TY - JOUR
T1 - Interplay of RFX transcription factors 1, 2 and 3 in motile ciliogenesis
AU - Lemeille, Sylvain
AU - Paschaki, Marie
AU - Baas, Dominique
AU - Morlé, Laurette
AU - Duteyrat, Jean Luc
AU - Ait-Lounis, Aouatef
AU - Barras, Emmanuèle
AU - Soulavie, Fabien
AU - Jerber, Julie
AU - Thomas, Joëelle
AU - Zhang, Yong
AU - Holtzman, Michael J.
AU - W. Stephen Kistler, Stephen Kistler
AU - Reith, Walter
AU - Durand, Bénédicte
N1 - Funding Information:
Fondation pour la Recherche Médicale (FRM) [DEQ20131029168 to B.D.]; ANR (Ciliopath-X); F.S. was supported by a doctoral fellowship from the Région Rhône-Alpes; J.J. was supported by a doctoral fellowship from the Fondation pour la Recherche Médicale (Prix Line Pomaret-Delalande); Work in the team of W. Reith was supported by multiple grants from the Swiss National Science Foundation. Funding for open access charge: Research grants.
Publisher Copyright:
© 2020 The Author(s) 2020. Published by Oxford University Press on behalf of Nucleic Acids Research.
PY - 2020/9/18
Y1 - 2020/9/18
N2 - Cilia assembly is under strict transcriptional control during animal development. In vertebrates, a hierarchy of transcription factors (TFs) are involved in controlling the specification, differentiation and function of multiciliated epithelia. RFX TFs play key functions in the control of ciliogenesis in animals. Whereas only one RFX factor regulates ciliogenesis in C. elegans, several distinct RFX factors have been implicated in this process in vertebrates. However, a clear understanding of the specific and redundant functions of different RFX factors in ciliated cells remains lacking. Using RNA-seq and ChIP-seq approaches we identified genes regulated directly and indirectly by RFX1, RFX2 and RFX3 in mouse ependymal cells. We show that these three TFs have both redundant and specific functions in ependymal cells. Whereas RFX1, RFX2 and RFX3 occupy many shared genomic loci, only RFX2 and RFX3 play a prominent and redundant function in the control of motile ciliogenesis in mice. Our results provide a valuable list of candidate ciliary genes. They also reveal stunning differences between compensatory processes operating in vivo and ex vivo.
AB - Cilia assembly is under strict transcriptional control during animal development. In vertebrates, a hierarchy of transcription factors (TFs) are involved in controlling the specification, differentiation and function of multiciliated epithelia. RFX TFs play key functions in the control of ciliogenesis in animals. Whereas only one RFX factor regulates ciliogenesis in C. elegans, several distinct RFX factors have been implicated in this process in vertebrates. However, a clear understanding of the specific and redundant functions of different RFX factors in ciliated cells remains lacking. Using RNA-seq and ChIP-seq approaches we identified genes regulated directly and indirectly by RFX1, RFX2 and RFX3 in mouse ependymal cells. We show that these three TFs have both redundant and specific functions in ependymal cells. Whereas RFX1, RFX2 and RFX3 occupy many shared genomic loci, only RFX2 and RFX3 play a prominent and redundant function in the control of motile ciliogenesis in mice. Our results provide a valuable list of candidate ciliary genes. They also reveal stunning differences between compensatory processes operating in vivo and ex vivo.
UR - http://www.scopus.com/inward/record.url?scp=85091263990&partnerID=8YFLogxK
U2 - 10.1093/nar/gkaa625
DO - 10.1093/nar/gkaa625
M3 - Article
C2 - 32725242
AN - SCOPUS:85091263990
VL - 48
SP - 9019
EP - 9036
JO - Nucleic Acids Research
JF - Nucleic Acids Research
SN - 0305-1048
IS - 16
ER -