The evolutionary conserved FOXJ1 target gene Fam183b is essential for motile cilia in Xenopus but dispensable for ciliary function in mice

Abstract The transcription factor FOXJ1 is essential for the formation of motile cilia throughout the animal kingdom. Target genes therefore likely constitute an important part of the motile cilia program. Here, we report on the analysis of one of these targets, Fam183b, in Xenopus and mice. Fam183b...

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Autores principales:	Anja Beckers, Tim Ott, Karin Schuster-Gossler, Karsten Boldt, Leonie Alten, Marius Ueffing, Martin Blum, Achim Gossler
Formato:	article
Lenguaje:	EN
Publicado:	Nature Portfolio 2018
Materias:	Motile Cilia mIMCD3 Cells Multiciliated Cells (MCCs) Epidermal MCCs Protein FAM107B Medicine R Science Q
Acceso en línea:	https://doaj.org/article/136882a3519e42bc9a7e2b67c949699d
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spelling	oai:doaj.org-article:136882a3519e42bc9a7e2b67c949699d2021-12-02T15:08:50ZThe evolutionary conserved FOXJ1 target gene Fam183b is essential for motile cilia in Xenopus but dispensable for ciliary function in mice10.1038/s41598-018-33045-22045-2322https://doaj.org/article/136882a3519e42bc9a7e2b67c949699d2018-10-01T00:00:00Zhttps://doi.org/10.1038/s41598-018-33045-2https://doaj.org/toc/2045-2322Abstract The transcription factor FOXJ1 is essential for the formation of motile cilia throughout the animal kingdom. Target genes therefore likely constitute an important part of the motile cilia program. Here, we report on the analysis of one of these targets, Fam183b, in Xenopus and mice. Fam183b encodes a protein with unknown function which is conserved from the green algae Chlamydomonas to humans. Fam183b is expressed in tissues harbouring motile cilia in both mouse and frog embryos. FAM183b protein localises to basal bodies of cilia in mIMCD3 cells and of multiciliated cells of the frog larval epidermis. In addition, FAM183b interacts with NUP93, which also localises to basal bodies. During frog embryogenesis, Fam183b was dispensable for laterality specification and brain development, but required for ciliogenesis and motility of epidermal multiciliated cells and nephrostomes, i.e. the embryonic kidney. Surprisingly, mice homozygous for a null allele did not display any defects indicative of disrupted motile ciliary function. The lack of a cilia phenotype in mouse and the limited requirements in frog contrast with high sequence conservation and the correlation of gene expression with the presence of motile cilia. This finding may be explained through compensatory mechanisms at sites where no defects were observed in our FAM183b-loss-of-function studies.Anja BeckersTim OttKarin Schuster-GosslerKarsten BoldtLeonie AltenMarius UeffingMartin BlumAchim GosslerNature PortfolioarticleMotile CiliamIMCD3 CellsMulticiliated Cells (MCCs)Epidermal MCCsProtein FAM107BMedicineRScienceQENScientific Reports, Vol 8, Iss 1, Pp 1-12 (2018)
institution	DOAJ
collection	DOAJ
language	EN
topic	Motile Cilia mIMCD3 Cells Multiciliated Cells (MCCs) Epidermal MCCs Protein FAM107B Medicine R Science Q
spellingShingle	Motile Cilia mIMCD3 Cells Multiciliated Cells (MCCs) Epidermal MCCs Protein FAM107B Medicine R Science Q Anja Beckers Tim Ott Karin Schuster-Gossler Karsten Boldt Leonie Alten Marius Ueffing Martin Blum Achim Gossler The evolutionary conserved FOXJ1 target gene Fam183b is essential for motile cilia in Xenopus but dispensable for ciliary function in mice
description	Abstract The transcription factor FOXJ1 is essential for the formation of motile cilia throughout the animal kingdom. Target genes therefore likely constitute an important part of the motile cilia program. Here, we report on the analysis of one of these targets, Fam183b, in Xenopus and mice. Fam183b encodes a protein with unknown function which is conserved from the green algae Chlamydomonas to humans. Fam183b is expressed in tissues harbouring motile cilia in both mouse and frog embryos. FAM183b protein localises to basal bodies of cilia in mIMCD3 cells and of multiciliated cells of the frog larval epidermis. In addition, FAM183b interacts with NUP93, which also localises to basal bodies. During frog embryogenesis, Fam183b was dispensable for laterality specification and brain development, but required for ciliogenesis and motility of epidermal multiciliated cells and nephrostomes, i.e. the embryonic kidney. Surprisingly, mice homozygous for a null allele did not display any defects indicative of disrupted motile ciliary function. The lack of a cilia phenotype in mouse and the limited requirements in frog contrast with high sequence conservation and the correlation of gene expression with the presence of motile cilia. This finding may be explained through compensatory mechanisms at sites where no defects were observed in our FAM183b-loss-of-function studies.
format	article
author	Anja Beckers Tim Ott Karin Schuster-Gossler Karsten Boldt Leonie Alten Marius Ueffing Martin Blum Achim Gossler
author_facet	Anja Beckers Tim Ott Karin Schuster-Gossler Karsten Boldt Leonie Alten Marius Ueffing Martin Blum Achim Gossler
author_sort	Anja Beckers
title	The evolutionary conserved FOXJ1 target gene Fam183b is essential for motile cilia in Xenopus but dispensable for ciliary function in mice
title_short	The evolutionary conserved FOXJ1 target gene Fam183b is essential for motile cilia in Xenopus but dispensable for ciliary function in mice
title_full	The evolutionary conserved FOXJ1 target gene Fam183b is essential for motile cilia in Xenopus but dispensable for ciliary function in mice
title_fullStr	The evolutionary conserved FOXJ1 target gene Fam183b is essential for motile cilia in Xenopus but dispensable for ciliary function in mice
title_full_unstemmed	The evolutionary conserved FOXJ1 target gene Fam183b is essential for motile cilia in Xenopus but dispensable for ciliary function in mice
title_sort	evolutionary conserved foxj1 target gene fam183b is essential for motile cilia in xenopus but dispensable for ciliary function in mice
publisher	Nature Portfolio
publishDate	2018
url	https://doaj.org/article/136882a3519e42bc9a7e2b67c949699d
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The evolutionary conserved FOXJ1 target gene Fam183b is essential for motile cilia in Xenopus but dispensable for ciliary function in mice

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