Eintrag weiter verarbeiten
RFX3 governs growth and beating efficiency of motile cilia in mouse and controls the expression of genes involved in human ciliopathies
Gespeichert in:
Zeitschriftentitel: | Journal of Cell Science |
---|---|
Personen und Körperschaften: | , , , , , , , |
In: | Journal of Cell Science, 122, 2009, 17, S. 3180-3189 |
Format: | E-Article |
Sprache: | Englisch |
veröffentlicht: |
The Company of Biologists
|
Schlagwörter: |
author_facet |
El Zein, Loubna Ait-Lounis, Aouatef Morlé, Laurette Thomas, Joëlle Chhin, Brigitte Spassky, Nathalie Reith, Walter Durand, Bénédicte El Zein, Loubna Ait-Lounis, Aouatef Morlé, Laurette Thomas, Joëlle Chhin, Brigitte Spassky, Nathalie Reith, Walter Durand, Bénédicte |
---|---|
author |
El Zein, Loubna Ait-Lounis, Aouatef Morlé, Laurette Thomas, Joëlle Chhin, Brigitte Spassky, Nathalie Reith, Walter Durand, Bénédicte |
spellingShingle |
El Zein, Loubna Ait-Lounis, Aouatef Morlé, Laurette Thomas, Joëlle Chhin, Brigitte Spassky, Nathalie Reith, Walter Durand, Bénédicte Journal of Cell Science RFX3 governs growth and beating efficiency of motile cilia in mouse and controls the expression of genes involved in human ciliopathies Cell Biology |
author_sort |
el zein, loubna |
spelling |
El Zein, Loubna Ait-Lounis, Aouatef Morlé, Laurette Thomas, Joëlle Chhin, Brigitte Spassky, Nathalie Reith, Walter Durand, Bénédicte 1477-9137 0021-9533 The Company of Biologists Cell Biology http://dx.doi.org/10.1242/jcs.048348 <jats:p>Cilia are cellular organelles that play essential physiological and developmental functions in various organisms. They can be classified into two categories, primary cilia and motile cilia, on the basis of their axonemal architecture. Regulatory factor X (RFX) transcription factors have been shown to be involved in the assembly of primary cilia in Caenorhabditis elegans, Drosophila and mice. Here, we have taken advantage of a novel primary-cell culture system derived from mouse brain to show that RFX3 is also necessary for biogenesis of motile cilia. We found that the growth and beating efficiencies of motile cilia are impaired in multiciliated Rfx3–/– cells. RFX3 was required for optimal expression of the FOXJ1 transcription factor, a key player in the differentiation program of motile cilia. Furthermore, we demonstrate for the first time that RFX3 regulates the expression of axonemal dyneins involved in ciliary motility by binding directly to the promoters of their genes. In conclusion, RFX proteins not only regulate genes involved in ciliary assembly, but also genes that are involved in ciliary motility and that are associated with ciliopathies such as primary ciliary dyskinesia in humans.</jats:p> RFX3 governs growth and beating efficiency of motile cilia in mouse and controls the expression of genes involved in human ciliopathies Journal of Cell Science |
doi_str_mv |
10.1242/jcs.048348 |
facet_avail |
Online Free |
finc_class_facet |
Biologie |
format |
ElectronicArticle |
fullrecord |
blob:ai-49-aHR0cDovL2R4LmRvaS5vcmcvMTAuMTI0Mi9qY3MuMDQ4MzQ4 |
id |
ai-49-aHR0cDovL2R4LmRvaS5vcmcvMTAuMTI0Mi9qY3MuMDQ4MzQ4 |
institution |
DE-Gla1 DE-Zi4 DE-15 DE-Pl11 DE-Rs1 DE-105 DE-14 DE-Ch1 DE-L229 DE-D275 DE-Bn3 DE-Brt1 DE-Zwi2 DE-D161 |
imprint |
The Company of Biologists, 2009 |
imprint_str_mv |
The Company of Biologists, 2009 |
issn |
1477-9137 0021-9533 |
issn_str_mv |
1477-9137 0021-9533 |
language |
English |
mega_collection |
The Company of Biologists (CrossRef) |
match_str |
elzein2009rfx3governsgrowthandbeatingefficiencyofmotileciliainmouseandcontrolstheexpressionofgenesinvolvedinhumanciliopathies |
publishDateSort |
2009 |
publisher |
The Company of Biologists |
recordtype |
ai |
record_format |
ai |
series |
Journal of Cell Science |
source_id |
49 |
title |
RFX3 governs growth and beating efficiency of motile cilia in mouse and controls the expression of genes involved in human ciliopathies |
title_unstemmed |
RFX3 governs growth and beating efficiency of motile cilia in mouse and controls the expression of genes involved in human ciliopathies |
title_full |
RFX3 governs growth and beating efficiency of motile cilia in mouse and controls the expression of genes involved in human ciliopathies |
title_fullStr |
RFX3 governs growth and beating efficiency of motile cilia in mouse and controls the expression of genes involved in human ciliopathies |
title_full_unstemmed |
RFX3 governs growth and beating efficiency of motile cilia in mouse and controls the expression of genes involved in human ciliopathies |
title_short |
RFX3 governs growth and beating efficiency of motile cilia in mouse and controls the expression of genes involved in human ciliopathies |
