Primary Cilia as Signaling Hubs in Health and Disease

The cholangiocyte primary cilium in health and disease

(2018) Adrian P. Mansini et al.   BIOCHIMICA ET BIOPHYSICA ACTA-MOLECULAR BASIS OF DISEASE

A highlight on Sonic hedgehog pathway

(2018) Gabriela Basile Carballo et al.   Cell Communication and Signaling

TSC1 and TSC2 regulate cilia length and canonical Hedgehog signaling via different mechanisms

(2018) Thomas Rosengren et al.   CELLULAR AND MOLECULAR LIFE SCIENCES

Proteomic analyses of signalling complexes associated with receptor tyrosine kinase identify novel members of fibroblast growth factor receptor 3 interactome

(2018) Lukas Balek et al.   CELLULAR SIGNALLING

Mechanistic insight into contextual TGF-β signaling

(2018) Ying E Zhang   CURRENT OPINION IN CELL BIOLOGY

Overcoming the resistance mechanisms of Smoothened inhibitors

(2018) Xiaochun Dong et al.   DRUG DISCOVERY TODAY

Export of membrane proteins from the Golgi complex to the primary cilium requires the kinesin motor, KIFC1

(2018) Si-Hyung Lee et al.   FASEB JOURNAL

40 YEARS of IGF1: IGF1: the Jekyll and Hyde of the aging brain

(2018) Sriram Gubbi et al.   JOURNAL OF MOLECULAR ENDOCRINOLOGY

Myosin-Va is required for preciliary vesicle transportation to the mother centriole during ciliogenesis

(2018) Chien-Ting Wu et al.   NATURE CELL BIOLOGY

Subcellular localization of MC4R with ADCY3 at neuronal primary cilia underlies a common pathway for genetic predisposition to obesity

(2018) Jacqueline E. Siljee et al.   NATURE GENETICS

Loss-of-function variants in ADCY3 increase risk of obesity and type 2 diabetes

(2018) Niels Grarup et al.   NATURE GENETICS

Loss-of-function mutations in ADCY3 cause monogenic severe obesity

(2018) Sadia Saeed et al.   NATURE GENETICS

EGF receptor kinase suppresses ciliogenesis through activation of USP8 deubiquitinase

(2018) Kousuke Kasahara et al.   Nature Communications

Primary Cilium-Mediated Retinal Pigment Epithelium Maturation Is Disrupted in Ciliopathy Patient Cells

(2018) Helen Louise May-Simera et al.   Cell Reports

CEP128 Localizes to the Subdistal Appendages of the Mother Centriole and Regulates TGF-β/BMP Signaling at the Primary Cilium

(2018) Maren Mönnich et al.   Cell Reports

Receptor Tyrosine Kinase Ubiquitination and De-Ubiquitination in Signal Transduction and Receptor Trafficking

(2018) William Critchley et al.   Cells

The daughter centriole controls ciliogenesis by regulating Neurl-4 localization at the centrosome

(2017) Abdelhalim Loukil et al.   JOURNAL OF CELL BIOLOGY

IFT20 modulates ciliary PDGFRα signaling by regulating the stability of Cbl E3 ubiquitin ligases

(2017) Fabian Marc Schmid et al.   JOURNAL OF CELL BIOLOGY

Microtubule stabilization drives 3D centrosome migration to initiate primary ciliogenesis

