Ciliogenesis articles within Nature Communications

Featured

• Article
  21 July 2023 | Open Access

  MOF-mediated histone H4 Lysine 16 acetylation governs mitochondrial and ciliary functions by controlling gene promoters

  Here the authors show that epigenetic regulation through the histone acetyltransferase MOF and the acetylation of histone H4 lysine 16 affect essential functions in the mitochondria and primary cilia. This regulation is important to promote epidermal differentiation and hair follicle formation.

  • Dongmei Wang
  • , Haimin Li
  •  & Rui Yi

• Article
  24 June 2023 | Open Access

  Hedgehog signaling via its ligand DHH acts as cell fate determinant during skeletal muscle regeneration

  Successful skeletal muscle regeneration relies on the interplay of multiple cell populations. Here, the authors describe how ciliary Hedgehog signaling coordinates the intercellular crosstalk required to balance wound healing and fatty fibrosis.

  • Alessandra M. Norris
  • , Ambili Bai Appu
  •  & Daniel Kopinke

• Article
  27 March 2023 | Open Access

  Single-molecule localization microscopy reveals the ultrastructural constitution of distal appendages in expanded mammalian centrioles

  The authors have combined direct stochastic optical reconstruction microscopy with expansion microscopy to describe the 3-dimensional molecular organization of centriolar distal appendages.

  • Ting-Jui Ben Chang
  • , Jimmy Ching-Cheng Hsu
  •  & T. Tony Yang

• Article
  06 March 2023 | Open Access

  Mucociliary Wnt signaling promotes cilia biogenesis and beating

  Motile cilia are thought of as signaling-inert nanomotors. Here, the authors show that motile cilia transduce a Wnt-protein phosphatase 1 signal that regulates cilia biogenesis and ciliary beating in Xenopus embryos and in human airway epithelia.

  • Carina Seidl
  • , Fabio Da Silva
  •  & Christof Niehrs

• Article
  13 February 2023 | Open Access

  Repair of airway epithelia requires metabolic rewiring towards fatty acid oxidation

  Airway epithelial repair, a key process in the recovery from lung injury, requires a metabolic shift from glycolysis to fatty acid oxidation (FAO). Pharmacological FAO promotion enhances epithelial differentiation, suggesting new therapeutic options.

  • Stefania Crotta
  • , Matteo Villa
  •  & Andreas Wack

• Article
  19 April 2022 | Open Access

  Microridge-like structures anchor motile cilia

  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
  04 October 2021 | Open Access

  Wdr47, Camsaps, and Katanin cooperate to generate ciliary central microtubules

  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
  27 September 2021 | Open Access

  Bardet-Biedl syndrome proteins modulate the release of bioactive extracellular vesicles

  Extracellular vesicles (EV) are known to be released from the primary cilium, but the role ciliary proteins play in EV biogenesis remains unexplored. Here, the authors demonstrate increased secretion of small EVs with altered cargo composition from cells with known ciliarelated mutations. Wnt related molecules made up a majority of altered cargo

  • Ann-Kathrin Volz
  • , Alina Frei
  •  & Helen L. May-Simera

• Article
  20 October 2020 | Open Access

  Siah2 integrates mitogenic and extracellular matrix signals linking neuronal progenitor ciliogenesis with germinal zone occupancy

  In neural development, progenitors transition from a proliferative to a differentiated state. Here, the authors show that cerebellar granule neurons retract primary cilia as they exit their proliferative niche upon decreased ECM engagement, enabling radial migration due to loss of Shh sensitivity.

  • Taren Ong
  • , Niraj Trivedi
  •  & David J. Solecki

• Article
  04 May 2020 | Open Access

  TALPID3 and ANKRD26 selectively orchestrate FBF1 localization and cilia gating

  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
  20 April 2020 | Open Access

  Wnt-PLC-IP₃-Connexin-Ca²⁺ axis maintains ependymal motile cilia in zebrafish spinal cord

  Ependymal cells are supporting cells in the central nervous system. Here the authors elucidate a signalling axis in zebrafish spinal cord ependymal cells that is important for motile cilia assembly and maintenance, demonstrating that it depends on intercellular propagation of calcium ions via connexin 43.

  • Jun Zhang
  • , Gopalakrishnan Chandrasekaran
  •  & Seok-Yong Choi

• Article
  06 September 2019 | Open Access

  Ciliary exclusion of Polycystin-2 promotes kidney cystogenesis in an autosomal dominant polycystic kidney disease model

  The molecular role of ciliary Polycystin-2 (PC2) in cyst formation and polycystic kidney disease (ADKPD) is unclear. Here, the authors identify a PC2 mutant lacking ciliary localisation but with active Ca2+ channel function in mice, that is sufficient to generate an ADPKD phenotype.

