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- References [743]
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- Product number
- 17711-1-AP - Provider product page
- Provider
- Proteintech Group
- Proper citation
- Proteintech Cat#17711-1-AP, RRID:AB_2060867
- Product name
- ARL13B antibody
- Antibody type
- Polyclonal
- Description
- KD/KO validated ARL13B antibody (Cat. #17711-1-AP) is a rabbit polyclonal antibody that shows reactivity with human, mouse, rat, Canine and has been validated for the following applications: IF, IHC, IP, WB, ELISA.
- Reactivity
- Human, Mouse, Rat, Canine
- Host
- Rabbit
- Conjugate
- Unconjugated
- Isotype
- IgG
- Vial size
- 20ul, 150ul
Submitted references POMC Neuron BBSome Regulation of Body Weight is Independent of its Ciliary Function.
Immunolabel-First-Expand-Later Expansion Microscopy Approach Using Stable STED Dyes.
Expansion Microscopy of Ciliary Proteins.
A Chemically Inducible Organelle Rerouting Assay to Probe Primary Cilium Assembly, Maintenance, and Disassembly in Cultured Cells.
Proximity Mapping of Ciliary Proteins by BioID.
Ultrastructure expansion microscopy (U-ExM) of mouse and human kidneys for analysis of subcellular structures.
Cytoskeletal β-tubulin and cysteine cathepsin L deregulation by SARS-CoV-2 spike protein interaction with the neuronal model cell line SH-SY5Y.
Rediscovering the Rete Ovarii: a secreting auxiliary structure to the ovary.
Primary Cilia as a Tumor Marker in Pituitary Neuroendocrine Tumors.
Targeting Rab-RILPL interactions as a strategy to downregulate pathogenic LRRK2 in Parkinson's disease.
MicroRNA‑155‑5p inhibits trophoblast cell proliferation and invasion by disrupting centrosomal function.
Engrailed 1 deficiency induces changes in ciliogenesis during human neuronal differentiation.
Extracellular matrix hyaluronan modulates fat cell differentiation and primary cilia dynamics.
Selective targeting of mu opioid receptors to primary cilia.
Assessment and Comparison of Early Developmental Toxicity of Six Per- and Polyfluoroalkyl Substances with Human Embryonic Stem Cell Models.
Bridge-like lipid transfer protein family member 2 suppresses ciliogenesis.
PI3KCIIα-Dependent Autophagy Program Protects From Endothelial Dysfunction and Atherosclerosis in Response to Low Shear Stress in Mice.
Store-operated calcium entry inhibits primary ciliogenesis via the activation of Aurora A.
XIAP-mediated degradation of IFT88 disrupts HSC cilia to stimulate HSC activation and liver fibrosis.
Bioengineered Tubular Biliary Organoids.
Primary Cilium in Neural Crest Cells Crucial for Anterior Segment Development and Corneal Avascularity.
Unraveling cellular complexity with transient adapters in highly multiplexed super-resolution imaging.
ESCRT-I protein UBAP1 controls ventricular expansion and cortical neurogenesis via modulating adherens junctions of radial glial cells.
Molecular basis promoting centriole triplet microtubule assembly.
Mechanisms underlying morphological and functional changes of cilia in fibroblasts derived from patients bearing ARL3(T31A) and ARL3(T31A/C118F) mutations.
Diesel exhaust particle exposure exacerbates ciliary and epithelial barrier dysfunction in the multiciliated bronchial epithelium models.
KIF3A tail domain phosphorylation is not required for ciliogenesis in mouse embryonic fibroblasts.
The CPLANE protein Fuzzy regulates ciliogenesis by suppressing actin polymerization at the base of the primary cilium via p190A RhoGAP.
DLG1 functions upstream of SDCCAG3 and IFT20 to control ciliary targeting of polycystin-2.
In vivo CRISPR screening directly targeting testicular cells.
The ARPKD Protein DZIP1L Regulates Ciliary Protein Entry by Modulating the Architecture and Function of Ciliary Transition Fibers.
The Immune Checkpoint Protein PD-L1 Regulates Ciliogenesis and Hedgehog Signaling.
Aurora kinase A inhibition plus Tumor Treating Fields suppress glioma cell proliferation in a cilium-independent manner.
A sterol analog inhibits hedgehog pathway by blocking cholesterylation of smoothened.
Bi-allelic variants in CEP295 cause Seckel-like syndrome presenting with primary microcephaly, developmental delay, intellectual disability, short stature, craniofacial and digital abnormalities.
Racgap1 knockdown results in cells with multiple cilia due to cytokinesis failure.
Human Kidney Proximal Tubule-Microvascular Model Facilitates High-Throughput Analyses of Structural and Functional Effects of Ischemia-Reperfusion Injury.
Interactome Analysis Reveals a Link of the Novel ALMS1-CEP70 Complex to Centrosomal Clusters.
Phosphodiesterase 4 activity uniquely regulates ciliary cAMP-dependent 3T3-L1 adipogenesis.
Upregulation of miR-20b-5p inhibits trophoblast invasion by blocking autophagy in recurrent miscarriage.
Variants in the WDR44 WD40-repeat domain cause a spectrum of ciliopathy by impairing ciliogenesis initiation.
TMEM216 promotes primary ciliogenesis and Hedgehog signaling through the SUFU-GLI2/GLI3 axis.
Cytoneme signaling provides essential contributions to mammalian tissue patterning.
Hedgehog signalling is involved in acquired resistance to KRAS(G12C) inhibitors in lung cancer cells.
The novel linkage between Fuz and Gpr161 genes regulates sonic hedgehog signaling during mouse embryonic development.
The Fuzzy planar cell polarity protein (FUZ), necessary for primary cilium formation, is essential for pituitary development.
Primary cilium participates in radiation-induced bystander effects through TGF-β1 signaling.
The miR-669a-5p/G3BP/HDAC6/AKAP12 Axis Regulates Primary Cilia Length.
DPCD is a regulator of R2TP in ciliogenesis initiation through Akt signaling.
Primary cilia promote the differentiation of human neurons through the WNT signaling pathway.
Mouse SAS-6 is required for centriole formation in embryos and integrity in embryonic stem cells.
The Primary Cilium and its Hedgehog Signaling in Nociceptors Contribute to Inflammatory and Neuropathic Pain.
Understanding the effects of per- and polyfluoroalkyl substances on early skin development: Role of ciliogenesis inhibition and altered microtubule dynamics.
Collagen VI Deficiency Impairs Tendon Fibroblasts Mechanoresponse in Ullrich Congenital Muscular Dystrophy.
The exocyst complex and intracellular vesicles mediate soluble protein trafficking to the primary cilium.
Early insights into the role of Exoc6B associated with spondyloepimetaphyseal dysplasia with joint laxity type 3 in primary ciliogenesis and chondrogenic differentiation in vitro.
Ultrastructure expansion microscopy (U-ExM) of mouse and human kidneys for analysis of subcellular structures.
Immuno-Scanning Electron Microscopy of Islet Primary Cilia.
Motor protein Kif6 regulates cilia motility and polarity in brain ependymal cells.
Kidney ischemia/reperfusion injury causes cholangiocytes primary cilia disruption and abnormal bile secretion.
Absence of the RING domain in MID1 results in patterning defects in the developing human brain.
INPP5E Regulates the Distribution of Phospholipids on Cilia in RPE1 Cells.
Differential alternative splicing analysis links variation in ZRSR2 to a novel type of oral-facial-digital syndrome.
A disease-associated PPP2R3C-MAP3K1 phospho-regulatory module controls centrosome function.
Roles for CEP170 in cilia function and dynein-2 assembly.
The Scaffold Protein KATNIP Enhances CILK1 Control of Primary Cilia.
Characterization of Primary Cilia Formation in Human ESC-Derived Retinal Organoids.
In vivo Polycystin-1 interactome using a novel Pkd1 knock-in mouse model.
Human LUHMES and NES cells as models for studying primary cilia in neurons.
Identification of novel TMEM231 gene splice variants and pathological findings in a fetus with Meckel Syndrome.
Morphological changes and two Nodal paralogs drive left-right asymmetry in the squamate veiled chameleon (C. calyptratus).
A targeted multi-proteomics approach generates a blueprint of the ciliary ubiquitinome.
Ciliary phenotyping in renal epithelial cells in a cranioectodermal dysplasia patient with WDR35 variants.
Brain microvascular endothelial cells possess a second cilium that arises from the daughter centriole.
INPP5E regulates CD3ζ enrichment at the immune synapse by phosphoinositide distribution control.
Pathogenic RAB34 variants impair primary cilium assembly and cause a novel oral-facial-digital syndrome.
Increasing Ciliary ARL13B Expression Drives Active and Inhibitor-Resistant Smoothened and GLI into Glioma Primary Cilia.
The IFT81-IFT74 complex acts as an unconventional RabL2 GTPase-activating protein during intraflagellar transport.
Dnajb11-Kidney Disease Develops from Reduced Polycystin-1 Dosage but not Unfolded Protein Response in Mice.