(2017) Amandine Pitaval et al.   JOURNAL OF CELL BIOLOGY

Genes and molecular pathways underpinning ciliopathies

(2017) Jeremy F. Reiter et al.   NATURE REVIEWS MOLECULAR CELL BIOLOGY

Toward personalized medicine in Bardet–Biedl syndrome

(2017) Joanna Kenny et al.   Personalized Medicine

Microtubule Motors Drive Hedgehog Signaling in Primary Cilia

(2017) Mu He et al.   TRENDS IN CELL BIOLOGY

The Cilium: Cellular Antenna and Central Processing Unit

(2017) Jarema J. Malicki et al.   TRENDS IN CELL BIOLOGY

Intraflagellar Transport and Ciliary Dynamics

(2017) Hiroaki Ishikawa et al.   Cold Spring Harbor Perspectives in Biology

Cilia and Obesity

(2017) Christian Vaisse et al.   Cold Spring Harbor Perspectives in Biology

Losers of Primary Cilia Gain the Benefit of Survival

(2017) Gladiola Goranci-Buzhala et al.   Cancer Discovery

A Transposon Screen Identifies Loss of Primary Cilia as a Mechanism of Resistance to SMO Inhibitors

(2017) Xuesong Zhao et al.   Cancer Discovery

The role of primary cilia in obesity and diabetes

(2016) Francesco Volta et al.   Annals of the New York Academy of Sciences

The Dark Side of Cell Signaling: Positive Roles for Negative Regulators

(2016) Mark A. Lemmon et al.   CELL

Mechanisms of ciliogenesis suppression in dividing cells

(2016) Hidemasa Goto et al.   CELLULAR AND MOLECULAR LIFE SCIENCES

Mechanism of ciliary disassembly

(2016) Yinwen Liang et al.   CELLULAR AND MOLECULAR LIFE SCIENCES

NDE1 and NDEL1 from genes to (mal)functions: parallel but distinct roles impacting on neurodevelopmental disorders and psychiatric illness

(2016) Nicholas J. Bradshaw et al.   CELLULAR AND MOLECULAR LIFE SCIENCES

Hedgehog Signal Transduction: Key Players, Oncogenic Drivers, and Cancer Therapy

(2016) Ekaterina Pak et al.   DEVELOPMENTAL CELL

CPAP promotes timely cilium disassembly to maintain neural progenitor pool

(2016) Elke Gabriel et al.   EMBO JOURNAL

Antennas of organ morphogenesis: the roles of cilia in vertebrate kidney development

(2016) Amanda N. Marra et al.   GENESIS

Ndel1 suppresses ciliogenesis in proliferating cells by regulating the trichoplein–Aurora A pathway

(2016) Hironori Inaba et al.   JOURNAL OF CELL BIOLOGY

Primary cilia are increased in number and demonstrate structural abnormalities in human cancer

(2016) Binnaz Yasar et al.   JOURNAL OF CLINICAL PATHOLOGY

Cilium assembly and disassembly

(2016) Irma Sánchez et al.   NATURE CELL BIOLOGY

Melanocortin-4 receptor–regulated energy homeostasis

(2016) Michael J Krashes et al.   NATURE NEUROSCIENCE

Endocytic Control of Cellular Signaling at the Primary Cilium

(2016) Lotte B. Pedersen et al.   TRENDS IN BIOCHEMICAL SCIENCES

Posttranslational Modifications of Tubulin and Cilia

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

Primary Cilia and Coordination of Receptor Tyrosine Kinase (RTK) and Transforming Growth Factor β (TGF-β) Signaling

(2016) Søren T. Christensen et al.   Cold Spring Harbor Perspectives in Biology

Phosphatidylinositol phosphate kinase PIPKIγ and phosphatase INPP5E coordinate initiation of ciliogenesis

(2016) Qingwen Xu et al.   Nature Communications

The deubiquitinase USP54 is overexpressed in colorectal cancer stem cells and promotes intestinal tumorigenesis

(2016) Julia M. Fraile et al.   Oncotarget

Smoothened Variants Explain the Majority of Drug Resistance in Basal Cell Carcinoma

(2015) Scott X. Atwood et al.   CANCER CELL

Genomic Analysis of Smoothened Inhibitor Resistance in Basal Cell Carcinoma

(2015) Hayley J. Sharpe et al.   CANCER CELL

Metabolic Regulation and Energy Homeostasis through the Primary Cilium

(2015) Edwin C. Oh et al.   Cell Metabolism

The tumor suppressor FBW7 controls ciliary length

(2015) A. S. Nikonova et al.   EMBO JOURNAL

Cell cycle-dependent ubiquitylation and destruction of NDE1 by CDK5-FBW7 regulates ciliary length

