ed4435a10fd923ac28beb1
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spelling	oai:doaj.org-article:4e99c332b9ed4435a10fd923ac28beb12021-12-02T17:12:21ZThe highly conserved FOXJ1 target CFAP161 is dispensable for motile ciliary function in mouse and Xenopus10.1038/s41598-021-92495-32045-2322https://doaj.org/article/4e99c332b9ed4435a10fd923ac28beb12021-06-01T00:00:00Zhttps://doi.org/10.1038/s41598-021-92495-3https://doaj.org/toc/2045-2322Abstract 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.Anja BeckersFranziska FuhlTim OttKarsten BoldtMagdalena Maria BrislingerPeter WalentekKarin Schuster-GosslerJan HegermannLeonie AltenElisabeth KremmerAdina PrzykopanskiKatrin SerthMarius UeffingMartin BlumAchim GosslerNature PortfolioarticleMedicineRScienceQENScientific Reports, Vol 11, Iss 1, Pp 1-14 (2021)
institution	DOAJ
collection	DOAJ
language	EN
topic	Medicine R Science Q
spellingShingle	Medicine R Science Q 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 Achim Gossler The highly conserved FOXJ1 target CFAP161 is dispensable for motile ciliary function in mouse and Xenopus
description	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.
format	article
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 Achim Gossler
author_facet	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 Achim Gossler
author_sort	Anja Beckers
title	The highly conserved FOXJ1 target CFAP161 is dispensable for motile ciliary function in mouse and Xenopus
title_short	The highly conserved FOXJ1 target CFAP161 is dispensable for motile ciliary function in mouse and Xenopus
title_full	The highly conserved FOXJ1 target CFAP161 is dispensable for motile ciliary function in mouse and Xenopus
title_fullStr	The highly conserved FOXJ1 target CFAP161 is dispensable for motile ciliary function in mouse and Xenopus
title_full_unstemmed	The highly conserved FOXJ1 target CFAP161 is dispensable for motile ciliary function in mouse and Xenopus
title_sort	highly conserved foxj1 target cfap161 is dispensable for motile ciliary function in mouse and xenopus
publisher	Nature Portfolio
publishDate	2021
url	https://doaj.org/article/4e99c332b9ed4435a10fd923ac28beb1
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The highly conserved FOXJ1 target CFAP161 is dispensable for motile ciliary function in mouse and Xenopus

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