ODF2 Negatively Regulates CP110 Levels at the Centrioles/Basal Bodies to Control the Biogenesis of Primary Cilia.
Primary cilia control oligodendrocyte precursor cell proliferation in white matter injury via Hedgehog-independent CREB signaling.
PATJ inhibits histone deacetylase 7 to control tight junction formation and cell polarity.
Parkinsonism Sac domain mutation in Synaptojanin-1 affects ciliary properties in iPSC-derived dopaminergic neurons.
Primary cilium-mediated signaling cascade suppresses age-related biliary fibrosis.
Certain heterozygous variants in the kinase domain of the serine/threonine kinase NEK8 can cause an autosomal dominant form of polycystic kidney disease.
Proteomic profiling of centrosomes across multiple mammalian cell and tissue types by an affinity capture method.
Oxidative Stress Induces Skin Pigmentation in Melasma by Inhibiting Hedgehog Signaling.
Absence of the primary cilia formation gene Talpid3 impairs muscle stem cell function.
MiniBAR/GARRE1 is a dual Rac and Rab effector required for ciliogenesis.
Thalamic Neuron Resilience during Osmotic Demyelination Syndrome (ODS) Is Revealed by Primary Cilium Outgrowth and ADP-ribosylation factor-like protein 13B Labeling in Axon Initial Segment.
Proximity Labeling Expansion Microscopy (PL-ExM) resolves structure of the interactome.
Polycystin-2-dependent transcriptome reveals early response of autosomal dominant polycystic kidney disease.
LRP2 contributes to planar cell polarity-dependent coordination of motile cilia function.
PDLIM3 supports hedgehog signaling in medulloblastoma by facilitating cilia formation.
Rab8 and TNPO1 are ciliary transport adaptors for GTPase Arl13b by interacting with its RVEP motif containing ciliary targeting sequence.
Scanning electron microscopy of human islet cilia.
Extracellular vesicle-mediated delivery of anti-miR-106b inhibits morphine-induced primary ciliogenesis in the brain.
Investigations on Primary Cilia of Nthy-ori 3-1 Cells upon Cysteine Cathepsin Inhibition or Thyrotropin Stimulation.
CCP5 and CCP6 retain CP110 and negatively regulate ciliogenesis.
Ciliary ARL13B inhibits developmental kidney cystogenesis in mouse.
Enhanced primary ciliogenesis via mitochondrial oxidative stress activates AKT to prevent neurotoxicity in HSPA9/mortalin-depleted SH-SY5Y cells.
Primary cilia sense glutamine availability and respond via asparagine synthetase.
The Rab GTPase-binding protein EHBP1L1 and its interactors CD2AP/CIN85 negatively regulate the length of primary cilia via actin remodeling.
Primary Cilium Identifies a Quiescent Cell Population in the Human Intestinal Crypt.
Reserpine maintains photoreceptor survival in retinal ciliopathy by resolving proteostasis imbalance and ciliogenesis defects.
An actin filament branching surveillance system regulates cell cycle progression, cytokinesis and primary ciliogenesis.
The developmental basis of fingerprint pattern formation and variation.
Primary cilia control cellular patterning of Meibomian glands during morphogenesis but not lipid composition.
Inactivation of Invs/Nphp2 in renal epithelial cells drives infantile nephronophthisis like phenotypes in mouse.
Interactions between TULP3 tubby domain and ARL13B amphipathic helix promote lipidated protein transport to cilia.
Canonical Wnt signaling regulates soft palate development by mediating ciliary homeostasis.
Disease-associated mutations in WDR34 lead to diverse impacts on the assembly and function of dynein-2.
Multiple interactions of the dynein-2 complex with the IFT-B complex are required for effective intraflagellar transport.
Arl13b promotes the proliferation, migration, osteogenesis, and mechanosensation of osteoblasts.
Generation of iPSC-derived human forebrain organoids assembling bilateral eye primordia.
Eupatilin Improves Cilia Defects in Human CEP290 Ciliopathy Models.
Polycomb Ezh1 maintains murine muscle stem cell quiescence through non-canonical regulation of Notch signaling.
Single-cell transcriptomics reveals ependymal subtypes related to cytoskeleton dynamics as the core driver of syringomyelia pathological development.
ULK4 and Fused/STK36 interact to mediate assembly of a motile flagellum.
Functional characterization of C21ORF2 association with the NEK1 kinase mutated in human in diseases.
Impairment of Connexin 43 may initiate cilia decline in syringomyelia.
MRCKα/β positively regulates Gli protein activity.
Coordination of canonical and noncanonical Hedgehog signalling pathways mediated by WDR11 during primordial germ cell development.
CEP164-GLI2 association ensures the hedgehog signaling in pancreatic cancer cells.
Dynein-2-driven intraciliary retrograde trafficking indirectly requires multiple interactions of IFT54 in the IFT-B complex with the dynein-2 complex.
Atp7b-dependent choroid plexus dysfunction causes transient copper deficit and metabolic changes in the developing mouse brain.
PDGF-AA activates AKT and ERK signaling for testicular interstitial Leydig cell growth via primary cilia.
CROCCP2 acts as a human-specific modifier of cilia dynamics and mTOR signaling to promote expansion of cortical progenitors.
Primary cilia are WNT-transducing organelles whose biogenesis is controlled by a WNT-PP1 axis.
Superenhancer activation of KLHDC8A drives glioma ciliation and hedgehog signaling.
Diverse but unique astrocytic phenotypes during embryonic stem cell differentiation, culturing and development.
A hierarchical pathway for assembly of the distal appendages that organize primary cilia.
Myristoylated Neuronal Calcium Sensor-1 captures the ciliary vesicle at distal appendages.
TTBK2 controls cilium stability by regulating distinct modules of centrosomal proteins.
XBP1 Activation Reduces Severity of Polycystic Kidney Disease due to a Nontruncating Polycystin-1 Mutation in Mice.
A SEC61A1 variant is associated with autosomal dominant polycystic liver disease.
Reduced SPAG17 Expression in Systemic Sclerosis Triggers Myofibroblast Transition and Drives Fibrosis.
Evaluation of Epithelial-Mesenchymal Transition Markers in Autoimmune Thyroid Diseases.
IHH, SHH, and primary cilia mediate epithelial-stromal cross-talk during decidualization in mice.
Deficiency of the minor spliceosome component U4atac snRNA secondarily results in ciliary defects in human and zebrafish.
Kif6 regulates cilia motility and polarity in brain ependymal cells.
Centriolar satellites expedite mother centriole remodeling to promote ciliogenesis.
Scanning electron microscopy of human islet cilia.
GRAMD1/ASTER-mediated cholesterol transport promotes Smoothened cholesterylation at the endoplasmic reticulum.
TTBK2 mutations associated with spinocerebellar ataxia type 11 disrupt peroxisome dynamics and ciliary localization of SHH signaling proteins.
Deletion of IFT20 exclusively in the RPE ablates primary cilia and leads to retinal degeneration.
CXCL12 targets the primary cilium cAMP/cGMP ratio to regulate cell polarity during migration.
Beta cell primary cilia mediate somatostatin responsiveness via SSTR3.
The Primary Cilium and its Hedgehog Signaling in Nociceptors Contribute to Inflammatory and Neuropathic Pain.
Human iPSC-derived renal collecting duct organoid model cystogenesis in ADPKD.
HDAC6 inhibition regulates substrate stiffness-mediated inflammation signaling in chondrocytes.
Identification of new ciliary signaling pathways in the brain and insights into neurological disorders.
WDR31 displays functional redundancy with GTPase-activating proteins (GAPs) ELMOD and RP2 in regulating IFT complex and recruiting the BBSome to cilium.
Ciliary ARL13B inhibits developmental kidney cystogenesis in mouse.
A Novel Genetic Variant in MBD5 Associated with Severe Epilepsy and Intellectual Disability: Potential Implications on Neural Primary Cilia.
Tau tubulin kinase 1 and 2 regulate ciliogenesis and human pluripotent stem cells-derived neural rosettes.
DOT1L activity affects neural stem cell division mode and reduces differentiation and ASNS expression.
Hedgehog target genes regulate lipid metabolism to drive basal cell carcinoma and medulloblastoma.
PCDH12 loss results in premature neuronal differentiation and impeded migration in a cortical organoid model.
Mutational burden of XPNPEP3 leads to defects in mitochondrial complex I and cilia in NPHPL1.
Increase in primary cilia number and length upon VDAC1 depletion contributes to attenuated proliferation of cancer cells.
Ubiquitylation of BBSome is required for ciliary assembly and signaling.
Septin-mediated RhoA activation engages the exocyst complex to recruit the cilium transition zone.
A stress-induced cilium-to-PML-NB route drives senescence initiation.
Identification of a heterogeneous and dynamic ciliome during embryonic development and cell differentiation.
Eupatilin improves cilia defects in human CEP290 ciliopathy models.
Pyrogallol-Phloroglucinol-6, 6-Bieckol Restored Primary Cilia Length, Which Was Decreased by High-Fat Diet in Visceral Adipose Tissue, and Decreased Adipogenesis.