(2015) D. Maskey et al.   EMBO JOURNAL

Ins and outs of GPCR signaling in primary cilia

(2015) K. B. Schou et al.   EMBO REPORTS

Efficacy of Hedgehog Pathway Inhibitors in Basal Cell Carcinoma

(2015) N. Basset-Seguin et al.   MOLECULAR CANCER THERAPEUTICS

G-protein-independent coupling of MC4R to Kir7.1 in hypothalamic neurons

(2015) Masoud Ghamari-Langroudi et al.   NATURE

A neural basis for melanocortin-4 receptor–regulated appetite

(2015) Alastair S Garfield et al.   NATURE NEUROSCIENCE

Molecular genetics and clinical features of Birt–Hogg–Dubé syndrome

(2015) Laura S. Schmidt et al.   Nature Reviews Urology

Targeting GLI factors to inhibit the Hedgehog pathway

(2015) Paola Infante et al.   TRENDS IN PHARMACOLOGICAL SCIENCES

Role of ErbB Receptors in Cancer Cell Migration and Invasion

(2015) Aline Appert-Collin et al.   Frontiers in Pharmacology

Linking the Primary Cilium to Cell Migration in Tissue Repair and Brain Development

(2014) Iben Rønn Veland et al.   BIOSCIENCE

Sonic Hedgehog Signaling in Basal Cell Nevus Syndrome

(2014) M. Athar et al.   CANCER RESEARCH

The CP110-interacting proteins Talpid3 and Cep290 play overlapping and distinct roles in cilia assembly

(2014) Tetsuo Kobayashi et al.   JOURNAL OF CELL BIOLOGY

INPP5E interacts with AURKA, linking phosphoinositide signaling to primary cilium stability

(2014) O. V. Plotnikova et al.   JOURNAL OF CELL SCIENCE

Leptin-promoted cilia assembly is critical for normal energy balance

(2014) Yu Mi Han et al.   JOURNAL OF CLINICAL INVESTIGATION

 -Catenin Links von Hippel-Lindau to Aurora Kinase A and Loss of Primary Cilia in Renal Cell Carcinoma

(2014) R. Dere et al.   JOURNAL OF THE AMERICAN SOCIETY OF NEPHROLOGY

Genome-wide association study of height-adjusted BMI in childhood identifies functional variant inADCY3

(2014) Evangelia Stergiakouli et al.   Obesity

How do cilia organize signalling cascades?

(2014) M. V. Nachury   PHILOSOPHICAL TRANSACTIONS OF THE ROYAL SOCIETY B-BIOLOGICAL SCIENCES

OFD1 and Flotillins Are Integral Components of a Ciliary Signaling Protein Complex Organized by Polycystins in Renal Epithelia and Odontoblasts

(2014) Stephanie Jerman et al.   PLoS One

Context-dependent signal integration by the GLI code: The oncogenic load, pathways, modifiers and implications for cancer therapy

(2014) Fritz Aberger et al.   SEMINARS IN CELL & DEVELOPMENTAL BIOLOGY

TGFβ signaling in establishing left–right asymmetry

(2014) Hidetaka Shiratori et al.   SEMINARS IN CELL & DEVELOPMENTAL BIOLOGY

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

(2014) Miju Kim et al.   Nature Communications

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

(2014) Kousuke Kasahara et al.   Nature Communications

Dido3-dependent HDAC6 targeting controls cilium size

(2014) Ainhoa Sánchez de Diego et al.   Nature Communications

Nephronophthisis-Associated CEP164 Regulates Cell Cycle Progression, Apoptosis and Epithelial-to-Mesenchymal Transition

(2014) Gisela G. Slaats et al.   PLoS Genetics

HDAC6 Inhibition Restores Ciliary Expression and Decreases Tumor Growth

(2013) S. A. Gradilone et al.   CANCER RESEARCH

Cell cycle progression by the repression of primary cilia formation in proliferating cells