title_sort |
rfx3 governs growth and beating efficiency of motile cilia in mouse and controls the expression of genes involved in human ciliopathies |
topic |
Cell Biology |
url |
http://dx.doi.org/10.1242/jcs.048348 |
publishDate |
2009 |
physical |
3180-3189 |
description |
<jats:p>Cilia are cellular organelles that play essential physiological and developmental functions in various organisms. They can be classified into two categories, primary cilia and motile cilia, on the basis of their axonemal architecture. Regulatory factor X (RFX) transcription factors have been shown to be involved in the assembly of primary cilia in Caenorhabditis elegans, Drosophila and mice. Here, we have taken advantage of a novel primary-cell culture system derived from mouse brain to show that RFX3 is also necessary for biogenesis of motile cilia. We found that the growth and beating efficiencies of motile cilia are impaired in multiciliated Rfx3–/– cells. RFX3 was required for optimal expression of the FOXJ1 transcription factor, a key player in the differentiation program of motile cilia. Furthermore, we demonstrate for the first time that RFX3 regulates the expression of axonemal dyneins involved in ciliary motility by binding directly to the promoters of their genes. In conclusion, RFX proteins not only regulate genes involved in ciliary assembly, but also genes that are involved in ciliary motility and that are associated with ciliopathies such as primary ciliary dyskinesia in humans.</jats:p> |
container_issue |
17 |
container_start_page |
3180 |
container_title |
Journal of Cell Science |
container_volume |
122 |
format_de105 |
Article, E-Article |
format_de14 |
Article, E-Article |
format_de15 |
Article, E-Article |
format_de520 |
Article, E-Article |
format_de540 |
Article, E-Article |
format_dech1 |
Article, E-Article |
format_ded117 |
Article, E-Article |
format_degla1 |
E-Article |
format_del152 |
Buch |
format_del189 |
Article, E-Article |
format_dezi4 |
Article |
format_dezwi2 |
Article, E-Article |
format_finc |
Article, E-Article |
format_nrw |
Article, E-Article |
_version_ |
1792342054128320512 |
geogr_code |
not assigned |
last_indexed |
2024-03-01T16:29:42.215Z |
geogr_code_person |
not assigned |
openURL |
url_ver=Z39.88-2004&ctx_ver=Z39.88-2004&ctx_enc=info%3Aofi%2Fenc%3AUTF-8&rfr_id=info%3Asid%2Fvufind.svn.sourceforge.net%3Agenerator&rft.title=RFX3+governs+growth+and+beating+efficiency+of+motile+cilia+in+mouse+and+controls+the+expression+of+genes+involved+in+human+ciliopathies&rft.date=2009-09-01&genre=article&issn=0021-9533&volume=122&issue=17&spage=3180&epage=3189&pages=3180-3189&jtitle=Journal+of+Cell+Science&atitle=RFX3+governs+growth+and+beating+efficiency+of+motile+cilia+in+mouse+and+controls+the+expression+of+genes+involved+in+human+ciliopathies&aulast=Durand&aufirst=Be%CC%81ne%CC%81dicte&rft_id=info%3Adoi%2F10.1242%2Fjcs.048348&rft.language%5B0%5D=eng |
SOLR | |
_version_ | 1792342054128320512 |
author | El Zein, Loubna, Ait-Lounis, Aouatef, Morlé, Laurette, Thomas, Joëlle, Chhin, Brigitte, Spassky, Nathalie, Reith, Walter, Durand, Bénédicte |
author_facet | El Zein, Loubna, Ait-Lounis, Aouatef, Morlé, Laurette, Thomas, Joëlle, Chhin, Brigitte, Spassky, Nathalie, Reith, Walter, Durand, Bénédicte, El Zein, Loubna, Ait-Lounis, Aouatef, Morlé, Laurette, Thomas, Joëlle, Chhin, Brigitte, Spassky, Nathalie, Reith, Walter, Durand, Bénédicte |
author_sort | el zein, loubna |
container_issue | 17 |
container_start_page | 3180 |
container_title | Journal of Cell Science |
container_volume | 122 |
description | <jats:p>Cilia are cellular organelles that play essential physiological and developmental functions in various organisms. They can be classified into two categories, primary cilia and motile cilia, on the basis of their axonemal architecture. Regulatory factor X (RFX) transcription factors have been shown to be involved in the assembly of primary cilia in Caenorhabditis elegans, Drosophila and mice. Here, we have taken advantage of a novel primary-cell culture system derived from mouse brain to show that RFX3 is also necessary for biogenesis of motile cilia. We found that the growth and beating efficiencies of motile cilia are impaired in multiciliated Rfx3–/– cells. RFX3 was required for optimal expression of the FOXJ1 transcription factor, a key player in the differentiation program of motile cilia. Furthermore, we demonstrate for the first time that RFX3 regulates the expression of axonemal dyneins involved in ciliary motility by binding directly to the promoters of their genes. In conclusion, RFX proteins not only regulate genes involved in ciliary assembly, but also genes that are involved in ciliary motility and that are associated with ciliopathies such as primary ciliary dyskinesia in humans.</jats:p> |
doi_str_mv | 10.1242/jcs.048348 |
facet_avail | Online, Free |
finc_class_facet | Biologie |
format | ElectronicArticle |
format_de105 | Article, E-Article |
format_de14 | Article, E-Article |
format_de15 | Article, E-Article |
format_de520 | Article, E-Article |
format_de540 | Article, E-Article |
format_dech1 | Article, E-Article |
format_ded117 | Article, E-Article |
format_degla1 | E-Article |
format_del152 | Buch |
format_del189 | Article, E-Article |
format_dezi4 | Article |
format_dezwi2 | Article, E-Article |
format_finc | Article, E-Article |
format_nrw | Article, E-Article |
geogr_code | not assigned |
geogr_code_person | not assigned |
id | ai-49-aHR0cDovL2R4LmRvaS5vcmcvMTAuMTI0Mi9qY3MuMDQ4MzQ4 |
imprint | The Company of Biologists, 2009 |
imprint_str_mv | The Company of Biologists, 2009 |
institution | DE-Gla1, DE-Zi4, DE-15, DE-Pl11, DE-Rs1, DE-105, DE-14, DE-Ch1, DE-L229, DE-D275, DE-Bn3, DE-Brt1, DE-Zwi2, DE-D161 |
issn | 1477-9137, 0021-9533 |
issn_str_mv | 1477-9137, 0021-9533 |
language | English |
last_indexed | 2024-03-01T16:29:42.215Z |
match_str | elzein2009rfx3governsgrowthandbeatingefficiencyofmotileciliainmouseandcontrolstheexpressionofgenesinvolvedinhumanciliopathies |
mega_collection | The Company of Biologists (CrossRef) |
physical | 3180-3189 |
publishDate | 2009 |
publishDateSort | 2009 |
publisher | The Company of Biologists |
record_format | ai |
recordtype | ai |
series | Journal of Cell Science |
source_id | 49 |
spelling | El Zein, Loubna Ait-Lounis, Aouatef Morlé, Laurette Thomas, Joëlle Chhin, Brigitte Spassky, Nathalie Reith, Walter Durand, Bénédicte 1477-9137 0021-9533 The Company of Biologists Cell Biology http://dx.doi.org/10.1242/jcs.048348 <jats:p>Cilia are cellular organelles that play essential physiological and developmental functions in various organisms. They can be classified into two categories, primary cilia and motile cilia, on the basis of their axonemal architecture. Regulatory factor X (RFX) transcription factors have been shown to be involved in the assembly of primary cilia in Caenorhabditis elegans, Drosophila and mice. Here, we have taken advantage of a novel primary-cell culture system derived from mouse brain to show that RFX3 is also necessary for biogenesis of motile cilia. We found that the growth and beating efficiencies of motile cilia are impaired in multiciliated Rfx3–/– cells. RFX3 was required for optimal expression of the FOXJ1 transcription factor, a key player in the differentiation program of motile cilia. Furthermore, we demonstrate for the first time that RFX3 regulates the expression of axonemal dyneins involved in ciliary motility by binding directly to the promoters of their genes. In conclusion, RFX proteins not only regulate genes involved in ciliary assembly, but also genes that are involved in ciliary motility and that are associated with ciliopathies such as primary ciliary dyskinesia in humans.</jats:p> RFX3 governs growth and beating efficiency of motile cilia in mouse and controls the expression of genes involved in human ciliopathies Journal of Cell Science |
spellingShingle | El Zein, Loubna, Ait-Lounis, Aouatef, Morlé, Laurette, Thomas, Joëlle, Chhin, Brigitte, Spassky, Nathalie, Reith, Walter, Durand, Bénédicte, Journal of Cell Science, RFX3 governs growth and beating efficiency of motile cilia in mouse and controls the expression of genes involved in human ciliopathies, Cell Biology |
title | RFX3 governs growth and beating efficiency of motile cilia in mouse and controls the expression of genes involved in human ciliopathies |
title_full | RFX3 governs growth and beating efficiency of motile cilia in mouse and controls the expression of genes involved in human ciliopathies |
title_fullStr | RFX3 governs growth and beating efficiency of motile cilia in mouse and controls the expression of genes involved in human ciliopathies |
title_full_unstemmed | RFX3 governs growth and beating efficiency of motile cilia in mouse and controls the expression of genes involved in human ciliopathies |
title_short | RFX3 governs growth and beating efficiency of motile cilia in mouse and controls the expression of genes involved in human ciliopathies |
title_sort | rfx3 governs growth and beating efficiency of motile cilia in mouse and controls the expression of genes involved in human ciliopathies |
title_unstemmed | RFX3 governs growth and beating efficiency of motile cilia in mouse and controls the expression of genes involved in human ciliopathies |
topic | Cell Biology |
url | http://dx.doi.org/10.1242/jcs.048348 |