  • Rebecca V. Walker
  • , Jennifer L. Keynton
  •  & Dominic P. Norris

• Article
  23 April 2019 | Open Access

  Primary cilia regulate hematopoietic stem and progenitor cell specification through Notch signaling in zebrafish

  Haematopoietic stem and progenitor cells (HSPCs) produce all blood lineages and arise from the haemogenic endothelium (HE) during embryogenesis. Here the authors show that genes specific to cilia formation are required for HSPC development in the HE in zebrafish through transduction of Notch signal.

  • Zhibin Liu
  • , Haiqing Tu
  •  & Feng Liu

• Article
  27 February 2019 | Open Access

  Secreted metalloproteases ADAMTS9 and ADAMTS20 have a non-canonical role in ciliary vesicle growth during ciliogenesis

  Ciliogenesis is a complex process requiring hundreds of molecules, although few secreted proteins have been implicated. Here, the authors show that the secreted metalloproteases ADAMTS9 and ADAMTS20 intracellularly regulate ciliogenesis from unique periciliary vesicles with proteolytic activity.

  • Sumeda Nandadasa
  • , Caroline M. Kraft
  •  & Suneel S. Apte

• Article
  11 December 2018 | Open Access

  Microtubule asters anchored by FSD1 control axoneme assembly and ciliogenesis

  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
  17 August 2018 | Open Access

  Axoneme polyglutamylation regulated by Joubert syndrome protein ARL13B controls ciliary targeting of signaling molecules

  The small GTPase ARL13B is mutated in the human ciliopathy Joubert syndrome. Here the authors show that ARL13B and the RAB11 effector FIP5 promote import of tubulin glutamylase into cilia and as such axoneme polyglutamylation, which is demonstrated to be required for cilia signaling.

  • Kai He
  • , Xiaoyu Ma
  •  & Jinghua Hu

• Article
  22 May 2018 | Open Access

  Super-resolution architecture of mammalian centriole distal appendages reveals distinct blade and matrix functional components

  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
  25 April 2018 | Open Access

  Uncovering inherent cellular plasticity of multiciliated ependyma leading to ventricular wall transformation and hydrocephalus

  Multiciliated ependymal cells (ECs) in the mammalian brain are glial cells facilitating cerebral spinal fluid movement. This study describes an inherent cellular plasticity of ECs as maintained by Foxj1 and IKK2 signaling, and shows resulting hydrocephalus when EC de-differentiation is triggered.

  • Khadar Abdi
  • , Chun-Hsiang Lai
  •  & Chay T. Kuo

• Article
  04 July 2017 | Open Access

  Cytoplasmic E2f4 forms organizing centres for initiation of centriole amplification during multiciliogenesis

  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
  09 January 2017 | Open Access

  Bi- and uniciliated ependymal cells define continuous floor-plate-derived tanycytic territories

  Ependymal cells lining the adult brain ventricles are comprised of multiciliated cells and a rare subpopulation with two cilia (E2 cells) whose origin and function remain unknown. Here the authors find E2 cells in the 3rd ventricle of mice and humans, along with a third ependymal cell type with only a primary cilium, and provide details of their marker profile and developmental origins.

  • Zaman Mirzadeh
  • , Yael Kusne
  •  & Arturo Alvarez-Buylla

• Article
  24 November 2016 | Open Access

  A mutation in VPS15 (PIK3R4) causes a ciliopathy and affects IFT20 release from the cis-Golgi

  VPS15 is known as a VPS34-associated protein that functions in intracellular trafficking and autophagy. Here the authors identify a role for VPS15 in ciliopathy and ciliary phenotypes, and show that it interacts with GM130 and functions in IFT20-dependent cis-Golgi to cilium trafficking.

  • Corinne Stoetzel
  • , Séverine Bär
  •  & Hélène Dollfus

• Article
  24 August 2016 | Open Access

  Severe NDE1-mediated microcephaly results from neural progenitor cell cycle arrests at multiple specific stages

  Human mutations in the NDE1 gene have been associated with cortical malformations and severe microcephaly. Here, the authors show in embryonic rat brains that NDE1-depleted neural progenitors arrest at three specific cell cycle stages before mitosis, resulting in a severe decrease in neurogenesis.

  • David J. Doobin
  • , Shahrnaz Kemal
  •  & Richard B. Vallee

• Article
  18 August 2016 | Open Access

  The hydrolethalus syndrome protein HYLS-1 regulates formation of the ciliary gate

  Transition fibres (TFs) and the transition zone (TZ) are basal ciliary structures thought to act as a gate to regulate protein transport. Here the authors show that the C. elegansorthologue of hydrolethalus protein 1, HYLS-1, disrupts both structures and compromises the trafficking of cargo through the ciliary gate.

