Articles

Systematic exploration of the ciliary protein landscape by large-scale affinity proteomics

Authors: K Boldt, J Van Reeuwijk, Q Lu, K Koutroumpas, N Horn, S Van Beersum, Y Texier, TM Nguyen, JR Willer, N Katsanis, F Képès, RB Russell, M Ueffing and R Roepman

Pkd2 affects the architecture of zebrafish left-right organizer

Authors: R Jacinto, P Sampaio, M Roxo-Rosa and S Lopes

Characterisation of homologues of known and putative dynein assembly factors in a Drosophila model

Authors: P zur Lage, D Moore, G Mali, E Hall, P Mill and A Jarman

Structure of outer arm dynein molecule in respiratory cilia suggests an alternative mechanism of force generation

Authors: H Ueno, H Bui, T Ishikawa, Y Imai, T Yamaguchi and T Ishikawa

CSPP-L and EB3 localize to centriolar satellites and are required for satellite-dependent recruitment of ciliopathy proteins to the centrosome

Authors: J Sternemalm, S Geimer, EK Aarnes, KM Frikstad, T Stokke, LB Pedersen and S Patzke

Developing a live cell assay for the centriole-cilium transition in flies

Authors: H Roque and J Raff

A systematic proximity map of the centriole-cilia interface

Authors: J Gonçalves, B Mojarad, G Gupta, E Coyaud, B Raught and L Pelletier

The role of Rabconnectin3a in cilia length regulation

Authors: B Tavares, P Pintado and SS Lopes

Paramecium tetraurelia basal body unit isolation for Cryo-electron tomography studies

Authors: S Trépout, M Lemullois, P Guichard, F Koll, A Aubusson-Fleury, J Beisson, J Cohen, S Marco and AM Tassin

Cep164, but not EB1, is critical for centriolar localization of TTBK2 and its function in ciliogenesis

Authors: Toshiaki Oda, Shuhei Chiba, Tomoaki Nagai and Kensaku Mizuno

Number of nexin links detectable at standard electron microscopy of normal human nasal cilia and at nexin link deficiency

Authors: U Stenram and C Cramnert

A reference to assess cilium phenotype in ciliopathy patients

Authors: M Oud, H Arts and R Roepman

Meckel-Gruber syndrome patient cells exhibit alterations in cell-substrate interaction, deformation response, and gene expression consistent with defects leading to liver fibrosis

Authors: B Meadows and H Dawe

Identification of human mutations in TRAF3IP1 in patients with nephronophthisis and retinal degeneration

Authors: A Becker-Heck, A Bizet, R Ryan, P Krug, E Filhol, B Linghu, E Oakeley, F Serluca, F Legendre, N Dörner, MC Lasbennes, J Duca, F Yang, A Damask, L Klickstein, M Labow…

Recruitment of IFT proteins during flagellum construction in Trypanosoma brucei

Authors: E Bertiaux, P Bastin and B Rotureau

FPC4: a new cytoskeletal component in T.brucei

Authors: A Albisetti, C Florimond, A Sahin, M Eggenspieler, O Cingal, D Robinson and M Bonhivers

Transgenic tools for proteomic analysis of ciliary transport

Authors: X Fang, U Jokopii and J Malicki

Evidence for a role of the ciliopathy protein MKS1 in cell polarity

Authors: M Collado-Hilly, C Fisch, B Desforges, J Jerber, L Combettes, C Campillo and P Dupuis-Williams

Using the talpid ² as novel model for determining the cellular and molecular etiology of Oral-facial-digital syndrome

Authors: EN Schock, CF Chang, JN Struve, J Chang and SA Brugmann

Does stopping left-right nodal flow mirror impaired mechanosensation in the left-right organizer?

