Ciliary Proteins: Filling the Gaps. Recent Advances in Deciphering the Protein Composition of Motile Ciliary Complexes

Cilia Distal Domain: Diversity in Evolutionarily Conserved Structures

(2019) Helena Soares et al.   Cells

Inner lumen proteins stabilize doublet microtubules in cilia and flagella

(2019) Mikito Owa et al.   Nature Communications

Proteome of the central apparatus of a ciliary axoneme

(2019) Lei Zhao et al.   JOURNAL OF CELL BIOLOGY

WDR92 Is Required for Axonemal Dynein Heavy Chain Stability in Cytoplasm

(2019) Ramila S. Patel-King et al.   MOLECULAR BIOLOGY OF THE CELL

PACRG and FAP20 form the inner junction of axonemal doublet microtubules and regulate ciliary motility

(2019) Erin E. Dymek et al.   MOLECULAR BIOLOGY OF THE CELL

Mutations in C11orf70 Cause Primary Ciliary Dyskinesia with Randomization of Left/Right Body Asymmetry Due to Defects of Outer and Inner Dynein Arms

(2018) Inga M. Höben et al.   AMERICAN JOURNAL OF HUMAN GENETICS

C11orf70 Mutations Disrupting the Intraflagellar Transport-Dependent Assembly of Multiple Axonemal Dyneins Cause Primary Ciliary Dyskinesia

(2018) Mahmoud R. Fassad et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Microtubule Inner Proteins: A Meshwork of Luminal Proteins Stabilizing the Doublet Microtubule

(2018) Muneyoshi Ichikawa et al.   BIOESSAYS

Flagellar-associated Protein FAP85 Is a Microtubule Inner Protein That Stabilizes Microtubules

(2018) Junya Kirima et al.   CELL STRUCTURE AND FUNCTION

Ciliary proteins Fap43 and Fap44 interact with each other and are essential for proper cilia and flagella beating

(2018) Paulina Urbanska et al.   CELLULAR AND MOLECULAR LIFE SCIENCES

Advances in the Genetics of Primary Ciliary Dyskinesia

(2018) Amjad Horani et al.   CHEST

Ciliary dynein motor preassembly is regulated by Wdr92 in association with HSP90 co-chaperone, R2TP

(2018) Petra zur Lage et al.   JOURNAL OF CELL BIOLOGY

DRC2/CCDC65 is a central hub for assembly of the nexin–dynein regulatory complex and other regulators of ciliary and flagellar motility

(2018) Raqual Bower et al.   MOLECULAR BIOLOGY OF THE CELL

A microtubule-dynein tethering complex regulates the axonemal inner dynein f (I1)

(2018) Tomohiro Kubo et al.   MOLECULAR BIOLOGY OF THE CELL

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

(2018) Gang Fu et al.   MOLECULAR BIOLOGY OF THE CELL

Asymmetric distribution and spatial switching of dynein activity generates ciliary motility

(2018) Jianfeng Lin et al.   SCIENCE

The Developmental Process of the Growing Motile Ciliary Tip Region

(2018) Matthew J. Reynolds et al.   Scientific Reports

ZMYND10 stabilizes intermediate chain proteins in the cytoplasmic pre-assembly of dynein arms

(2018) Kyeong Jee Cho et al.   PLoS Genetics

ZMYND10 functions in a chaperone relay during axonemal dynein assembly

(2018) Girish R Mali et al.   eLife

Proteins that control the geometry of microtubules at the ends of cilia

(2018) Panagiota Louka et al.   JOURNAL OF CELL BIOLOGY

Tetrahymena RIB72A and RIB72B are Microtubule Inner Proteins in the ciliary doublet microtubules

(2018) Daniel Stoddard et al.   MOLECULAR BIOLOGY OF THE CELL

CFAP70 Is a Novel Axoneme-Binding Protein That Localizes at the Base of the Outer Dynein Arm and Regulates Ciliary Motility

(2018) Noritoshi Shamoto et al.   Cells

Emerging Picture of Deuterosome-Dependent Centriole Amplification in MCCs

(2018) Umama Shahid et al.   Cells

Composition, structure and function of the eukaryotic flagellum distal tip

(2018) Jacob T. Croft et al.   Essays in Biochemistry

Trafficking of ciliary membrane proteins by the intraflagellar transport/BBSome machinery

(2018) Jenna L. Wingfield et al.   Essays in Biochemistry

The roles of a flagellar HSP40 ensuring rhythmic beating

(2018) Xiaoyan Zhu et al.   MOLECULAR BIOLOGY OF THE CELL

A liquid-like organelle at the root of motile ciliopathy

(2018) Ryan L Huizar et al.   eLife

Intraflagellar transport: mechanisms of motor action, cooperation, and cargo delivery