  • Qing Wei
  • , Yingyi Zhang
  •  & Jinghua Hu

• Article
  26 February 2016 | Open Access

  Phosphatidylinositol phosphate kinase PIPKIγ and phosphatase INPP5E coordinate initiation of ciliogenesis

  The primary cilium is essential for embryonic development and tissue pattern formation. Here the authors show that PIPKIγ localizes to the basal body of the primary cilium and cooperates with the Joubert Syndrome associated protein INPP5E to regulate the initiation of ciliogenesis.

  • Qingwen Xu
  • , Yuxia Zhang
  •  & Kun Ling

• Article
  18 September 2015 | Open Access

  miR-34/449 control apical actin network formation during multiciliogenesis through small GTPase pathways

  MicroRNAs of the miR-34/449 family initiate formation of multiciliated cells through the suppression of cell cycle genes and Notch. Here the authors show that miR-34/449 also regulate the assembly of an apical actin network necessary for basal body anchoring by regulating the expression of R-Ras.

  • Benoît Chevalier
  • , Anna Adamiok
  •  & Brice Marcet

• Article
  24 July 2015 | Open Access

  Developmental disruptions underlying brain abnormalities in ciliopathies

  Primary cilia are essential conveyors of signals underlying major cellular functions but their role in brain development is not completely understood. Here the authors compiled a shRNA library targeting ciliopathy genes known to cause brain disorders, and used it to query how ciliopathy genes affect distinct stages of mouse cortical development.

  • Jiami Guo
  • , Holden Higginbotham
  •  & E.S. Anton

• Article | 07 April 2015

  Actin remodelling factors control ciliogenesis by regulating YAP/TAZ activity and vesicle trafficking

  Destabilizing the actin cytoskeleton induces the formation of primary cilia, but the mechanism is unknown. Here, Kim et al.show that activity of the transcriptional coactivators YAP/TAZ is regulated by actin remodelling factors, and YAP/TAZ activity correlates with ciliogenesis.

  • Jongshin Kim
  • , Haiin Jo
  •  & Joon Kim

• Article | 31 March 2015

  ERK7 regulates ciliogenesis by phosphorylating the actin regulator CapZIP in cooperation with Dishevelled

  The actin regulator CapZIP has been shown to regulate ciliogenesis in a phosphorylation-dependent manner. Miyatake et al.show that the atypical MAP kinase ERK7 promotes ciliogenesis by phophorylating CapZIP, and that interactions between both proteins and Dishevelled are required for CapZIP phosphorylation.

  • Koichi Miyatake
  • , Morioh Kusakabe
  •  & Eisuke Nishida

• Article | 04 November 2014

  The MST1/2-SAV1 complex of the Hippo pathway promotes ciliogenesis

  Although much is known about the structural and trafficking molecules involved in generation of primary cilia, the signalling proteins that regulate ciliogenesis are poorly defined. Here, Kim et al. identify the MST1/2-SAV1 complex, a core component of the Hippo pathway, as a key regulator of ciliogenesis in cells and zebrafish.

  • Miju Kim
  • , Minchul Kim
  •  & Dae-Sik Lim

• Article
  01 October 2014 | Open Access

  Ubiquitin-proteasome system controls ciliogenesis at the initial step of axoneme extension

  Biogenesis of the primary cilium begins after cell cycle exit, but the regulatory steps for its formation are poorly defined. Here the authors show that proteasome-mediated removal of the ciliogenesis inhibitor, trichoplein, from mother centrioles initiates the first step of ciliogenesis.

  • Kousuke Kasahara
  • , Yoshitaka Kawakami
  •  & Masaki Inagaki

• Article | 19 August 2014

  The deubiquitinating enzyme CYLD controls apical docking of basal bodies in ciliated epithelial cells

  Mutations in the deubiquitinase gene CYLD are associated with cylindromatosis, a disease characterized by the development of skin appendage tumours. Eguether et al.discover that CYLD localizes to centrosomes and is required for basal body migration and docking, providing insight into its tumour suppressor activity.

  • Thibaut Eguether
  • , Maria A. Ermolaeva
  •  & Anne-Marie Tassin

• Article | 20 June 2014

  Ciliopathy-associated gene Cc2d2a promotes assembly of subdistal appendages on the mother centriole during cilia biogenesis

  Mutations in the centrosome-cilia gene, Cc2d2a, result in Meckel and Joubert syndromes in humans. By creating Cc2d2a-mutant mice, Veleri et al.show that this gene encodes a component of subdistal appendages; ciliary structures thought to be required to anchor cilia to the microtubule network.

  • Shobi Veleri
  • , Souparnika H. Manjunath
  •  & Anand Swaroop