Cilia articles within Nature Communications

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  • Article
    | Open Access

    The precise regulatory mechanisms controlling ciliary Hedgehog signaling remain incomplete. Here, the authors use ciliary proteomics to reveal that Numb facilitates the endocytosis of the receptor Ptch1 from the ciliary pocket, thereby enabling activation of Hedgehog signaling.

    • Xiaoliang Liu
    • , Patricia T. Yam
    •  & Xuecai Ge

  • Article
    | Open Access

    Intraflagellar transport is essential for the formation and function of cilia. Here, the authors use single-molecule imaging in live C. elegans to show that transport trains are formed by the sequential attachment of proteins before departing into the cilium.

    • Aniruddha Mitra
    • , Elizaveta Loseva
    •  & Erwin J. G. Peterman

  • Article
    | Open Access

    Centrioles are characterized by an atypical triplet microtubule structure. Here, the authors discover that the ciliopathy protein HYLS1 promotes the assembly of triplet microtubules within human centrioles.

    • Yutaka Takeda
    • , Takumi Chinen
    •  & Daiju Kitagawa

  • Article
    | Open Access

    Microtubule inner proteins (MIPs) contribute to species-specific motility characteristics but are largely unstudied. Here, the authors combine functional, structural and proteomic analysis in T. brucei to advance fundamental understanding of MIP assembly and identify trypanosome-specific MIPs required for motility.

    • Michelle M. Shimogawa
    • , Angeline S. Wijono
    •  & Kent L. Hill

  • Article
    | Open Access

    Exposure to irreparable stresses induces transient ciliogenesis, enabling communication with PML-NBs via a FBF1 pathway to trigger senescence in mammalian cells. Fbf1 ablation reduces senescence and associated health decline in mice, highlighting cilia as a promising senotherapy target.

    • Xiaoyu Ma
    • , Yingyi Zhang
    •  & Jinghua Hu

  • Article
    | Open Access

    Motile cilia beat in a defined direction to orchestrate developmental programs, but also to execute janitorial tasks such as clearing airways. Here they show that motile cilia of the Xenopus epidermis are anchored to microridge-like membrane protrusions to maintain their directionality.

    • Takayuki Yasunaga
    • , Johannes Wiegel
    •  & Gerd Walz

  • Article
    | Open Access

    Primary cilia are key sensory organelles whose dysfunction leads to ciliopathy disorders such as Bardet-Biedl syndrome (BBS). Here they identify a role for Bbs1 in lipid homeostasis of photoreceptor outer segments in zebrafish, which may contribute to vision loss in patients with Bardet-Biedl syndrome.

    • Markus Masek
    • , Christelle Etard
    •  & Ruxandra Bachmann-Gagescu

  • Article
    | Open Access

    Cilia are microtubule-based organelles containing proteins transported from the cell body. Here, the authors show that the multicilia of mouse ependymal cells contain ribosomal components, tubulin mRNA,18 S rRNA and nascent tubulin peptides, suggesting local translation in the ciliary compartment.

    • Kai Hao
    • , Yawen Chen
    •  & Xueliang Zhu

  • Article
    | Open Access

    The γ-tubulin ring complex (γTuRC) nucleates microtubules at the centrosome, but how this function is related to γTuRC subcentrosomal distribution is unclear. Here the authors show that γTuRC in the centriole lumen has a nucleation-independent role in centriole integrity and cilium assembly.

    • Nina Schweizer
    • , Laurence Haren
    •  & Jens Lüders

  • Article
    | Open Access

    Ciliary beating is mediated by the axonemal central pair microtubules, though how these non-centrosomal microtubules form is poorly understood. Here the authors show that a trio of proteins act cooperatively to initiate central microtubule formation in mammals.

    • Hao Liu
    • , Jianqun Zheng
    •  & Xiumin Yan

  • Article
    | Open Access

    How the developing skin epidermis is transformed from a simple single-layered epithelium to a complex and stratified barrier is still an open question. Here, the authors provide a model based on high proliferation and delamination of the keratinocyte progenitors that support the stratification process.

    • Mareike Damen
    • , Lisa Wirtz
    •  & Hisham Bazzi

  • Article
    | Open Access

    Sensory perception and metabolic homeostasis are known to deteriorate with ageing, while mechanisms underlying their deterioration remain poorly understood. Here, the authors demonstrate that decrease of intraflagellar transport in the cilia of sensory neurons impairs sensory perception and metabolism in ageing C. elegans.