(2013) Hidemasa Goto et al.   CELLULAR AND MOLECULAR LIFE SCIENCES

Cluap1 localizes preferentially to the base and tip of cilia and is required for ciliogenesis in the mouse embryo

(2013) Yanick Botilde et al.   DEVELOPMENTAL BIOLOGY

IGF-1 Activates a Cilium-Localized Noncanonical Gβγ Signaling Pathway that Regulates Cell-Cycle Progression

(2013) Celine Yeh et al.   DEVELOPMENTAL CELL

Centriole distal appendages promote membrane docking, leading to cilia initiation

(2013) B. E. Tanos et al.   GENES & DEVELOPMENT

Presence of primary cilia in cancer cells correlates with prognosis of pancreatic ductal adenocarcinoma

(2013) Katsura Emoto et al.   HUMAN PATHOLOGY

Arl13b-regulated cilia activities are essential for polarized radial glial scaffold formation

(2013) Holden Higginbotham et al.   NATURE NEUROSCIENCE

Primary cilia in neurodevelopmental disorders

(2013) Enza Maria Valente et al.   Nature Reviews Neurology

Shaping Our Minds: Stem and Progenitor Cell Diversity in the Mammalian Neocortex

(2013) Santos J. Franco et al.   NEURON

Primary Cilia Are Lost in Preinvasive and Invasive Prostate Cancer

(2013) Nadia B. Hassounah et al.   PLoS One

CCDC41 is required for ciliary vesicle docking to the mother centriole

(2013) K. Joo et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

TGF-β Signaling Is Associated with Endocytosis at the Pocket Region of the Primary Cilium

(2013) Christian Alexandro Clement et al.   Cell Reports

Protein tyrosine kinase regulation by ubiquitination: Critical roles of Cbl-family ubiquitin ligases

(2012) Bhopal Mohapatra et al.   BIOCHIMICA ET BIOPHYSICA ACTA-MOLECULAR CELL RESEARCH

TTBK2 Kinase: Linking Primary Cilia and Cerebellar Ataxias

(2012) Peter K. Jackson   CELL

The Spinocerebellar Ataxia-Associated Gene Tau Tubulin Kinase 2 Controls the Initiation of Ciliogenesis

(2012) Sarah C. Goetz et al.   CELL

Polycystins and cellular Ca2+ signaling

(2012) D. Mekahli et al.   CELLULAR AND MOLECULAR LIFE SCIENCES

Molecular Pathways: The Role of Primary Cilia in Cancer Progression and Therapeutics with a Focus on Hedgehog Signaling

(2012) N. B. Hassounah et al.   CLINICAL CANCER RESEARCH

Arl13b in Primary Cilia Regulates the Migration and Placement of Interneurons in the Developing Cerebral Cortex

(2012) Holden Higginbotham et al.   DEVELOPMENTAL CELL

Identification of a novel Wnt5a-CK1ε-Dvl2-Plk1-mediated primary cilia disassembly pathway

(2012) Kyung Ho Lee et al.   EMBO JOURNAL

Neurl4, a novel daughter centriole protein, prevents formation of ectopic microtubule organizing centres

(2012) Ji Li et al.   EMBO REPORTS

The Fbw7 and BetaTRCP E3 ubiquitin ligases and their roles in tumorigenesis

(2012) Alan et al.   Frontiers in Bioscience-Landmark

Trichoplein and Aurora A block aberrant primary cilia assembly in proliferating cells

(2012) Akihito Inoko et al.   JOURNAL OF CELL BIOLOGY

Cep164 mediates vesicular docking to the mother centriole during early steps of ciliogenesis

(2012) Kerstin N. Schmidt et al.   JOURNAL OF CELL BIOLOGY

PDGFR  signaling in the primary cilium regulates NHE1-dependent fibroblast migration via coordinated differential activity of MEK1/2-ERK1/2-p90RSK and AKT signaling pathways