Authors: R Jacinto, P Sampaio, M Roxo-Rosa and SS Lopes

FOXA2 controls Pkd1l1 expression in the mouse node during left-right determination

Authors: D Leordean, D Grimes, J Keynton, J Maier, B Harfe, M Benson, A Gray, S Bhattacharya and D Norris

NrCAM modulates sonic hedgehog signalling by controlling smoothened translocation in the cilium

Authors: B Basu, O Weeranantanapan, D Xenaki and A Furley

Effects of the novel formin INF1 on ciliogenesis

Authors: S Copeland, C Tenneson, W Kulacz and J Copeland

Yeast as a simple eukaryotic model to study human diseases linked to membrane trafficking

Authors: J De Craene, S Bär, J Laporte, V Marion, H Dollfus and S Friant

Gene discovery for motile cilia disorders: mutation spectrum in primary ciliary dyskinesia and discovery of mutations in CCDC151

Authors: A Onoufriadis, R Hjeij, CM Watson, CE Slagle, NT Klena, GW Dougherty, M Kurkowiak, NT Loges, CP Diggle, NF Morante, GC Gabriel, KL Lemke, Y Li, P Pennekamp, T Menchen, JK Marthin…

OFD1 and VFL3/CCDC61 in basal body positioning and docking in Paramecium

Authors: H Bengueddach, M Lemullois, J Cohen, AM Tassin, A Aubusson-Fleury and F Koll

MARK4 contributes to cilia formation by regulating the degradation of inhibitory OFD1 from the centriolar satellites

Authors: R Carvalho, P Wiedemann and G Pereira

Integrative approaches to investigate the structure and assembly of Trypanosoma brucei BILBO1, a multidomain cytoskeletal protein at the flagellar pocket collar

Authors: K Vidilaseris, B Morriswood and G Dong

Crumbs proteins control ciliogenesis and centrosome organization: what about the mechanism?

Authors: M Barthelemy-Requin, JP Chauvin, CL Baron-Gaillard, P Rashbass, D Massey-Harroche and A Le Bivic

Developing stem cell therapy for retinal dystrophies

Authors: JC Sowden

Pericentrin interacts with KASH domain-containing protein Syne-2

Authors: N Falk, K Kessler, J Glöckner, K Boldt, M Ueffing, R Roepman, C Thiel, JH Brandstätter and A Gießl

Uncoupling flagellum formation and maintenance

Authors: C Fort, J Santi-Rocca and P Bastin

A novel form of PCD that impacts nodal, but not tracheal cilia

Authors: J Keynton, E Adams, K Riley, N Powles-Glover, K Shinohara, J Lucas, P Lackie and D Norris

PDZD7 connects the Usher protein complex to the intraflagellar transport machinery

Authors: E De Vrieze, L Creemers, N Sorusch, M Gorris, J Van Reeuwijk, S Van Beersum, B Knapp, K Boldt, M Ueffing, R Roepman, E Van Wijk, U Wolfram and H Kremer

DNA Methylation and Ciliopathies: a way to be explored

Authors: M Alvarez-Satta, L De Chiara, S Castro-Sánchez and D Valverde

Investigation into the Importance of genes encoding ciliary proteins in congenital heart disease using whole exome sequencing

Authors: V Hartill, C Logan, DA Parry, K Szymanska, K Ashcroft, K English, K Prescott, A Dobbie, S Barwick, C Bennett, J Goodship, E Sheridan and C Johnson

Mutations of IFT81, encoding an IFT-B core protein, as a rare cause of a ciliopathy

Authors: I Perrault, J Halbritter, J Porath, X Gerard, D Braun, H Gee, H Fathy, S Saunier, V Cormier-Daire, S Thomas, T Attié-Bitach, N Boddaert, M Taschner, M Schueler, E Lorentzen, R Lifton…

The regulation of mechanosensory motile cilium formation

Authors: A Jarman, P zur Lage, G Mali and P Mill

The study of a total and two hypothalamic-specific BBS10 knockout models highlights the importance of systemic inactivation in the obese phenotype in Bardet Biedl Syndrome