(2017) Bram Prevo et al.   FEBS Journal

DNAAF1 links heart laterality with the AAA+ ATPase RUVBL1 and ciliary intraflagellar transport

(2017) Verity L Hartill et al.   HUMAN MOLECULAR GENETICS

Mutation of serine/threonine protein kinase 36 (STK36 ) causes primary ciliary dyskinesia with a central pair defect

(2017) Christine Edelbusch et al.   HUMAN MUTATION

Structural basis of outer dynein arm intraflagellar transport by the transport adaptor protein ODA16 and the intraflagellar transport protein IFT46

(2017) Michael Taschner et al.   JOURNAL OF BIOLOGICAL CHEMISTRY

Outer dense fibers stabilize the axoneme to maintain sperm motility

(2017) Wenlong Zhao et al.   JOURNAL OF CELLULAR AND MOLECULAR MEDICINE

Quantitative Proteomic Analysis of Human Airway Cilia Identifies Previously Uncharacterized Proteins of High Abundance

(2017) Kevin Blackburn et al.   JOURNAL OF PROTEOME RESEARCH

Electrostatic interaction between polyglutamylated tubulin and the nexin–dynein regulatory complex regulates flagellar motility

(2017) Tomohiro Kubo et al.   MOLECULAR BIOLOGY OF THE CELL

The N-terminus of IFT46 mediates intraflagellar transport of outer arm dynein and its cargo-adaptor ODA16

(2017) Yuqing Hou et al.   MOLECULAR BIOLOGY OF THE CELL

The Ciliary Transition Zone: Finding the Pieces and Assembling the Gate

(2017) João Gonçalves et al.   MOLECULES AND CELLS

Characterization of a new oda3 allele, oda3-6, defective in assembly of the outer dynein arm-docking complex in Chlamydomonas reinhardtii

(2017) Jason M. Brown et al.   PLoS One

Subnanometre-resolution structure of the doublet microtubule reveals new classes of microtubule-associated proteins

(2017) Muneyoshi Ichikawa et al.   Nature Communications

X-linked primary ciliary dyskinesia due to mutations in the cytoplasmic axonemal dynein assembly factor PIH1D3

(2017) Chiara Olcese et al.   Nature Communications

Chlamydomonas DYX1C1/PF23 is essential for axonemal assembly and proper morphology of inner dynein arms

(2017) Ryosuke Yamamoto et al.   PLoS Genetics

TTC25 Deficiency Results in Defects of the Outer Dynein Arm Docking Machinery and Primary Ciliary Dyskinesia with Left-Right Body Asymmetry Randomization

(2016) Julia Wallmeier et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Myc-binding protein orthologue interacts with AKAP240 in the central pair apparatus of the Chlamydomonas flagella

(2016) Venkatramanan G. Rao et al.   BMC CELL BIOLOGY

Mammalian axoneme central pair complex proteins: Broader roles revealed by gene knockout phenotypes

(2016) Maria E. Teves et al.   Cytoskeleton

The nexin link and B-tubule glutamylation maintain the alignment of outer doublets in the ciliary axoneme

(2016) Lea M. Alford et al.   Cytoskeleton

Transport of the outer dynein arm complex to cilia requires a cytoplasmic protein Lrrc6

(2016) Yasuko Inaba et al.   GENES TO CELLS

Docking-complex-independent alignment ofChlamydomonasouter dynein arms with 24-nm periodicityin vitro

(2016) Toshiyuki Oda et al.   JOURNAL OF CELL SCIENCE

Cilium transition zone proteome reveals compartmentalization and differential dynamics of ciliopathy complexes

(2016) Samuel Dean et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

Posttranslational Modifications of Tubulin and Cilia

(2016) Dorota Wloga et al.   Cold Spring Harbor Perspectives in Biology

Axonemal Dynein Arms

(2016) Stephen M. King   Cold Spring Harbor Perspectives in Biology

The Intraflagellar Transport Machinery

(2016) Michael Taschner et al.   Cold Spring Harbor Perspectives in Biology

Open Sesame: How Transition Fibers and the Transition Zone Control Ciliary Composition

(2016) Francesc R. Garcia-Gonzalo et al.   Cold Spring Harbor Perspectives in Biology

c21orf59/kurly Controls Both Cilia Motility and Polarization

(2016) Kimberly M. Jaffe et al.   Cell Reports

Mutation of Growth Arrest Specific 8 Reveals a Role in Motile Cilia Function and Human Disease