(2012) D. L. Clement et al.   JOURNAL OF CELL SCIENCE

Hedgehog-GLI Pathway in Medulloblastoma

(2012) Tenley C. Archer et al.   JOURNAL OF CLINICAL ONCOLOGY

Interaction Proteomics Identify NEURL4 and the HECT E3 Ligase HERC2 as Novel Modulators of Centrosome Architecture

(2012) Abdallah K. Al-Hakim et al.   MOLECULAR & CELLULAR PROTEOMICS

Calmodulin activation of Aurora-A kinase (AURKA) is required during ciliary disassembly and in mitosis

(2012) Olga V. Plotnikova et al.   MOLECULAR BIOLOGY OF THE CELL

Medulloblastoma exome sequencing uncovers subtype-specific somatic mutations

(2012) Trevor J. Pugh et al.   NATURE

Primary cilia attenuate hedgehog signalling in neoplastic chondrocytes

(2012) L Ho et al.   ONCOGENE

Fluid flow and interlinked feedback loops establish left–right asymmetric decay of Cerl2 mRNA

(2012) Tetsuya Nakamura et al.   Nature Communications

The ciliary pocket: a once-forgotten membrane domain at the base of cilia

(2011) Rania Ghossoub et al.   BIOLOGY OF THE CELL

Left–right asymmetry in the level of active Nodal protein produced in the node is translated into left–right asymmetry in the lateral plate of mouse embryos

(2011) Aiko Kawasumi et al.   DEVELOPMENTAL BIOLOGY

Trichoplein controls microtubule anchoring at the centrosome by binding to Odf2 and ninein

(2011) M. Ibi et al.   JOURNAL OF CELL SCIENCE

Primary cilia and coordination of receptor tyrosine kinase (RTK) signalling

(2011) Søren T Christensen et al.   JOURNAL OF PATHOLOGY

Ciliary transition zone activation of phosphorylated Tctex-1 controls ciliary resorption, S-phase entry and fate of neural progenitors

(2011) Aiqun Li et al.   NATURE CELL BIOLOGY

Nde1-mediated inhibition of ciliogenesis affects cell cycle re-entry

(2011) Sehyun Kim et al.   NATURE CELL BIOLOGY

The Cerebrospinal Fluid Provides a Proliferative Niche for Neural Progenitor Cells

(2011) Maria K. Lehtinen et al.   NEURON

Primary Cilium Depletion Typifies Cutaneous Melanoma In Situ and Malignant Melanoma

(2011) Jinah Kim et al.   PLoS One

Cell Signaling by Receptor Tyrosine Kinases

(2010) Mark A. Lemmon et al.   CELL

Role of primary cilia in brain development and cancer

(2010) Young-Goo Han et al.   CURRENT OPINION IN NEUROBIOLOGY

The Hippo Tumor Pathway Promotes TAZ Degradation by Phosphorylating a Phosphodegron and Recruiting the SCFβ-TrCPE3 Ligase

(2010) Chen-Ying Liu et al.   JOURNAL OF BIOLOGICAL CHEMISTRY

The ciliary pocket: an endocytic membrane domain at the base of primary and motile cilia

(2010) A. Molla-Herman et al.   JOURNAL OF CELL SCIENCE

Medulloblastoma Comprises Four Distinct Molecular Variants

(2010) Paul A. Northcott et al.   JOURNAL OF CLINICAL ONCOLOGY

Primary Cilia Are Decreased in Breast Cancer: Analysis of a Collection of Human Breast Cancer Cell Lines and Tissues

(2010) Kun Yuan et al.   JOURNAL OF HISTOCHEMISTRY & CYTOCHEMISTRY

Roles for Growth Factors in Cancer Progression

(2010) Esther Witsch et al.   PHYSIOLOGY

Arsenic antagonizes the Hedgehog pathway by preventing ciliary accumulation and reducing stability of the Gli2 transcriptional effector