    • Yincong Zhang
    • , Xiaona Zhang
    •  & Yidong Shen

  • Article
    | Open Access

    Multicilia are complex, and how they achieve accurate assembly is unclear. Here, the authors show that fibrogranular materials condense into spherical cores and function in multicilia formation by tightly associating with deuterosomes and concentrating specific proteins to promote proper assembly.

    • Huijie Zhao
    • , Qingxia Chen
    •  & Xueliang Zhu

  • Article
    | Open Access

    Axonemal dyneins are tethered to doublet microtubules inside cilia to drive ciliary beating but the mechanisms regulating their localization and function are poorly understood. Here authors report a cryo-EM reconstruction of a three-headed axonemal dynein natively bound to doublet microtubules isolated from cilia which provides a framework to understand the roles of individual subunits.

    • Travis Walton
    • , Hao Wu
    •  & Alan Brown

  • Article
    | Open Access

    Most cells possess sensory cilia, which need to be gated properly. Here the authors show that the C. elegans proteins TALP-3 and ANKR-26 coordinate cilia gating in the context of transition fibers and that this mechanism is conserved in mammalian cells and likely implicated in certain ciliopathies.

    • Hao Yan
    • , Chuan Chen
    •  & Qing Wei

  • Article
    | Open Access

    Microtubule asters originate from centrosomes but their role during interphase remains largely unknown. Here, the authors find that microtubule asters anchored by previously-uncharacterized FSD1 play a role in ciliogenesis by maintaining the dynamic localization of centriolar satellites.

    • Hai-Qing Tu
    • , Xuan-He Qin
    •  & Hui-Yan Li

  • Article
    | Open Access

    Ependymal ciliary beating contributes to the flow of cerebrospinal fluid in the brain ventricles and these cilia resist the flow forces. Here the authors show that the assembly of a dense actin network around the centrioles is induced by cilia beating to protect centrioles against the shear stress generated by ciliary motility.

    • Alexia Mahuzier
    • , Asm Shihavuddin
    •  & Nathalie Delgehyr

  • Article
    | Open Access

    Distal appendages (DAPs) at the cilia base mediate membrane docking during ciliogenesis. Here the authors use super-resolution microscopy to map 16 centriole distal end components, revealing the structure of the backbone of the DAP, as well as a previously undescribed distal appendage matrix.

    • T. Tony Yang
    • , Weng Man Chong
    •  & Jung-Chi Liao

  • Article
    | Open Access

    Tubulin post-translational modifications (PTMs) occur spatiotemporally throughout cells, therefore assessing the physiological roles in specific subcellular compartments has been challenging. Here the authors develop a method to rapidly deplete tubulin glutamylation inside the primary cilia by targeting an engineered deglutamylase to the axoneme.

    • Shi-Rong Hong
    • , Cuei-Ling Wang
    •  & Yu-Chun Lin

  • Article
    | Open Access

    The trichoplein-Aurora A pathway inhibits ciliogenesis in proliferating cells. Here the authors EGFR-mediated phosphorylation of the deubiquitinating enzyme USP8 leads to its activation, and this suppresses trichoplein degradation, allowing inhibition of ciliogenesis.

    • Kousuke Kasahara
    • , Hiromasa Aoki
    •  & Masaki Inagaki

  • Article
    | Open Access

    Asthenozoospermia is a major cause of male infertility, and multiple morphological abnormalities of the flagella (MMAF) is a particularly severe form. Here, using whole-exome sequencing of 78 MMAF patients, the authors identify mutations in two WDR proteins, CFAP43 and CFAP44, and confirm that these proteins are required for flagellogenesis in mouse and Trypanosoma brucei.

    • Charles Coutton
    • , Alexandra S. Vargas
    •  & Pierre F. Ray

  • Article
    | Open Access

    Multiciliogenesis requires activation of transcriptional and protein assembly programs; however, the mechanisms that initiate the formation of these multiprotein complexes are unclear. Here the authors show that after inducing centriole biogenesis genes, the transcription factor E2f4 is required in the cytoplasm for assembly and nucleation of deuterosomes.

    • Munemasa Mori
    • , Renin Hazan
    •  & Wellington V. Cardoso

  • Article
    | Open Access

    Cilia are hair-like appendages involved in cell motility and sensory reception. Here, the authors report a high resolution cryo-EM structure of the microtubule doublet from motile cilia and identify microtubule inner proteins (MIPs) bound to the inner surface of the doublet that appear to stabilize its structure.