(2016) Wesley R. Lewis et al.   PLoS Genetics

Loss-of-Function GAS8 Mutations Cause Primary Ciliary Dyskinesia and Disrupt the Nexin-Dynein Regulatory Complex

(2015) Heike Olbrich et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Proteomic Analysis of Isolated Ciliary Transition Zones Reveals the Presence of ESCRT Proteins

(2015) Dennis R. Diener et al.   CURRENT BIOLOGY

In SituLocalization of N and C Termini of Subunits of the Flagellar Nexin-Dynein Regulatory Complex (N-DRC) Using SNAP Tag and Cryo-electron Tomography

(2015) Kangkang Song et al.   JOURNAL OF BIOLOGICAL CHEMISTRY

The Oligomeric Outer Dynein Arm Assembly Factor CCDC103 Is Tightly Integrated within the Ciliary Axoneme and Exhibits Periodic Binding to Microtubules

(2015) Stephen M. King et al.   JOURNAL OF BIOLOGICAL CHEMISTRY

Detailed structural and biochemical characterization of the nexin-dynein regulatory complex

(2015) Toshiyuki Oda et al.   MOLECULAR BIOLOGY OF THE CELL

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

(2015) Paulina Urbanska et al.   MOLECULAR BIOLOGY OF THE CELL

DRC3 connects the N-DRC to dynein g to regulate flagellar waveform

(2015) Junya Awata et al.   MOLECULAR BIOLOGY OF THE CELL

FAP206 is a microtubule-docking adapter for ciliary radial spoke 2 and dynein c

(2015) Krishna Kumar Vasudevan et al.   MOLECULAR BIOLOGY OF THE CELL

Late steps in cytoplasmic maturation of assembly-competent axonemal outer arm dynein in Chlamydomonas require interaction of ODA5 and ODA10 in a complex

(2015) Anudariya B. Dean et al.   MOLECULAR BIOLOGY OF THE CELL

α- and β-Tubulin Lattice of the Axonemal Microtubule Doublet and Binding Proteins Revealed by Single Particle Cryo-Electron Microscopy and Tomography

(2015) Aditi Maheshwari et al.   STRUCTURE

CCDC151 Mutations Cause Primary Ciliary Dyskinesia by Disruption of the Outer Dynein Arm Docking Complex Formation

(2014) Rim Hjeij et al.   AMERICAN JOURNAL OF HUMAN GENETICS

A Structural Basis for How Motile Cilia Beat

(2014) Peter Satir et al.   BIOSCIENCE

Insights into the Structure and Function of Ciliary and Flagellar Doublet Microtubules

(2014) Richard Linck et al.   JOURNAL OF BIOLOGICAL CHEMISTRY

FAP20 is an inner junction protein of doublet microtubules essential for both the planar asymmetrical waveform and stability of flagella in Chlamydomonas

(2014) Haru-aki Yanagisawa et al.   MOLECULAR BIOLOGY OF THE CELL

Cooperative binding of the outer arm-docking complex underlies the regular arrangement of outer arm dynein in the axoneme

(2014) Mikito Owa et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

A molecular ruler determines the repeat length in eukaryotic cilia and flagella

(2014) T. Oda et al.   SCIENCE

HEATR2 Plays a Conserved Role in Assembly of the Ciliary Motile Apparatus

(2014) Christine P. Diggle et al.   PLoS Genetics

Zebrafish Ciliopathy Screen Plus Human Mutational Analysis Identifies C21orf59 and CCDC65 Defects as Causing Primary Ciliary Dyskinesia

(2013) Christina Austin-Tse et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Mutations in SPAG1 Cause Primary Ciliary Dyskinesia Associated with Defective Outer and Inner Dynein Arms

(2013) Michael R. Knowles et al.   AMERICAN JOURNAL OF HUMAN GENETICS

ARMC4 Mutations Cause Primary Ciliary Dyskinesia with Randomization of Left/Right Body Asymmetry

(2013) Rim Hjeij et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Mutations in ZMYND10, a Gene Essential for Proper Axonemal Assembly of Inner and Outer Dynein Arms in Humans and Flies, Cause Primary Ciliary Dyskinesia

(2013) Daniel J. Moore et al.   AMERICAN JOURNAL OF HUMAN GENETICS

ZMYND10 Is Mutated in Primary Ciliary Dyskinesia and Interacts with LRRC6

(2013) Maimoona A. Zariwala et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Sperm-Associated Antigen–17 Gene Is Essential for Motile Cilia Function and Neonatal Survival