Systematic analysis of cilia characteristics and Hedgehog signaling in five immortal cell lines.
Physical Exercise-Induced Astrocytic Neuroprotection and Cognitive Improvement Through Primary Cilia and Mitogen-Activated Protein Kinases Pathway in Rats With Chronic Cerebral Hypoperfusion.
Renal primary cilia lengthen in the progression of diabetic kidney disease.
Microtubule modification defects underlie cilium degeneration in cell models of retinitis pigmentosa associated with pre-mRNA splicing factor mutations.
Fluorescence imaging of beta cell primary cilia.
Hedgehog signaling is controlled by Rac1 activity.
Concurrent Germline and Somatic Mutations in FLCN and Preliminary Exploration of Its Function: A Case Report.
Human Organotypic Airway and Lung Organoid Cells of Bronchiolar and Alveolar Differentiation Are Permissive to Infection by Influenza and SARS-CoV-2 Respiratory Virus.
Modeling Human Primary Microcephaly With hiPSC-Derived Brain Organoids Carrying CPAP-E1235V Disease-Associated Mutant Protein.
Biallelic KIF24 Variants Are Responsible for a Spectrum of Skeletal Disorders Ranging From Lethal Skeletal Ciliopathy to Severe Acromesomelic Dysplasia.
KLC3 Regulates Ciliary Trafficking and Cyst Progression in CILK1 Deficiency-Related Polycystic Kidney Disease.
Deletion of CEP164 in mouse photoreceptors post-ciliogenesis interrupts ciliary intraflagellar transport (IFT).
Ttc21b deficiency attenuates autosomal dominant polycystic kidney disease in a kidney tubular- and maturation-dependent manner.
Plexins promote Hedgehog signaling through their cytoplasmic GAP activity.
Islet primary cilia motility controls insulin secretion.
AMBRA1 p.Gln30Arg Mutation, Identified in a Cowden Syndrome Family, Exhibits Hyperproliferative Potential in hTERT-RPE1 Cells.
The Nicotinic Receptor Polymorphism rs16969968 Is Associated with Airway Remodeling and Inflammatory Dysregulation in COPD Patients.
The intraflagellar transport protein IFT52 associated with short-rib thoracic dysplasia is essential for ciliary function in osteogenic differentiation in vitro and for sensory perception in Drosophila.
Carnitine Protects against MPP(+)-Induced Neurotoxicity and Inflammation by Promoting Primary Ciliogenesis in SH-SY5Y Cells.
A PKA inhibitor motif within SMOOTHENED controls Hedgehog signal transduction.
Heterozygous variants in MYH10 associated with neurodevelopmental disorders and congenital anomalies with evidence for primary cilia-dependent defects in Hedgehog signaling.
The Primary Ciliary Deficits in Cerebellar Bergmann Glia of the Mouse Model of Fragile X Syndrome.
Endothelial β-arrestins regulate mechanotransduction by the type II bone morphogenetic protein receptor in primary cilia.
Mutations in SCNM1 cause orofaciodigital syndrome due to minor intron splicing defects affecting primary cilia.
Analysis of Primary Cilium Expression and Hedgehog Pathway Activation in Mesothelioma Throws Back Its Complex Biology.
KIF24 depletion induces clustering of supernumerary centrosomes in PDAC cells.
Nuclear factor (erythroid-derived 2)-like 2 counter-regulates thymosin beta-4 expression and primary cilium formation for HeLa cervical cancer cell survival.
HeLa Cervical Cancer Cells Are Maintained by Nephronophthisis 3-Associated Primary Cilium Formation via ROS-Induced ERK and HIF-1α Activation under Serum-Deprived Normoxic Condition.
A Potent Antagonist of Smoothened in Hedgehog Signaling for Epilepsy.
A high-throughput electron tomography workflow reveals over-elongated centrioles in relapsed/refractory multiple myeloma.
Metabolomic fingerprinting of renal disease progression in Bardet-Biedl syndrome reveals mitochondrial dysfunction in kidney tubular cells.
Cilia-Mediated Insulin/Akt and ST2/JNK Signaling Pathways Regulate the Recovery of Muscle Injury.
LRRK1-mediated NDEL1 phosphorylation promotes cilia disassembly via dynein-2-driven retrograde intraflagellar transport.
Midbody plays an active role in fibroblast-myofibroblast transition by mediating TGF-β signaling.
Agonists of prostaglandin E(2) receptors as potential first in class treatment for nephronophthisis and related ciliopathies.
Inhibition of Ciliogenesis Enhances the Cellular Sensitivity to Temozolomide and Ionizing Radiation in Human Glioblastoma Cells.
The ciliary gene INPP5E confers dorsal telencephalic identity to human cortical organoids by negatively regulating Sonic hedgehog signaling.
Cholesterylation of Smoothened is a calcium-accelerated autoreaction involving an intramolecular ester intermediate.
Identification of a novel variant of the ciliopathic gene FUZZY associated with craniosynostosis.
The carboxyl-terminal region of SDCCAG8 comprises a functional module essential for cilia formation as well as organ development and homeostasis.
TMED2 binding restricts SMO to the ER and Golgi compartments.
Biallelic variants in CENPF causing a phenotype distinct from Strømme syndrome.
Evolutionary conservation of centriole rotational asymmetry in the human centrosome.
Cilia proteins are biomarkers of altered flow in the vasculature.
3D-Structured Illumination Microscopy of Centrosomes in Human Cell Lines.
A protocol for generation and live-cell imaging analysis of primary cilia reporter cell lines.
ALKBH3-dependent m(1)A demethylation of Aurora A mRNA inhibits ciliogenesis.
Primary Cilium by Polyinosinic:Polycytidylic Acid Regulates the Regenerative Migration of Beas-2B Bronchial Epithelial Cells.
Ciliary IFT88 Protects Coordinated Adolescent Growth Plate Ossification From Disruptive Physiological Mechanical Forces.
Preliminary results on a proposed histopathological assessment of predictive factors for basal cell carcinoma recurrence after primary free margin excision.
Ciliary Proteins Repurposed by the Synaptic Ribbon: Trafficking Myristoylated Proteins at Rod Photoreceptor Synapses.
Control of meiotic chromosomal bouquet and germ cell morphogenesis by the zygotene cilium.
Pathogenic LRRK2 regulates centrosome cohesion via Rab10/RILPL1-mediated CDK5RAP2 displacement.
Angiomotin isoform 2 promotes binding of PALS1 to KIF13B at primary cilia and regulates ciliary length and signaling.
The developmental stage of the medulloblastoma cell-of-origin restricts Sonic hedgehog pathway usage and drug sensitivity.
Soft, Dynamic Hydrogel Confinement Improves Kidney Organoid Lumen Morphology and Reduces Epithelial-Mesenchymal Transition in Culture.
Differential requirements for the Eps15 homology domain proteins EHD4 and EHD2 in the regulation of mammalian ciliogenesis.
A pair of primers facing at the double-strand break site enables to detect NHEJ-mediated indel mutations at a 1-bp resolution.
Palmitic acid control of ciliogenesis modulates insulin signaling in hypothalamic neurons through an autophagy-dependent mechanism.
Modulation of Primary Cilia by Alvocidib Inhibition of CILK1.
Chondrocyte-specific response to stiffness-mediated primary cilia formation and centriole positioning.
Porcine choroid plexus-Riems cell line demonstrates altered polarization of transport proteins compared with the native epithelium.
TTC30A and TTC30B Redundancy Protects IFT Complex B Integrity and Its Pivotal Role in Ciliogenesis.
Ciliary Type III Adenylyl Cyclase in the VMH Is Crucial for High-Fat Diet-Induced Obesity Mediated by Autophagy.
Vangl2 participates in the primary ciliary assembly under low fluid shear stress in hUVECs.
The N-terminal Leu-Pro-Gln sequence of Rab34 is required for ciliogenesis in hTERT-RPE1 cells.
Molecular mechanisms underlying the role of the centriolar CEP164-TTBK2 complex in ciliopathies.
Genome-wide screening in human kidney organoids identifies developmental and disease-related aspects of nephrogenesis.
Evolutionarily conserved genetic interactions between nphp-4 and bbs-5 mutations exacerbate ciliopathy phenotypes.
LUBAC regulates ciliogenesis by promoting CP110 removal from the mother centriole.
Theobroma cacao improves bone growth by modulating defective ciliogenesis in a mouse model of achondroplasia.
C2cd6-encoded CatSperτ targets sperm calcium channel to Ca(2+) signaling domains in the flagellar membrane.
KIAA0319 influences cilia length, cell migration and mechanical cell-substrate interaction.
Hedgehog-induced ciliary trafficking of kinesin-4 motor KIF7 requires intraflagellar transport but not KIF7's microtubule binding.
Regulators of tubulin polyglutamylation control nuclear shape and cilium disassembly by balancing microtubule and actin assembly.
Mutations in OSBPL2 cause hearing loss associated with primary cilia defects via sonic hedgehog signaling.
TP53 promotes lineage commitment of human embryonic stem cells through ciliogenesis and sonic hedgehog signaling.
