scholarly journals Rsph4a is essential for the triplet radial spoke head assembly of the mouse motile cilia

PLoS Genetics ◽  
2020 ◽  
Vol 16 (3) ◽  
pp. e1008664
Author(s):  
Hiroshi Yoke ◽  
Hironori Ueno ◽  
Akihiro Narita ◽  
Takafumi Sakai ◽  
Kahoru Horiuchi ◽  
...  
Keyword(s):  
Radial Spoke ◽  
Head Assembly ◽  
Motile Cilia

scholarly journals The CSC proteins FAP61 and FAP251 build the basal substructures of radial spoke 3 in cilia

2015 ◽  
Vol 26 (8) ◽  
pp. 1463-1475 ◽  
Author(s):  
Paulina Urbanska ◽  
Kangkang Song ◽  
Ewa Joachimiak ◽  
Lucja Krzemien-Ojak ◽  
Piotr Koprowski ◽  
...  
Keyword(s):  
Radial Spoke ◽  
Ciliary Motility ◽  
Distinct Structure ◽  
Radial Spokes ◽  
Dynein Arms ◽  
Motile Cilia ◽  
The Individual ◽  
And Function

Dynein motors and regulatory complexes repeat every 96 nm along the length of motile cilia. Each repeat contains three radial spokes, RS1, RS2, and RS3, which transduct signals between the central microtubules and dynein arms. Each radial spoke has a distinct structure, but little is known about the mechanisms of assembly and function of the individual radial spokes. In Chlamydomonas, calmodulin and spoke-associated complex (CSC) is composed of FAP61, FAP91, and FAP251 and has been linked to the base of RS2 and RS3. We show that in Tetrahymena, loss of either FAP61 or FAP251 reduces cell swimming and affects the ciliary waveform and that RS3 is either missing or incomplete, whereas RS1 and RS2 are unaffected. Specifically, FAP251-null cilia lack an arch-like density at the RS3 base, whereas FAP61-null cilia lack an adjacent portion of the RS3 stem region. This suggests that the CSC proteins are crucial for stable and functional assembly of RS3 and that RS3 and the CSC are important for ciliary motility.

scholarly journals The I1 dynein-associated tether and tether head complex is a conserved regulator of ciliary motility

2018 ◽  
Vol 29 (9) ◽  
pp. 1048-1059 ◽  
Author(s):  
Gang Fu ◽  
Qian Wang ◽  
Nhan Phan ◽  
Paulina Urbanska ◽  
Ewa Joachimiak ◽  
...  
Keyword(s):  
Signaling Pathway ◽  
Regulatory Mechanisms ◽  
Ciliary Beating ◽  
Radial Spoke ◽  
Ciliary Motility ◽  
Dynein Motor ◽  
Axonemal Dynein ◽  
Motile Cilia ◽  
Mechanical Feedback

Motile cilia are essential for propelling cells and moving fluids across tissues. The activity of axonemal dynein motors must be precisely coordinated to generate ciliary motility, but their regulatory mechanisms are not well understood. The tether and tether head (T/TH) complex was hypothesized to provide mechanical feedback during ciliary beating because it links the motor domains of the regulatory I1 dynein to the ciliary doublet microtubule. Combining genetic and biochemical approaches with cryoelectron tomography, we identified FAP44 and FAP43 (plus the algae-specific, FAP43-redundant FAP244) as T/TH components. WT-mutant comparisons revealed that the heterodimeric T/TH complex is required for the positional stability of the I1 dynein motor domains, stable anchoring of CK1 kinase, and proper phosphorylation of the regulatory IC138-subunit. T/TH also interacts with inner dynein arm d and radial spoke 3, another important motility regulator. The T/TH complex is a conserved regulator of I1 dynein and plays an important role in the signaling pathway that is critical for normal ciliary motility.

scholarly journals TGF-β1 increases permeability of ciliated airway epithelia via redistribution of claudin 3 from tight junction into cell nuclei

2021 ◽  
Author(s):  
Carolin Schilpp ◽  
Robin Lochbaum ◽  
Peter Braubach ◽  
Danny Jonigk ◽  
Manfred Frick ◽  
...  
Keyword(s):  
Time Course ◽  
Atopic Asthma ◽  
Ciliated Cells ◽  
Cell Nuclei ◽  
Tissue Remodelling ◽  
Similar Time ◽  
Airway Epithelia ◽  
Epithelial Integrity ◽  
Motile Cilia ◽  
Tgf Β1