(2013) Maria Eugenia Teves et al.   AMERICAN JOURNAL OF RESPIRATORY CELL AND MOLECULAR BIOLOGY

Identification of the Outer-Inner Dynein Linker as a Hub Controller for Axonemal Dynein Activities

(2013) Toshiyuki Oda et al.   CURRENT BIOLOGY

Fused (Stk36) is a ciliary protein required for central pair assembly and motile cilia orientation in the mammalian oviduct

(2013) Yoko Inès Nozawa et al.   DEVELOPMENTAL DYNAMICS

The MIA complex is a conserved and novel dynein regulator essential for normal ciliary motility

(2013) Ryosuke Yamamoto et al.   JOURNAL OF CELL BIOLOGY

The N-DRC forms a conserved biochemical complex that maintains outer doublet alignment and limits microtubule sliding in motile axonemes

(2013) Raqual Bower et al.   MOLECULAR BIOLOGY OF THE CELL

The nexin-dynein regulatory complex subunit DRC1 is essential for motile cilia function in algae and humans

(2013) Maureen Wirschell et al.   NATURE GENETICS

DYX1C1 is required for axonemal dynein assembly and ciliary motility

(2013) Aarti Tarkar et al.   NATURE GENETICS

LRRC6 Mutation Causes Primary Ciliary Dyskinesia with Dynein Arm Defects

(2013) Amjad Horani et al.   PLoS One

The Zebrafish Orthologue of the Dyslexia Candidate Gene DYX1C1 Is Essential for Cilia Growth and Function

(2013) Gayathri Chandrasekar et al.   PLoS One

CCDC65 Mutation Causes Primary Ciliary Dyskinesia with Normal Ultrastructure and Hyperkinetic Cilia

(2013) Amjad Horani et al.   PLoS One

Whole-Exome Capture and Sequencing Identifies HEATR2 Mutation as a Cause of Primary Ciliary Dyskinesia

(2012) Amjad Horani et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Loss-of-Function Mutations in LRRC6 , a Gene Essential for Proper Axonemal Assembly of Inner and Outer Dynein Arms, Cause Primary Ciliary Dyskinesia

(2012) Esther Kott et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Exome Sequencing Identifies Mutations in CCDC114 as a Cause of Primary Ciliary Dyskinesia

(2012) Michael R. Knowles et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Splice-Site Mutations in the Axonemal Outer Dynein Arm Docking Complex Gene CCDC114 Cause Primary Ciliary Dyskinesia

(2012) Alexandros Onoufriadis et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Analyses of functional domains within the PF6 protein of the central apparatus reveal a role for PF6 sub-complex members in regulating flagellar beat frequency

(2012) Daniel J. Goduti et al.   Cytoskeleton

Conserved structural motifs in the central pair complex of eukaryotic flagella

(2012) Blanca I. Carbajal-González et al.   Cytoskeleton

Mutations inCCDC39andCCDC40are the Major Cause of Primary Ciliary Dyskinesia with Axonemal Disorganization and Absent Inner Dynein Arms

(2012) Dinu Antony et al.   HUMAN MUTATION

A flagellar A-kinase anchoring protein with two amphipathic helices forms a structural scaffold in the radial spoke complex

(2012) Priyanka Sivadas et al.   JOURNAL OF CELL BIOLOGY

Polarity and asymmetry in the arrangement of dynein and related structures in theChlamydomonasaxoneme

(2012) Khanh Huy Bui et al.   JOURNAL OF CELL BIOLOGY

PF19 encodes the p60 catalytic subunit of katanin and is required for assembly of the flagellar central apparatus in Chlamydomonas

(2012) E. E. Dymek et al.   JOURNAL OF CELL SCIENCE

A FAP46 mutant provides new insights into the function and assembly of the C1d complex of the ciliary central apparatus

(2012) J. M. Brown et al.   JOURNAL OF CELL SCIENCE

Comparative structural analysis of eukaryotic flagella and cilia from Chlamydomonas, Tetrahymena, and sea urchins

(2012) Gaia Pigino et al.   JOURNAL OF STRUCTURAL BIOLOGY

The DPY-30 Domain and Its Flanking Sequence Mediate the Assembly and Modulation of Flagellar Radial Spoke Complexes

(2012) R. Gopal et al.   MOLECULAR AND CELLULAR BIOLOGY

The CSC connects three major axonemal complexes involved in dynein regulation

(2012) Thomas Heuser et al.   MOLECULAR BIOLOGY OF THE CELL

Mutations in axonemal dynein assembly factor DNAAF3 cause primary ciliary dyskinesia