Cytoskeletal Protein 4.1G Is Essential for the Primary Ciliogenesis and Osteoblast Differentiation in Bone Formation.
GLI transcriptional repression is inert prior to Hedgehog pathway activation.
The ARF GAPs ELMOD1 and ELMOD3 act at the Golgi and cilia to regulate ciliogenesis and ciliary protein traffic.
ExSTED microscopy reveals contrasting functions of dopamine and somatostatin CSF-c neurons along the lamprey central canal.
Biallelic loss-of-function variants in EXOC6B are associated with impaired primary ciliogenesis and cause spondylo-epi-metaphyseal dysplasia with joint laxity type 3.
ATM- and ATR-induced primary ciliogenesis promotes cisplatin resistance in pancreatic ductal adenocarcinoma.
Primary Cilia Restrain PI3K-AKT Signaling to Orchestrate Human Decidualization.
IFT-A structure reveals carriages for membrane protein transport into cilia.
Primary cilia suppress Ripk3-mediated necroptosis.
Non-cell-autonomous activation of hedgehog signaling contributes to disease progression in a mouse model of renal cystic ciliopathy.
FOXA1 is a transcriptional activator of Odf2/Cenexin and regulates primary ciliation.
Characterization of TRPV4-mediated signaling pathways in an optimized human choroid plexus epithelial cell line.
Distribution of prototypical primary cilia markers in subtypes of retinal ganglion cells.
Disruption of GMNC-MCIDAS multiciliogenesis program is critical in choroid plexus carcinoma development.
Ciliogenesis mechanisms mediated by PAK2-ARL13B signaling in brain endothelial cells is responsible for vascular stability.
Monoallelic pathogenic ALG5 variants cause atypical polycystic kidney disease and interstitial fibrosis.
A cAMP signalosome in primary cilia drives gene expression and kidney cyst formation.
Heterogeneity of the NIH3T3 Fibroblast Cell Line.
GJA1 depletion causes ciliary defects by affecting Rab11 trafficking to the ciliary base.
Subtype-Selective Positive Modulation of K(Ca)2.3 Channels Increases Cilia Length.
Primary cilia and SHH signaling impairments in human and mouse models of Parkinson's disease.
The LRRK2 signaling network converges on a centriolar phospho-Rab10/RILPL1 complex to cause deficits in centrosome cohesion and cell polarization.
Precise control of microtubule disassembly in living cells.
Modelling ciliopathy phenotypes in human tissues derived from pluripotent stem cells with genetically ablated cilia.
CDK7/12/13 inhibition targets an oscillating leukemia stem cell network and synergizes with venetoclax in acute myeloid leukemia.
Phylogenetic profiling and cellular analyses of ARL16 reveal roles in traffic of IFT140 and INPP5E.
Mitotic Maturation Compensates for Premature Centrosome Splitting and PCM Loss in Human cep135 Knockout Cells.
ATXN10 Is Required for Embryonic Heart Development and Maintenance of Epithelial Cell Phenotypes in the Adult Kidney and Pancreas.
Centriolar Protein C2cd3 Is Required for Craniofacial Development.
Primary Cilia Formation Does Not Rely on WNT/β-Catenin Signaling.
Loss of Ciliary Gene Bbs8 Results in Physiological Defects in the Retinal Pigment Epithelium.
Palmitoylation of acetylated tubulin and association with ceramide-rich platforms is critical for ciliogenesis.
Primary Cilia as a Biomarker in Mesenchymal Stem Cells Senescence: Influencing Osteoblastic Differentiation Potency Associated with Hedgehog Signaling Regulation.
Differential requirement of NPHP1 for compartmentalized protein localization during photoreceptor outer segment development and maintenance.
TALPID3/KIAA0586 Regulates Multiple Aspects of Neuromuscular Patterning During Gastrointestinal Development in Animal Models and Human.
Primary Cilia Mediate TSH-Regulated Thyroglobulin Endocytic Pathways.
Quantitative lineage analysis identifies a hepato-pancreato-biliary progenitor niche.
The type 3 adenylyl cyclase is crucial for intestinal mucosal neural network in the gut lamina propria.
Recombinant Antithrombin Attenuates Acute Respiratory Distress Syndrome in Experimental Endotoxemia.
Cilium induction triggers differentiation of glioma stem cells.
2-IPMA Ameliorates PM2.5-Induced Inflammation by Promoting Primary Ciliogenesis in RPE Cells.
A ciliopathy complex builds distal appendages to initiate ciliogenesis.
MAPK15 Controls Hedgehog Signaling in Medulloblastoma Cells by Regulating Primary Ciliogenesis.
Bardet-Biedl syndrome proteins modulate the release of bioactive extracellular vesicles.
Elucidation of Distinct Modular Assemblies of Smoothened Receptor by Bitopic Ligand Measurement.
Identification of disease-relevant modulators of the SHH pathway in the developing brain.
Oxidative stress following acute kidney injury causes disruption of lung cell cilia and their release into the bronchoaveolar lavage fluid and lung injury, which are exacerbated by Idh2 deletion.
DZIP1 regulates mammalian cardiac valve development through a Cby1-β-catenin mechanism.
Cep55 regulation of PI3K/Akt signaling is required for neocortical development and ciliogenesis.
Human brain organoids assemble functionally integrated bilateral optic vesicles.
Induction of inverted morphology in brain organoids by vertical-mixing bioreactors.
Ciliary Hedgehog signaling regulates cell survival to build the facial midline.
Primary Ciliogenesis by 2-Isopropylmalic Acid Prevents PM2.5-Induced Inflammatory Response and MMP-1 Activation in Human Dermal Fibroblasts and a 3-D-Skin Model.
Δ9-Tetrahydrocannabinol inhibits Hedgehog-dependent patterning during development.
Gene replacement therapy restores RCBTB1 expression and cilium length in patient-derived retinal pigment epithelium.
A WDR35-dependent coat protein complex transports ciliary membrane cargo vesicles to cilia.
Extracellular LGALS3BP regulates neural progenitor position and relates to human cortical complexity.
Hydrogen sulfide, a gaseous signaling molecule, elongates primary cilia on kidney tubular epithelial cells by activating extracellular signal-regulated kinase.
Kif3a deletion prevents primary cilia assembly on oligodendrocyte progenitor cells, reduces oligodendrogenesis and impairs fine motor function.
LRRK2-phosphorylated Rab10 sequesters Myosin Va with RILPL2 during ciliogenesis blockade.
Deletion of Sox9 in the liver leads to hepatic cystogenesis in mice by transcriptionally downregulating Sec63.
De novo purine biosynthesis is a major driver of chemoresistance in glioblastoma.
Time-resolved proteomics profiling of the ciliary Hedgehog response.
The TBC1D31/praja2 complex controls primary ciliogenesis through PKA-directed OFD1 ubiquitylation.
Primary cilia-dependent lipid raft/caveolin dynamics regulate adipogenesis.
CiliaQ: a simple, open-source software for automated quantification of ciliary morphology and fluorescence in 2D, 3D, and 4D images.
Investigating Primary Cilia during Peripheral Nervous System Formation.
Identification of a potent antagonist of smoothened in hedgehog signaling.
Sub-toxic levels of cobalt ions impair chondrocyte mechanostranduction via HDAC6-dependent primary cilia shortening.
Exovesicular-Shh confers Imatinib resistance by upregulating Bcl2 expression in chronic myeloid leukemia with variant chromosomes.
Exon junction complex dependent mRNA localization is linked to centrosome organization during ciliogenesis.
Ciliopathy protein HYLS1 coordinates the biogenesis and signaling of primary cilia by activating the ciliary lipid kinase PIPKIγ.
Increase in primary cilia in the epidermis of patients with atopic dermatitis and psoriasis.
Genotoxic stress-activated DNA-PK-p53 cascade and autophagy cooperatively induce ciliogenesis to maintain the DNA damage response.
Disrupted intraflagellar transport due to IFT74 variants causes Joubert syndrome.
Ciliogenesis is Not Directly Regulated by LRRK2 Kinase Activity in Neurons.
Automatic and unbiased segmentation and quantification of myofibers in skeletal muscle.
Identification of Cilia in Different Mouse Tissues.
Modeling retinitis pigmentosa through patient-derived retinal organoids.
Comprehensive interactome profiling of the human Hsp70 network highlights functional differentiation of J domains.
Trace Amine-Associated Receptor 1 Trafficking to Cilia of Thyroid Epithelial Cells.
TRIM37 prevents formation of condensate-organized ectopic spindle poles to ensure mitotic fidelity.
Myocardin-related transcription factor and serum response factor regulate cilium turnover by both transcriptional and local mechanisms.
CEP78 functions downstream of CEP350 to control biogenesis of primary cilia by negatively regulating CP110 levels.
Rab34 GTPase mediates ciliary membrane formation in the intracellular ciliogenesis pathway.
Rab34 is necessary for early stages of intracellular ciliogenesis.
Targeting an anchored phosphatase-deacetylase unit restores renal ciliary homeostasis.