Authors: M Scerbo, F Costa, C Obringer, H Dollfus and V Marion

9+2 to 9+0 axoneme conversion in Leishmania

Authors: R Wheeler, K Gull and E Gluenz

Kinesin-3 motor protein KIF13B localizes to centrosomes and primary cilia and regulates ciliary length and signaling

Authors: K B Schou, JB Mogensen, A Aleliunaite, S Morthorst, I Veland, S Christensen and LB Pedersen

Phenotypic variability in Meckel-Gruber syndrome

Authors: S Darouich, I Bettaieb, M Khila, T Kitova, D Chelli, E Sfar, MB Channoufi, H Reziga, S Gaigi and A Masmoudi

PI3K-C2α regulates Polycystin-2 ciliary entry to prevent kidney cyst formation

Authors: JP Margaria, I Franco, A Ranghino, D Monteyne, M Chiaravalli, M Pema, C Campa, F Gulluni, D Perez-Morga, G Merlo, A Boletta and E Hirsch

The Drosophila homologue of Rootletin is required for mechanosensory function and ciliary rootlet formation in chordotonal sensory neurons

In vertebrates, rootletin is the major structural component of the ciliary rootlet and is also part of the tether linking the centrioles of the centrosome. Various functions have been ascribed to the rootlet, ...

Authors: Katarzyna Styczynska-Soczka and Andrew P. Jarman

Non-invasive sources of cells with primary cilia from pediatric and adult patients

Ciliopathies give rise to a multitude of organ-specific pathologies; obtaining relevant primary patient material is useful for both diagnostics and research. However, acquisition of primary ciliated cells from...

Authors: Henry Ajzenberg, Gisela G. Slaats, Marijn F. Stokman, Heleen H. Arts, Ive Logister, Hester Y. Kroes, Kirsten Y. Renkema, Mieke M. van Haelst, Paulien A. Terhal, Iris A. van Rooij, Mandy G. Keijzer-Veen, Nine V. Knoers, Marc R. Lilien, Michael A. Jewett and Rachel H. Giles

The primary cilium functions as a mechanical and calcium signaling nexus

The primary cilium is an antenna-like, nonmotile structure that extends from the surface of most mammalian cell types and is critical for chemosensing and mechanosensing in a variety of tissues including carti...

Authors: Kristen L Lee, Marie D Guevarra, An M Nguyen, Mardonn C Chua, Yingxiao Wang and Christopher R Jacobs

Calcium sensors of ciliary outer arm dynein: functions and phylogenetic considerations for eukaryotic evolution

The motility of eukaryotic cilia and flagella is modulated in response to several extracellular stimuli. Ca²⁺ is the most critical intracellular factor for these changes in motility, directly acting on the axonem...

Authors: Kazuo Inaba

The more we know, the more we have to discover: an exciting future for understanding cilia and ciliopathies

The Cilia 2014 conference was organised by four European networks: the Ciliopathy Alliance, the Groupement de Recherche CIL, the Nordic Cilia and Centrosome Network and the EU FP7 programme SYSCILIA. More than...

Authors: Alexandre Benmerah, Bénédicte Durand, Rachel H Giles, Tess Harris, Linda Kohl, Christine Laclef, Sigolène M Meilhac, Hannah M Mitchison, Lotte B Pedersen, Ronald Roepman, Peter Swoboda, Marius Ueffing and Philippe Bastin

Ultrastructural visualization of trans-ciliary rhodopsin cargoes in mammalian rods

Cilia are vital to various cellular and sensory functions. The pathway by which ciliary membrane proteins translocate through the transition zone is not well understood. Direct morphological characterization o...

Authors: Jen-Zen Chuang, Ya-Chu Hsu and Ching-Hwa Sung

Cryo-electron tomography of motile cilia and flagella

Cryo-electron tomography has been a valuable tool in the analysis of 3D structures of cilia at molecular and cellular levels. It opened a way to reconstruct 3D conformations of proteins in cilia at 3-nm resolu...

Authors: Takashi Ishikawa