(2012) Hannah M Mitchison et al.   NATURE GENETICS

Cryoelectron tomography reveals doublet-specific structures and unique interactions in the I1 dynein

(2012) Thomas Heuser et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

Regulation of ciliary motility: Conserved protein kinases and phosphatases are targeted and anchored in the ciliary axoneme

(2011) Maureen Wirschell et al.   ARCHIVES OF BIOCHEMISTRY AND BIOPHYSICS

Loss of SPEF2 Function in Mice Results in Spermatogenesis Defects and Primary Ciliary Dyskinesia1

(2011) Anu Sironen et al.   BIOLOGY OF REPRODUCTION

The structural heterogeneity of radial spokes in cilia and flagella is conserved

(2011) Jianfeng Lin et al.   Cytoskeleton

Building Blocks of the Nexin-Dynein Regulatory Complex inChlamydomonasFlagella

(2011) Jianfeng Lin et al.   JOURNAL OF BIOLOGICAL CHEMISTRY

Cryoelectron tomography of radial spokes in cilia and flagella

(2011) Gaia Pigino et al.   JOURNAL OF CELL BIOLOGY

The CSC is required for complete radial spoke assembly and wild-type ciliary motility

(2011) Erin E. Dymek et al.   MOLECULAR BIOLOGY OF THE CELL

The Pcdp1 complex coordinates the activity of dynein isoforms to produce wild-type ciliary motility

(2011) Christen G. DiPetrillo et al.   MOLECULAR BIOLOGY OF THE CELL

Cryo-electron tomography reveals conserved features of doublet microtubules in flagella

(2011) D. Nicastro et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

Tubulin Glutamylation Regulates Ciliary Motility by Altering Inner Dynein Arm Activity

(2010) Swati Suryavanshi et al.   CURRENT BIOLOGY

Tubulin Polyglutamylation Regulates Axonemal Motility by Modulating Activities of Inner-Arm Dyneins

(2010) Tomohiro Kubo et al.   CURRENT BIOLOGY

Bug22p, a Conserved Centrosomal/Ciliary Protein Also Present in Higher Plants, Is Required for an Effective Ciliary Stroke in Paramecium

(2010) C. Laligné et al.   EUKARYOTIC CELL

Pcdp1 is a central apparatus protein that binds Ca2+-calmodulin and regulates ciliary motility

(2010) Christen G. DiPetrillo et al.   JOURNAL OF CELL BIOLOGY

The coiled-coil domain containing protein CCDC40 is essential for motile cilia function and left-right axis formation

(2010) Anita Becker-Heck et al.   NATURE GENETICS

CCDC39 is required for assembly of inner dynein arms and the dynein regulatory complex and for normal ciliary motility in humans and dogs

(2010) Anne-Christine Merveille et al.   NATURE GENETICS

Loss-of-Function Mutations in the Human Ortholog of Chlamydomonas reinhardtii ODA7 Disrupt Dynein Arm Assembly and Cause Primary Ciliary Dyskinesia

(2009) Philippe Duquesnoy et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Deletions and Point Mutations of LRRC50 Cause Primary Ciliary Dyskinesia Due to Dynein Arm Defects

(2009) Niki Tomas Loges et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Proteomic characterization of sperm radial spokes identifies a novel spoke protein with an ubiquitin domain

(2009) Yuhkoh Satouh et al.   FEBS LETTERS

The paraflagellar rod of kinetoplastid parasites: From structure to components and function

(2009) Neil Portman et al.   INTERNATIONAL JOURNAL FOR PARASITOLOGY

The dynein regulatory complex is the nexin link and a major regulatory node in cilia and flagella

(2009) Thomas Heuser et al.   JOURNAL OF CELL BIOLOGY

Central pair apparatus enhances outer-arm dynein activities through regulation of inner-arm dyneins

(2009) Kenji Kikushima   CELL MOTILITY AND THE CYTOSKELETON

Mutations inHydinimpair ciliary motility in mice

(2008) Karl-Ferdinand Lechtreck et al.   JOURNAL OF CELL BIOLOGY

ODA16 aids axonemal outer row dynein assembly through an interaction with the intraflagellar transport machinery

(2008) Noveera T. Ahmed et al.   JOURNAL OF CELL BIOLOGY

Molecular architecture of inner dynein arms in situ inChlamydomonas reinhardtiiflagella

(2008) Khanh Huy Bui et al.   JOURNAL OF CELL BIOLOGY

Ktu/PF13 is required for cytoplasmic pre-assembly of axonemal dyneins

(2008) Heymut Omran et al.   NATURE