Sequences in the stalk domain regulate auto-inhibition and ciliary tip localization of the immotile kinesin-4 KIF7.
TMEM16A/ANO1 calcium-activated chloride channel as a novel target for the treatment of human respiratory syncytial virus infection.
DAB2IP modulates primary cilia formation associated with renal tumorigenesis.
Nonredundant roles of DIAPHs in primary ciliogenesis.
BBS Proteins Affect Ciliogenesis and Are Essential for Hedgehog Signaling, but Not for Formation of iPSC-Derived RPE-65 Expressing RPE-Like Cells.
LPA signaling acts as a cell-extrinsic mechanism to initiate cilia disassembly and promote neurogenesis.
Human cells lacking CDC14A and CDC14B show differences in ciliogenesis but not in mitotic progression.
Application of green tea catechins, polysaccharides, and flavonol prevent fine dust induced bronchial damage by modulating inflammation and airway cilia.
Interaction of INPP5E with ARL13B is essential for its ciliary membrane retention but dispensable for its ciliary entry.
EFA6A, an exchange factor for Arf6, regulates early steps in ciliogenesis.
Rabl2 GTP hydrolysis licenses BBSome-mediated export to fine-tune ciliary signaling.
Cooperation of the IFT-A complex with the IFT-B complex is required for ciliary retrograde protein trafficking and GPCR import.
Phosphosite T674A mutation in kinesin family member 3A fails to reproduce tissue and ciliary defects characteristic of CILK1 loss of function.
Functional inhibition of fatty acid binding protein 4 ameliorates impaired ciliogenesis in GCs.
Loss of primary cilia promotes mitochondria-dependent apoptosis in thyroid cancer.
Expression of α-Tubulin Acetyltransferase 1 and Tubulin Acetylation as Selective Forces in Cell Competition.
CEP55 promotes cilia disassembly through stabilizing Aurora A kinase.
YAP/TAZ maintain the proliferative capacity and structural organization of radial glial cells during brain development.
The 3-beta-hydroxysteroid-Delta(8), Delta(7)-isomerase EBP inhibits cholesterylation of Smoothened.
Cisplatin induces lung cell cilia disruption and lung damage via oxidative stress.
Quantitative Determination of Primary Cilia Protein Distribution Using Immunofluorescence Staining and MATLAB Analysis.
Loss of Planar Cell Polarity Effector Fuzzy Causes Renal Hypoplasia by Disrupting Several Signaling Pathways.
Mitral Valve Prolapse Induces Regionalized Myocardial Fibrosis.
Endoderm development requires centrioles to restrain p53-mediated apoptosis in the absence of ERK activity.
Exocyst complex mediates recycling of internal cilia.
Biallelic deep intronic variant c.5457+81T>A in TRIP11 causes loss of function and results in achondrogenesis 1A.
Identification and characterization of primary cilia-positive salivary gland tumours exhibiting basaloid/myoepithelial differentiation.
Differential Roles of Tubby Family Proteins in Ciliary Formation and Trafficking.
Impaired Ciliary Beat Frequency and Ciliogenesis Alteration during Airway Epithelial Cell Differentiation in COPD.
FBF1 deficiency promotes beiging and healthy expansion of white adipose tissue.
Loss of the ciliary protein Chibby1 in mice leads to exocrine pancreatic degeneration and pancreatitis.
A polycystin-2 protein with modified channel properties leads to an increased diameter of renal tubules and to renal cysts.
Targeted Ablation of Primary Cilia in Differentiated Dopaminergic Neurons Reduces Striatal Dopamine and Responsiveness to Metabolic Stress.
Multifaceted Functions of Rab23 on Primary Cilium-Mediated and Hedgehog Signaling-Mediated Cerebellar Granule Cell Proliferation.
Xpo7 negatively regulates Hedgehog signaling by exporting Gli2 from the nucleus.
A CRISPR and high-content imaging assay compliant with ACMG/AMP guidelines for clinical variant interpretation in ciliopathies.
A mouse model of BBS identifies developmental and homeostatic effects of BBS5 mutation and identifies novel pituitary abnormalities.
WDR35 is involved in subcellular localization of acetylated tubulin in 293T cells.
Roles for ELMOD2 and Rootletin in ciliogenesis.
Rpgrip1l controls ciliary gating by ensuring the proper amount of Cep290 at the vertebrate transition zone.
A ZFYVE19 gene mutation associated with neonatal cholestasis and cilia dysfunction: case report with a novel pathogenic variant.
HDAC6 Signaling at Primary Cilia Promotes Proliferation and Restricts Differentiation of Glioma Cells.
Characterization of primary cilia features reveal cell-type specific variability in in vitro models of osteogenic and chondrogenic differentiation.
FOP Negatively Regulates Ciliogenesis and Promotes Cell Cycle Re-entry by Facilitating Primary Cilia Disassembly.
Effect of Cell Spreading on Rosette Formation by Human Pluripotent Stem Cell-Derived Neural Progenitor Cells.
CEP164 Deficiency Causes Hyperproliferation of Pancreatic Cancer Cells.
Distinct Roles of TRAPPC8 and TRAPPC12 in Ciliogenesis via Their Interactions With OFD1.
Patient-Specific Retinal Organoids Recapitulate Disease Features of Late-Onset Retinitis Pigmentosa.
Analysis of Caveolin in Primary Cilia.
A Shh/Gli-driven three-node timer motif controls temporal identity and fate of neural stem cells.
Loss of polycystins suppresses deciliation via the activation of the centrosomal integrity pathway.
Loss of Cilia Does Not Slow Liver Disease Progression in Mouse Models of Autosomal Recessive Polycystic Kidney Disease.
Live imaging of hair bundle polarity acquisition demonstrates a critical timeline for transcription factor Emx2.
Cell Types of the Human Retina and Its Organoids at Single-Cell Resolution.
Mosaic Analysis with Double Markers reveals IGF1R function in granule cell progenitors during cerebellar development.
Protein phosphatase 1 regulatory subunit 35 is required for ciliogenesis, notochord morphogenesis, and cell-cycle progression during murine development.
Primary cilia mediate parathyroid hormone receptor type 1 osteogenic actions in osteocytes and osteoblasts via Gli activation.
Mutations in the exocyst component EXOC2 cause severe defects in human brain development.
A novel hypomorphic allele of Spag17 causes primary ciliary dyskinesia phenotypes in mice.
Comparative Super-Resolution Mapping of Basal Feet Reveals a Modular but Distinct Architecture in Primary and Motile Cilia.
Roles of TOG and jelly-roll domains of centrosomal protein CEP104 in its functions in cilium elongation and Hedgehog signaling.
Use of patient derived urine renal epithelial cells to confirm pathogenicity of PKHD1 alleles.
Differentiation of ciliated human midbrain-derived LUHMES neurons.
Centrosome and ciliary abnormalities in fetal akinesia deformation sequence human fibroblasts.
Mutations in GRK2 cause Jeune syndrome by impairing Hedgehog and canonical Wnt signaling.
LRP2 controls sonic hedgehog-dependent differentiation of cardiac progenitor cells during outflow tract formation.
Primary cilia mediate early life programming of adiposity through lysosomal regulation in the developing mouse hypothalamus.
Primary cilium remodeling mediates a cell signaling switch in differentiating neurons.
Pannexin-1 mediates fluid shear stress-sensitive purinergic signaling and cyst growth in polycystic kidney disease.
Genetic interaction of mammalian IFT-A paralogs regulates cilia disassembly, ciliary entry of membrane protein, Hedgehog signaling, and embryogenesis.
ECE2 regulates neurogenesis and neuronal migration during human cortical development.
TALPID3 and ANKRD26 selectively orchestrate FBF1 localization and cilia gating.
Profilin1-Dependent F-Actin Assembly Controls Division of Apical Radial Glia and Neocortex Development.
Prominins control ciliary length throughout the animal kingdom: New lessons from human prominin-1 and zebrafish prominin-3.
ARL13B regulates Sonic hedgehog signaling from outside primary cilia.
Conditional switching of KIF2A mutation provides new insights into cortical malformation pathogeny.
Nek2 kinase displaces distal appendages from the mother centriole prior to mitosis.
Functional Alterations in Ciliogenesis-Associated Kinase 1 (CILK1) that Result from Mutations Linked to Juvenile Myoclonic Epilepsy.
Primary cilia mediate Klf2-dependant Notch activation in regenerating heart.
A novel CDK-independent function of p27(Kip1) in preciliary vesicle trafficking during ciliogenesis.
Epb41l5 interacts with Iqcb1 and regulates ciliary function in zebrafish embryos.
Nanobody-directed targeting of optogenetic tools to study signaling in the primary cilium.
Loss of CPAP in developing mouse brain and its functional implication for human primary microcephaly.
mTOR and S6K1 drive polycystic kidney by the control of Afadin-dependent oriented cell division.
CCDC61/VFL3 Is a Paralog of SAS6 and Promotes Ciliary Functions.
Insufficiency of ciliary cholesterol in hereditary Zellweger syndrome.
Robust classification of cell cycle phase and biological feature extraction by image-based deep learning.