AbstractTGF-β1 is a major mediator of airway tissue remodelling during atopic asthma and affects tight junctions (TJs) of airway epithelia. However, its impact on TJs of ciliated epithelia is sparsely investigated. Herein we elaborated effects of TGF-β1 on TJs of primary human bronchial epithelial cells. We demonstrate that TGF-β1 activates TGF-β1 receptors TGFBR1 and TGFBR2 resulting in ALK5-mediated phosphorylation of SMAD2. We observed that TGFBR1 and -R2 localize specifically on motile cilia. TGF-β1 activated accumulation of phosphorylated SMAD2 (pSMAD2-C) at centrioles of motile cilia and at cell nuclei. This triggered an increase in paracellular permeability via cellular redistribution of claudin 3 (CLDN3) from TJs into cell nuclei followed by disruption of epithelial integrity and formation of epithelial lesions. Only ciliated cells express TGF-β1 receptors; however, nuclear accumulations of pSMAD2-C and CLDN3 redistribution were observed with similar time course in ciliated and non-ciliated cells. In summary, we demonstrate a role of motile cilia in TGF-β1 sensing and showed that TGF-β1 disturbs TJ permeability of conductive airway epithelia by redistributing CLDN3 from TJs into cell nuclei. We conclude that the observed effects contribute to loss of epithelial integrity during atopic asthma.

scholarly journals Correction: Foxn4 promotes gene expression required for the formation of multiple motile cilia

Development ◽  
2017 ◽  
Vol 144 (4) ◽  
pp. 731-731 ◽  
Author(s):  
Evan P. Campbell ◽  
Ian K. Quigley ◽  
Chris Kintner
Keyword(s):  
Gene Expression ◽  
Motile Cilia

scholarly journals IC138 Defines a Subdomain at the Base of the I1 Dynein That Regulates Microtubule Sliding and Flagellar Motility

2009 ◽  
Vol 20 (13) ◽  
pp. 3055-3063 ◽  
Author(s):  
Raqual Bower ◽  
Kristyn VanderWaal ◽  
Eileen O'Toole ◽  
Laura Fox ◽  
Catherine Perrone ◽  
...  
Keyword(s):  
Double Mutant ◽  
Essential Role ◽  
Null Allele ◽  
Flagellar Motility ◽  
Wild Type ◽  
Gene Encoding ◽  
Radial Spoke ◽  
Mutant Strains ◽  
Dynein Arms ◽  
Image Averaging

To understand the mechanisms that regulate the assembly and activity of flagellar dyneins, we focused on the I1 inner arm dynein (dynein f) and a null allele, bop5-2, defective in the gene encoding the IC138 phosphoprotein subunit. I1 dynein assembles in bop5-2 axonemes but lacks at least four subunits: IC138, IC97, LC7b, and flagellar-associated protein (FAP) 120—defining a new I1 subcomplex. Electron microscopy and image averaging revealed a defect at the base of the I1 dynein, in between radial spoke 1 and the outer dynein arms. Microtubule sliding velocities also are reduced. Transformation with wild-type IC138 restores assembly of the IC138 subcomplex and rescues microtubule sliding. These observations suggest that the IC138 subcomplex is required to coordinate I1 motor activity. To further test this hypothesis, we analyzed microtubule sliding in radial spoke and double mutant strains. The results reveal an essential role for the IC138 subcomplex in the regulation of I1 activity by the radial spoke/phosphorylation pathway.

scholarly journals The actin nucleator Cordon-bleu is required for development of motile cilia in zebrafish

2011 ◽  
Vol 350 (1) ◽  
pp. 101-111 ◽  
Author(s):  
Andrew M. Ravanelli ◽  
John Klingensmith
Keyword(s):  
Motile Cilia ◽  
Actin Nucleator

scholarly journals Maturation of the Human Motile Cilia Waveform in Airway Cells

2018 ◽  
Vol 114 (3) ◽  
pp. 649a
Author(s):  
Alina Oltean ◽  
Philip V. Bayly ◽  
Steven L. Brody
Keyword(s):  
Motile Cilia ◽  
Airway Cells
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