    • Muneyoshi Ichikawa
    • , Dinan Liu
    •  & Khanh Huy Bui

  • Article
    | Open Access

    The centriole is an organelle composed of rings of SAS-6 proteins that form a cartwheel structure. Here the authors develop a cell-free system to examine core cartwheel assembly ofC. reinhardtiiproteins and discover that CrSAS-6 has autonomous properties that facilitates self-organized stacking of pairs of rings.

    • P. Guichard
    • , V. Hamel
    •  & P. Gönczy

  • Article
    | Open Access

    Cytoplasmic dynein 2 drives retrograde intraflagellar transport but little is known about its dynamics. Here the authors use fluorescence microscopy to track labelled dynein 2 inC. elegansat the single-molecule level and report diffusion at the ciliary base, and pausing and directional switches along the cilium.

    • Jona Mijalkovic
    • , Bram Prevo
    •  & Erwin J. G. Peterman

  • Article
    | Open Access

    Centrioles and cilia are microtubule-based structures of defined architecture, but what regulates this architecture is not clear. Here the authors discover that centrosomal-P4.1-associated-protein (CPAP) binds the α/β-tubulin dimer and licenses it for tubulin delivery contributing to centriolar/ciliary length and architecture control.

    • Xiangdong Zheng
    • , Anand Ramani
    •  & Haitao Li

  • Article
    | Open Access

    PDE6δ regulates the sorting of prenylated cargo proteins. Here Fansa et al. propose that the affinity of the interaction between PDE6δ and its cargo protein determines whether they are released by cytoplasmic or cilia-specific release factors ultimately determining their subcellular localization.

    • Eyad Kalawy Fansa
    • , Stefanie Kristine Kösling
    •  & Shehab Ismail

  • Article
    | Open Access

    Whether basal bodies are pre-committed to form 9+2 motile or 9+0 sensory axonemes and whether interconversion occurs between the two types of axonemes is not clear. Here, the authors used the unicellular eukaryote Leishmania as a model system to demonstrate that 9+0 axonemes can be formed de novoor by restructuring of 9+2 axonemes.

    • R. J. Wheeler
    • , E. Gluenz
    •  & K. Gull

  • Article
    | Open Access

    The formation of motile cilia is regulated by Dishevelled (DVL), a central component in WNT signalling and planar cell polarity (PCP). Here the authors identify DVL as a novel substrate of the phosphatase PTEN, coupling PTEN to cilia dynamics and PCP.

    • Iryna Shnitsar
    • , Mikhail Bashkurov
    •  & Miriam Barrios-Rodiles

  • Article
    | Open Access

    Most differentiated mammalian cells assemble a primary cilium, which serves as a cellular ‘antenna’ for sensing and responding to the extracellular environment. Here the authors show that Nek2-mediated phosphorylation of Kif24 further promotes the loss of primary cilia, triggered by Aurora A and HDAC6 on cell cycle re-entry.

    • Sehyun Kim
    • , Kwanwoo Lee
    •  & Brian David Dynlacht

  • Article |

    The ciliary margin of the eye functions as a source of multipotent progenitor cells in certain organisms but whether it plays this role in humans has not been easy to study. Here the authors culture human embryonic stem cells that self-organize into retinal tissue, and show that ciliary margin-like growth zones emerge from the developing human retinal tissue and contain stem cell niches.

    • Atsushi Kuwahara
    • , Chikafumi Ozone
    •  & Yoshiki Sasai

  • Article
    | Open Access

    Loss of olfactory function is one of the many symptoms of the ciliopathy Bardet–Biedl syndrome. Williams et al. show that Bardet–Biedl proteins are components of intraflagellar transport particles within cilia, and directly visualize their trafficking in native mammalian olfactory neurons.

    • Corey L. Williams
    • , Jeremy C. McIntyre
    •  & Jeffrey R. Martens

  • Article |

    Cilia are hair-like protuberances on the cellular surface that have been implicated in sensing and signal transduction. Here Gerdes et al. show cilia are involved in insulin signalling and secretion in pancreatic β-cells of rodents, and suggest that ciliary dysfunction could contribute to type 2 diabetes.

    • Jantje M. Gerdes
    • , Sonia Christou-Savina
    •  & Per-Olof Berggren

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