Intracellular calcium response of primary cilia of tubular cells to modulated shear stress under oxidative stress.
Ciliary proteins specify the cell inflammatory response by tuning NFκB signalling, independently of primary cilia.
Ubiquitin links smoothened to intraflagellar transport to regulate Hedgehog signaling.
Ciliogenesis and Hedgehog signalling are suppressed downstream of KRAS during acinar-ductal metaplasia in mouse.
Meningioma cells express primary cilia but do not transduce ciliary Hedgehog signals.
Transient Primary Cilia Mediate Robust Hedgehog Pathway-Dependent Cell Cycle Control.
Desert hedgehog-primary cilia cross talk shapes mitral valve tissue by organizing smooth muscle actin.
Airway epithelial cell differentiation relies on deficient Hedgehog signalling in COPD.
Intraflagellar-transport A dysfunction causes hyperphagia-induced systemic insulin resistance in a pre-obese state.
ESCRT subunit CHMP4B localizes to primary cilia and is required for the structural integrity of the ciliary membrane.
CENPV Is a CYLD-Interacting Molecule Regulating Ciliary Acetylated α-Tubulin.
hVFL3/CCDC61 is a component of mother centriole subdistal appendages required for centrosome cohesion and positioning.
The novel testicular enrichment protein Cfap58 is required for Notch-associated ciliogenesis.
MetAP2 inhibition reduces food intake and body weight in a ciliopathy mouse model of obesity.
The association of microcephaly protein WDR62 with CPAP/IFT88 is required for cilia formation and neocortical development.
PI3KC2α-dependent and VPS34-independent generation of PI3P controls primary cilium-mediated autophagy in response to shear stress.
TTBK2 and primary cilia are essential for the connectivity and survival of cerebellar Purkinje neurons.
NRF2 negatively regulates primary ciliogenesis and hedgehog signaling.
CEP41-mediated ciliary tubulin glutamylation drives angiogenesis through AURKA-dependent deciliation.
Nubp2 is required for cranial neural crest survival in the mouse.
Altered gene regulation as a candidate mechanism by which ciliopathy gene SDCCAG8 contributes to schizophrenia and cognitive function.
DIAPH1 regulates ciliogenesis and trafficking in primary cilia.
Serotonin Receptor 2A Activation Promotes Evolutionarily Relevant Basal Progenitor Proliferation in the Developing Neocortex.
Gpr37l1/prosaposin receptor regulates Ptch1 trafficking, Shh production, and cell proliferation in cerebellar primary astrocytes.
Sonic hedgehog signalling as a potential endobronchial biomarker in COPD.
The novel ciliogenesis regulator DYRK2 governs Hedgehog signaling during mouse embryogenesis.
Polycystin-1 regulates ARHGAP35-dependent centrosomal RhoA activation and ROCK signaling.
An interphase pool of KIF11 localizes at the basal bodies of primary cilia and a reduction in KIF11 expression alters cilia dynamics.
Primary Ciliary Deficits in the Dentate Gyrus of Fragile X Syndrome.
An IFT20 mechanotrafficking axis is required for integrin recycling, focal adhesion dynamics, and polarized cell migration.
A reduction of primary cilia but not hedgehog signaling disrupts morphogenesis in testicular organoids.
Ptch2/Gas1 and Ptch1/Boc differentially regulate Hedgehog signalling in murine primordial germ cell migration.
The HOPS complex subunit VPS39 controls ciliogenesis through autophagy.
The primary cilium dampens proliferative signaling and represses a G2/M transcriptional network in quiescent myoblasts.
Roles for IFT172 and Primary Cilia in Cell Migration, Cell Division, and Neocortex Development.
Ocular Ciliopathies: Genetic and Mechanistic Insights into Developing Therapies.
Pharmacological cholesterol depletion disturbs ciliogenesis and ciliary function in developing zebrafish.
Induction of Neural Crest Stem Cells From Bardet-Biedl Syndrome Patient Derived hiPSCs.
661W Photoreceptor Cell Line as a Cell Model for Studying Retinal Ciliopathies.
Centrosome-derived microtubule radial array, PCM-1 protein, and primary cilia formation.
Patient-derived lung cancer organoids as in vitro cancer models for therapeutic screening.
Ahi1 promotes Arl13b ciliary recruitment, regulates Arl13b stability and is required for normal cell migration.
NRF2-dependent gene expression promotes ciliogenesis and Hedgehog signaling.
Requirement of IFT-B-BBSome complex interaction in export of GPR161 from cilia.
Neddylation inhibitor MLN4924 suppresses cilia formation by modulating AKT1.
Planar Cell Polarity Effector Proteins Inturned and Fuzzy Form a Rab23 GEF Complex.
Centrosomal and ciliary targeting of CCDC66 requires cooperative action of centriolar satellites, microtubules and molecular motors.
Differential requirements for the EF-hand domains of human centrin 2 in primary ciliogenesis and nucleotide excision repair.
KCTD15 inhibits the Hedgehog pathway in Medulloblastoma cells by increasing protein levels of the oncosuppressor KCASH2.
Omega-3 Fatty Acids Activate Ciliary FFAR4 to Control Adipogenesis.
microRNAs Regulating Human and Mouse Naïve Pluripotency.
RAB8, RAB10 and RILPL1 contribute to both LRRK2 kinase-mediated centrosomal cohesion and ciliogenesis deficits.
A Sexually Dimorphic Role for STAT3 in Sonic Hedgehog Medulloblastoma.
Primary Cilia Exhibit Mechanosensitivity to Cyclic Tensile Strain and Lineage-Dependent Expression in Adipose-Derived Stem Cells.
The ciliary GTPase Arl3 maintains tissue architecture by directing planar spindle orientation during epidermal morphogenesis.
Endosomal trafficking defects in patient cells with KIAA1109 biallelic variants.
Indian Hedgehog links obesity to development of hepatocellular carcinoma.
Fine particulate matter (PM2.5) inhibits ciliogenesis by increasing SPRR3 expression via c-Jun activation in RPE cells and skin keratinocytes.
Fibroblast growth factor receptor influences primary cilium length through an interaction with intestinal cell kinase.
Homeostatic and tumourigenic activity of SOX2+ pituitary stem cells is controlled by the LATS/YAP/TAZ cascade.
HDAC1 and HDAC2 Regulate Intermediate Progenitor Positioning to Safeguard Neocortical Development.
Decreased Expression of Cilia Genes in Pancreatic Islets as a Risk Factor for Type 2 Diabetes in Mice and Humans.
The deubiquitinating enzyme Usp14 controls ciliogenesis and Hedgehog signaling.
Elevated miR-200a and miR-141 inhibit endocrine gland-derived vascular endothelial growth factor expression and ciliogenesis in preeclampsia.
Fbxo41 Promotes Disassembly of Neuronal Primary Cilia.
Silencing Y-box binding protein-1 inhibits triple-negative breast cancer cell invasiveness via regulation of MMP1 and beta-catenin expression.
Extraciliary roles of the ciliopathy protein JBTS17 in mitosis and neurogenesis.
Characterization of Functional Primary Cilia in Human Induced Pluripotent Stem Cell-Derived Neurons.
Mechanical loading inhibits cartilage inflammatory signalling via an HDAC6 and IFT-dependent mechanism regulating primary cilia elongation.
Chloroquine inhibits human retina pigmented epithelial cell growth and microtubule nucleation by downregulating p150(glued).
Primary cilium loss in mammalian cells occurs predominantly by whole-cilium shedding.
Ciliopathy-Associated Protein Kinase ICK Requires Its Non-Catalytic Carboxyl-Terminal Domain for Regulation of Ciliogenesis.
Targeted exon skipping rescues ciliary protein composition defects in Joubert syndrome patient fibroblasts.
Spastin Contributes to Neural Development through the Regulation of Microtubule Dynamics in the Primary Cilia of Neural Stem Cells.
Unexpected Activities in Regulating Ciliation Contribute to Off-target Effects of Targeted Drugs.
CCDC114 is mutated in patient with a complex phenotype combining primary ciliary dyskinesia, sensorineural deafness, and renal disease.
Fragmentation of kidney epithelial cell primary cilia occurs by cisplatin and these cilia fragments are excreted into the urine.
Reproducibility and staging of 3D human retinal organoids across multiple pluripotent stem cell lines.
Hydrocephalus in a rat model of Meckel Gruber syndrome with a TMEM67 mutation.
Prominin-1 controls stem cell activation by orchestrating ciliary dynamics.
Bardet-Biedl Syndrome proteins regulate cilia disassembly during tissue maturation.
Carbocisteine stimulated an increase in ciliary bend angle via a decrease in [Cl(-)](i) in mouse airway cilia.
Loss of Primary Cilia Results in the Development of Cancer in the Murine Thyroid Gland.
Recycling endosomal CD133 functions as an inhibitor of autophagy at the pericentrosomal region.
Integrated miRNA and mRNA expression profiling identifies novel targets and pathological mechanisms in autoimmune thyroid diseases.
Rab7 regulates primary cilia disassembly through cilia excision.