(2010) J. Kim et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

Rapid calcium-dependent activation of Aurora-A kinase

(2010) Olga V. Plotnikova et al.   Nature Communications

Novel epigenetic regulation of skeletal muscle myosin heavy chain genes. Focus on “Differential epigenetic modifications of histones at the myosin heavy chain genes in fast and slow skeletal muscle fibers and in response to muscle unloading”

(2009) Kevin A. Zwetsloot et al.   AMERICAN JOURNAL OF PHYSIOLOGY-CELL PHYSIOLOGY

Primary ciliogenesis defects are associated with human astrocytoma/glioblastoma cells

(2009) Joanna J Moser et al.   BMC CANCER

Pancreatic Cancer and Precursor Pancreatic Intraepithelial Neoplasia Lesions Are Devoid of Primary Cilia

(2009) E. S. Seeley et al.   CANCER RESEARCH

Cdk4/CyclinD1 Overexpression in Neural Stem Cells Shortens G1, Delays Neurogenesis, and Promotes the Generation and Expansion of Basal Progenitors

(2009) Christian Lange et al.   Cell Stem Cell

Characterization of Primary Cilia in Human Airway Smooth Muscle Cells

(2009) Jun Wu et al.   CHEST

The Primary Cilium as a Complex Signaling Center

(2009) Nicolas F. Berbari et al.   CURRENT BIOLOGY

Adenylate cyclase regulates elongation of mammalian primary cilia

(2009) Young Ou et al.   EXPERIMENTAL CELL RESEARCH

Enhanced Stat3 Activation in POMC Neurons Provokes Negative Feedback Inhibition of Leptin and InsulinSignaling in Obesity

(2009) M. B. Ernst et al.   JOURNAL OF NEUROSCIENCE

Dual and opposing roles of primary cilia in medulloblastoma development

(2009) Young-Goo Han et al.   NATURE MEDICINE

Primary cilia can both mediate and suppress Hedgehog pathway–dependent tumorigenesis

(2009) Sunny Y Wong et al.   NATURE MEDICINE

Small-molecule inhibitors reveal multiple strategies for Hedgehog pathway blockade

(2009) J. M. Hyman et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

Left-Right Determination: Involvement of Molecular Motor KIF3, Cilia, and Nodal Flow

(2009) N. Hirokawa et al.   Cold Spring Harbor Perspectives in Biology

Mutations in the Cilia Gene ARL13B Lead to the Classical Form of Joubert Syndrome

(2008) Vincent Cantagrel et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Activation of Stat3 Signaling in AgRP Neurons Promotes Locomotor Activity

(2008) Andrea Mesaros et al.   Cell Metabolism

CP110 Suppresses Primary Cilia Formation through Its Interaction with CEP290, a Protein Deficient in Human Ciliary Disease

(2008) William Y. Tsang et al.   DEVELOPMENTAL CELL

The role of the EGF family of ligands and receptors in renal development, physiology and pathophysiology

(2008) Fenghua Zeng et al.   EXPERIMENTAL CELL RESEARCH

Association of common variants in the Joubert syndrome gene (AHI1) with autism

(2008) Ana I. Alvarez Retuerto et al.   HUMAN MOLECULAR GENETICS

Sporadic clear cell renal cell carcinoma but not the papillary type is characterized by severely reduced frequency of primary cilia

(2008) Peter Schraml et al.   MODERN PATHOLOGY

Mice Lacking Histone Deacetylase 6 Have Hyperacetylated Tubulin but Are Viable and Develop Normally

(2008) Y. Zhang et al.   MOLECULAR AND CELLULAR BIOLOGY

Integrated Genomics Identifies Five Medulloblastoma Subtypes with Distinct Genetic Profiles, Pathway Signatures and Clinicopathological Features

(2008) Marcel Kool et al.   PLoS One

Bardet-Biedl syndrome proteins are required for the localization of G protein-coupled receptors to primary cilia

(2008) N. F. Berbari et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA