A function for the Joubert syndrome protein Arl13b in ciliary membrane extension and ciliary length regulation

Developmental Biology
Hao LuSudipto Roy

Abstract

Cilia perform a variety of functions in a number of developmental and physiological contexts, and are implicated in the pathogenesis of a wide spectrum of human disorders. While the ciliary axoneme is assembled by intraflagellar transport, how ciliary membrane length is regulated is not completely understood. Here, we show that zebrafish embryos as well as mammalian cells overexpressing the ciliary membrane protein Arl13b, an ARF family small GTPase that is essential for ciliary differentiation, showed pronounced increase in ciliary length. Intriguingly, this increase in cilia length occurred as a function of the amounts of overexpressed Arl13b. While the motility of Arl13b overexpressing excessively long motile cilia was obviously disrupted, surprisingly, the abnormally long immotile primary cilia seemed to retain their signaling capacity. arl13b is induced by FoxJ1 and Rfx, and these ciliogenic transcription factors are unable to promote ciliary length increase when Arl13b activity is inhibited. Conversely, overexpression of Arl13b was sufficient to restore ciliary length in zebrafish embryos deficient in FoxJ1 function. We show that Arl13b increases cilia length by inducing protrusion of the ciliary membrane, which is then f...Continue Reading

References

Mar 2, 2005·Proceedings of the National Academy of Sciences of the United States of America·Viktor StolcWallace F Marshall
Oct 3, 2006·Annual Review of Physiology·Peter Satir, Søren Tvorup Christensen
Apr 17, 2007·Current Biology : CB·Christine BlaineauPatrick Bastien
May 10, 2007·Developmental Cell·Tamara CasparyKathryn V Anderson
Oct 24, 2007·The Journal of Cell Biology·Susanne GraserErich A Nigg
Apr 26, 2008·Developmental Dynamics : an Official Publication of the American Association of Anatomists·Nicole Santos, Jeremy F Reiter
Jun 17, 2008·Biochemical and Biophysical Research Communications·Yuji HoriToshiaki Katada
Aug 5, 2008·American Journal of Human Genetics·Vincent CantagrelJoseph G Gleeson
Aug 12, 2008·Proceedings of the National Academy of Sciences of the United States of America·Rainer BeckFelix Wieland
Nov 18, 2008·Nature Genetics·Jennifer L StubbsChris Kintner
Dec 17, 2009·Developmental Dynamics : an Official Publication of the American Association of Anatomists·Shang Yew TaySudipto Roy
Mar 23, 2010·Nature Cell Biology·Antonia BorovinaBrian Ciruna
Apr 17, 2010·Nature Reviews. Genetics·Sarah C Goetz, Kathryn V Anderson
Jun 10, 2010·The Journal of Cell Biology·Yujie LiJinghua Hu
Dec 15, 2010·Proceedings of the National Academy of Sciences of the United States of America·Yoshihiro OmoriTakahisa Furukawa
Apr 22, 2011·The New England Journal of Medicine·Friedhelm HildebrandtNicholas Katsanis
Jul 5, 2011·Nature Genetics·Francesc R Garcia-GonzaloJeremy F Reiter
Oct 7, 2011·Molecular Biology of the Cell·Christine E LarkinsTamara Caspary
Dec 20, 2011·Differentiation; Research in Biological Diversity·Prachee Avasthi, Wallace F Marshall
Sep 8, 2012·Science·Yee-Hung M Chan, Wallace F Marshall
Nov 7, 2012·The Journal of Cell Biology·Yujie LiJinghua Hu
Nov 15, 2012·Proceedings of the National Academy of Sciences of the United States of America·Melissa C HumbertSeongjin Seo
Nov 22, 2012·Developmental Cell·Shinsuke NiwaNobutaka Hirokawa
May 31, 2013·Open Biology·Deepak Babu, Sudipto Roy
Mar 20, 2014·Development·Semil P ChoksiSudipto Roy

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Citations

Mar 30, 2016·Journal of Genetics and Genomics = Yi Chuan Xue Bao·Zhu SongChengtian Zhao
Aug 27, 2016·Modern Pathology : an Official Journal of the United States and Canadian Academy of Pathology, Inc·Shannon CoySandro Santagata
Oct 12, 2016·PLoS Genetics·Chen-Jei Hong, Bruce A Hamilton
Nov 20, 2016·The Journal of Pathology·Hannah M Mitchison, Enza Maria Valente
Feb 22, 2018·Journal of Molecular Neuroscience : MN·Éva SiposPéter Ács
Dec 20, 2017·European Journal of Human Genetics : EJHG·Rafiullah RafiullahGudrun A Rappold
Jan 1, 2019·FASEB Journal : Official Publication of the Federation of American Societies for Experimental Biology·Eric H J VerschurenRené J M Bindels
Dec 31, 2019·Philosophical Transactions of the Royal Society of London. Series B, Biological Sciences·Tanya T Whitfield
Mar 14, 2020·FASEB Journal : Official Publication of the Federation of American Societies for Experimental Biology·Wei WangPamela V Tran
Mar 5, 2020·ELife·Eduardo D GiganteTamara Caspary
Aug 23, 2019·EMBO Reports·Stefanie KuhnsOliver E Blacque
Apr 3, 2016·Genetics·Niedharsan Pooranachandran, Jarema J Malicki
Aug 7, 2018·Cilia·Sarbjot KaurMarie-Louise Ward
Sep 19, 2018·Development·Lei Wang, Brian D Dynlacht
Jun 5, 2020·Science Advances·Gabriela Toro-Tapia, Raman M Das
Apr 14, 2017·Royal Society Open Science·P PintadoS S Lopes
Aug 11, 2018·International Journal of Gynecological Cancer : Official Journal of the International Gynecological Cancer Society·Zakia A AbdelhamedColin A Johnson
Feb 5, 2019·Microscopy·Faryal Ijaz, Koji Ikegami
Jan 24, 2021·Journal of Cell Science·Mariagrazia PartisaniMichel Franco
Mar 9, 2021·The European Physical Journal. E, Soft Matter·Jan Niklas HansenDagmar Wachten
Sep 17, 2020·Science Advances·Gabriela Toro-Tapia, Raman M Das
Jun 8, 2021·Frontiers in Cell and Developmental Biology·Wei WangPamela V Tran
Jun 23, 2021·Journal of Cell Science·Magdalena Cardenas-RodriguezIain A Drummond
Aug 26, 2021·Journal of Cell Science·Mariagrazia PartisaniMichel Franco

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