Primary cilia deficiency in neural crest cells models anterior segment dysgenesis in mouse.
Primary cilia mediate mitochondrial stress responses to promote dopamine neuron survival in a Parkinson's disease model.
AAV-Mediated Gene Augmentation Therapy Restores Critical Functions in Mutant PRPF31(+/-) iPSC-Derived RPE Cells.
Neutrophil gelatinase-associated lipocalin (NGAL) is localised to the primary cilium in renal tubular epithelial cells - A novel source of urinary biomarkers of renal injury.
Mutations in ARL2BP, a protein required for ciliary microtubule structure, cause syndromic male infertility in humans and mice.
HDAC3 and HDAC8 are required for cilia assembly and elongation.
Mutations in the Heterotopia Gene Eml1/EML1 Severely Disrupt the Formation of Primary Cilia.
Characterization of an immortalized human small airway basal stem/progenitor cell line with airway region-specific differentiation capacity.
A null allele of Dnaaf2 displays embryonic lethality and mimics human ciliary dyskinesia.
Human IFT52 mutations uncover a novel role for the protein in microtubule dynamics and centrosome cohesion.
Restoration of primary cilia in obese adipose-derived mesenchymal stem cells by inhibiting Aurora A or extracellular signal-regulated kinase.
A CEP104-CSPP1 Complex Is Required for Formation of Primary Cilia Competent in Hedgehog Signaling.
Mutagenesis of putative ciliary genes with the CRISPR/Cas9 system in zebrafish identifies genes required for retinal development.
Tubular flow activates magnesium transport in the distal convoluted tubule.
The RNA-Protein Interactome of Differentiated Kidney Tubular Epithelial Cells.
CEP120 interacts with C2CD3 and Talpid3 and is required for centriole appendage assembly and ciliogenesis.
Acute Inhibition of Heterotrimeric Kinesin-2 Function Reveals Mechanisms of Intraflagellar Transport in Mammalian Cilia.
Glioma cell proliferation is enhanced in the presence of tumor-derived cilia vesicles.
Adenylyl cyclases and TRPV4 mediate Ca(2+)/cAMP dynamics to enhance fluid flow-induced osteogenesis in osteocytes.
Ciliogenesis is reciprocally regulated by PPARA and NR1H4/FXR through controlling autophagy in vitro and in vivo.
Dorsoventral differences in cAMP levels and correlated changes in the subcellular distribution of the PKA catalytic domain, provide further evidence that PKA signaling coordinates dorsoventral patterning of the otocyst.
A distal centriolar protein network controls organelle maturation and asymmetry.
LRRC45 contributes to early steps of axoneme extension.
A slow-cycling LGR5 tumour population mediates basal cell carcinoma relapse after therapy.
Homozygous variants in KIAA1549, encoding a ciliary protein, are associated with autosomal recessive retinitis pigmentosa.
Characterization of Primary Cilia in Normal Fallopian Tube Epithelium and Serous Tubal Intraepithelial Carcinoma.
The Ciliary Machinery Is Repurposed for T Cell Immune Synapse Trafficking of LCK.
ARL3 Mutations Cause Joubert Syndrome by Disrupting Ciliary Protein Composition.
M-Phase Phosphoprotein 9 regulates ciliogenesis by modulating CP110-CEP97 complex localization at the mother centriole.
Dynein-2 intermediate chains play crucial but distinct roles in primary cilia formation and function.
Disrupted alternative splicing for genes implicated in splicing and ciliogenesis causes PRPF31 retinitis pigmentosa.
Mutation of FOP/FGFR1OP in mice recapitulates human short rib-polydactyly ciliopathy.
Membrane association and remodeling by intraflagellar transport protein IFT172.
A Cell/Cilia Cycle Biosensor for Single-Cell Kinetics Reveals Persistence of Cilia after G1/S Transition Is a General Property in Cells and Mice.
Oncoprotein CIP2A promotes the disassembly of primary cilia and inhibits glycolytic metabolism.
Primary cilia sensitize endothelial cells to BMP and prevent excessive vascular regression.
The Joubert syndrome protein ARL13B binds tubulin to maintain uniform distribution of proteins along the ciliary membrane.
Regulator of calcineurin-2 is a centriolar protein with a role in cilia length control.
Cell type-specific regulation of ciliary transition zone assembly in vertebrates.
Loss of Tctn3 causes neuronal apoptosis and neural tube defects in mice.
Quantitative Comparison of Primary Cilia Marker Expression and Length in the Mouse Brain.
The actin-MRTF-SRF transcriptional circuit controls tubulin acetylation via α-TAT1 gene expression.
Insm1 Induces Neural Progenitor Delamination in Developing Neocortex via Downregulation of the Adherens Junction Belt-Specific Protein Plekha7.
Regulation of ciliary function by fibroblast growth factor signaling identifies FGFR3-related disorders achondroplasia and thanatophoric dysplasia as ciliopathies.
Mice Expressing Myc in Neural Precursors Develop Choroid Plexus and Ciliary Body Tumors.
Primary Cilia Mediate Diverse Kinase Inhibitor Resistance Mechanisms in Cancer.
Targeted deletion of the AAA-ATPase Ruvbl1 in mice disrupts ciliary integrity and causes renal disease and hydrocephalus.
Autophagy alteration prevents primary cilium disassembly in RPE1 cells.
Cilia protein IFT88 regulates extracellular protease activity by optimizing LRP-1-mediated endocytosis.
Primary cilia-regulated transcriptome in the renal collecting duct.
CRISPR/Cas9-mediated Genomic Editing of Cluap1/IFT38 Reveals a New Role in Actin Arrangement.
EZH2-Mediated Primary Cilium Deconstruction Drives Metastatic Melanoma Formation.
Brain Somatic Mutations in MTOR Disrupt Neuronal Ciliogenesis, Leading to Focal Cortical Dyslamination.
Glucose deprivation induces primary cilium formation through mTORC1 inactivation.
CDKL Family Kinases Have Evolved Distinct Structural Features and Ciliary Function.
Primary Cilium-Mediated Retinal Pigment Epithelium Maturation Is Disrupted in Ciliopathy Patient Cells.
Functional characterization of tektin-1 in motile cilia and evidence for TEKT1 as a new candidate gene for motile ciliopathies.
Ciliogenesis and cell cycle alterations contribute to KIF2A-related malformations of cortical development.
Constitutively-active FGFR3 disrupts primary cilium length and IFT20 trafficking in various chondrocyte models of achondroplasia.
Adenylyl cyclase 5 deficiency reduces renal cyclic AMP and cyst growth in an orthologous mouse model of polycystic kidney disease.
Export of membrane proteins from the Golgi complex to the primary cilium requires the kinesin motor, KIFC1.
WDR11-mediated Hedgehog signalling defects underlie a new ciliopathy related to Kallmann syndrome.
Deficiency of primary cilia in kidney epithelial cells induces epithelial to mesenchymal transition.
DNAAF1 links heart laterality with the AAA+ ATPase RUVBL1 and ciliary intraflagellar transport.
Truncated SALL1 Impedes Primary Cilia Function in Townes-Brocks Syndrome.
Reconstructing the Human Renal Vascular-Tubular Unit In Vitro.
Targeted exon skipping of a CEP290 mutation rescues Joubert syndrome phenotypes in vitro and in a murine model.
Kinesin-2 and IFT-A act as a complex promoting nuclear localization of β-catenin during Wnt signalling.
Cullin-3-KCTD10-mediated CEP97 degradation promotes primary cilium formation.
Microtubule asters anchored by FSD1 control axoneme assembly and ciliogenesis.
Loss-of-function of IFT88 determines metabolic phenotypes in thyroid cancer.
Axoneme polyglutamylation regulated by Joubert syndrome protein ARL13B controls ciliary targeting of signaling molecules.
Basal exon skipping and nonsense-associated altered splicing allows bypassing complete CEP290 loss-of-function in individuals with unusually mild retinal disease.
Aspm knockout ferret reveals an evolutionary mechanism governing cerebral cortical size.
Mapping GPR88-Venus illuminates a novel role for GPR88 in sensory processing.
Robust interaction of IFT70 with IFT52-IFT88 in the IFT-B complex is required for ciliogenesis.
Centrobin controls primary ciliogenesis in vertebrates.
Mutations in IFT-A satellite core component genes IFT43 and IFT121 produce short rib polydactyly syndrome with distinctive campomelia.
Abnormal glycosylation in Joubert syndrome type 10.
Jasplakinolide induces primary cilium formation through cell rounding and YAP inactivation.
Widespread GLI expression but limited canonical hedgehog signaling restricted to the ductular reaction in human chronic liver disease.
Missense mutations in the WD40 domain of AHI1 cause non-syndromic retinitis pigmentosa.
Reduced primary cilia length and altered Arl13b expression are associated with deregulated chondrocyte Hedgehog signaling in alkaptonuria.
The ciliary membrane-associated proteome reveals actin-binding proteins as key components of cilia.
CDK10 Mutations in Humans and Mice Cause Severe Growth Retardation, Spine Malformations, and Developmental Delays.
Chondrocyte expansion is associated with loss of primary cilia and disrupted hedgehog signalling.
Primary cilia modulate TLR4-mediated inflammatory responses in hippocampal neurons.
GPR37L1 modulates seizure susceptibility: Evidence from mouse studies and analyses of a human GPR37L1 variant.
Palmitoylation of the ciliary GTPase ARL13b is necessary for its stability and its role in cilia formation.
Loss of OCRL increases ciliary PI(4,5)P(2) in Lowe oculocerebrorenal syndrome.
A novel Cre-inducible knock-in ARL13B-tRFP fusion cilium reporter.
Neural crest cells utilize primary cilia to regulate ventral forebrain morphogenesis via Hedgehog-dependent regulation of oriented cell division.
Human Embryonic Stem Cell-Derived Cardiomyocytes Self-Arrange with Areas of Different Subtypes During Differentiation.
An ovine hepatorenal fibrocystic model of a Meckel-like syndrome associated with dysmorphic primary cilia and TMEM67 mutations.
Afadin controls cell polarization and mitotic spindle orientation in developing cortical radial glia.
Recent Zika Virus Isolates Induce Premature Differentiation of Neural Progenitors in Human Brain Organoids.
Centriole splitting caused by loss of the centrosomal linker protein C-NAP1 reduces centriolar satellite density and impedes centrosome amplification.
SOX4 regulates gonad morphogenesis and promotes male germ cell differentiation in mice.
Ciliary entry of KIF17 is dependent on its binding to the IFT-B complex via IFT46-IFT56 as well as on its nuclear localization signal.
Primary Cilium-Regulated EG-VEGF Signaling Facilitates Trophoblast Invasion.
Mutations in DZIP1L, which encodes a ciliary-transition-zone protein, cause autosomal recessive polycystic kidney disease.
Guided self-organization and cortical plate formation in human brain organoids.
Ciliary Hedgehog Signaling Restricts Injury-Induced Adipogenesis.
In Vitro Modeling Using Ciliopathy-Patient-Derived Cells Reveals Distinct Cilia Dysfunctions Caused by CEP290 Mutations.
The CEP19-RABL2 GTPase Complex Binds IFT-B to Initiate Intraflagellar Transport at the Ciliary Base.
Arl3 and RP2 regulate the trafficking of ciliary tip kinesins.
Primary Cilia in the Murine Cerebellum and in Mutant Models of Medulloblastoma.
KIF13B establishes a CAV1-enriched microdomain at the ciliary transition zone to promote Sonic hedgehog signalling.
Dynamic Remodeling of Membrane Composition Drives Cell Cycle through Primary Cilia Excision.
HDAC2 promotes loss of primary cilia in pancreatic ductal adenocarcinoma.
Zfp462 deficiency causes anxiety-like behaviors with excessive self-grooming in mice.
Regulation of ciliary retrograde protein trafficking by the Joubert syndrome proteins ARL13B and INPP5E.
A novel homozygous ARL13B variant in patients with Joubert syndrome impairs its guanine nucleotide-exchange factor activity.
Primary Cilium Formation and Ciliary Protein Trafficking Is Regulated by the Atypical MAP Kinase MAPK15 in Caenorhabditis elegans and Human Cells.
IFT proteins spatially control the geometry of cleavage furrow ingression and lumen positioning.
An apicosome initiates self-organizing morphogenesis of human pluripotent stem cells.
The guanine nucleotide exchange factor Arf-like protein 13b is essential for assembly of the mouse photoreceptor transition zone and outer segment.
Evolutionary Proteomics Uncovers Ancient Associations of Cilia with Signaling Pathways.
Eml1 loss impairs apical progenitor spindle length and soma shape in the developing cerebral cortex.
A human patient-derived cellular model of Joubert syndrome reveals ciliary defects which can be rescued with targeted therapies.
Folate-dependent methylation of septins governs ciliogenesis during neural tube closure.
A novel role for primary cilia in airway remodeling.
Primary cilia on porcine testicular somatic cells and their role in hedgehog signaling and tubular morphogenesis in vitro.
Estrogen receptor α is required for oviductal transport of embryos.
Mutations in KIAA0753 cause Joubert syndrome associated with growth hormone deficiency.
KDM3A coordinates actin dynamics with intraflagellar transport to regulate cilia stability.
Cilia-dependent GLI processing in neural crest cells is required for tongue development.
Generation of iPSC-derived Human Brain Organoids to Model Early Neurodevelopmental Disorders.
Practical method for targeted disruption of cilia-related genes by using CRISPR/Cas9-mediated, homology-independent knock-in system.
A novel ICK mutation causes ciliary disruption and lethal endocrine-cerebro-osteodysplasia syndrome.
Measuring the primary cilium length: improved method for unbiased high-throughput analysis.
Three-dimensional retinal organoids from mouse pluripotent stem cells mimic in vivo development with enhanced stratification and rod photoreceptor differentiation.
Morphological and Functional Characterization of the Ciliary Pocket by Electron and Fluorescence Microscopy.
Methods for Visualization of Neuronal Cilia.
Targeting of ASH Domain-Containing Proteins to the Centrosome.
Immunofluorescent staining of septins in primary cilia.
Primary Cilia Negatively Regulate Melanogenesis in Melanocytes and Pigmentation in a Human Skin Model.
KIF3A binds to β-arrestin for suppressing Wnt/β-catenin signalling independently of primary cilia in lung cancer.
Destabilization of the IFT-B cilia core complex due to mutations in IFT81 causes a Spectrum of Short-Rib Polydactyly Syndrome.
Nestin Mediates Hedgehog Pathway Tumorigenesis.
RPGR, a prenylated retinal ciliopathy protein, is targeted to cilia in a prenylation- and PDE6D-dependent manner.
IFT52 mutations destabilize anterograde complex assembly, disrupt ciliogenesis and result in short rib polydactyly syndrome.
An inactivating mutation in intestinal cell kinase, ICK, impairs hedgehog signalling and causes short rib-polydactyly syndrome.
Zfp423 Regulates Sonic Hedgehog Signaling via Primary Cilium Function.
Loss of MACF1 Abolishes Ciliogenesis and Disrupts Apicobasal Polarity Establishment in the Retina.
Prenylated retinal ciliopathy protein RPGR interacts with PDE6δ and regulates ciliary localization of Joubert syndrome-associated protein INPP5E.
Craniofacial Ciliopathies Reveal Specific Requirements for GLI Proteins during Development of the Facial Midline.
Defective ciliogenesis in thyroid hürthle cell tumors is associated with increased autophagy.
Neuroanatomical characterization of the cellular and axonal architecture of subcortical band heterotopia in the BXD29-Tlr4(lps-2J)/J mouse cortex.
Role for the IFT-A Complex in Selective Transport to the Primary Cilium.
Cyclic expression of the voltage-gated potassium channel KV10.1 promotes disassembly of the primary cilium.
An organelle-specific protein landscape identifies novel diseases and molecular mechanisms.
Recessive NEK9 mutation causes a lethal skeletal dysplasia with evidence of cell cycle and ciliary defects.
Centrosomal protein CP110 controls maturation of the mother centriole during cilia biogenesis.
Arf-like Protein 3 (ARL3) Regulates Protein Trafficking and Ciliogenesis in Mouse Photoreceptors.
MCRS1 associates with cytoplasmic dynein and mediates pericentrosomal material recruitment.
Identification and Correction of Mechanisms Underlying Inherited Blindness in Human iPSC-Derived Optic Cups.
Hedgehog signaling promotes basal progenitor expansion and the growth and folding of the neocortex.
MKS1 regulates ciliary INPP5E levels in Joubert syndrome.
TMEM107 recruits ciliopathy proteins to subdomains of the ciliary transition zone and causes Joubert syndrome.
Arl13b and the exocyst interact synergistically in ciliogenesis.
Lithium chloride modulates chondrocyte primary cilia and inhibits Hedgehog signaling.
NudC regulates actin dynamics and ciliogenesis by stabilizing cofilin 1.
Endothelial primary cilia inhibit atherosclerosis.
Disruption of KIF3A in patient-derived glioblastoma cells: effects on ciliogenesis, hedgehog sensitivity, and tumorigenesis.
WDR8 is a centriolar satellite and centriole-associated protein that promotes ciliary vesicle docking during ciliogenesis.
Deletion of ADP Ribosylation Factor-Like GTPase 13B Leads to Kidney Cysts.
Airway Epithelial KIF3A Regulates Th2 Responses to Aeroallergens.
Neonatal seizures induced by pentylenetetrazol or kainic acid disrupt primary cilia growth on developing mouse cortical neurons.
Severe NDE1-mediated microcephaly results from neural progenitor cell cycle arrests at multiple specific stages.
Arl13b Interacts With Vangl2 to Regulate Cilia and Photoreceptor Outer Segment Length in Zebrafish.
Sonic Hedgehog promotes proliferation of Notch-dependent monociliated choroid plexus tumour cells.
CPAP promotes timely cilium disassembly to maintain neural progenitor pool.
Culture and detection of primary cilia in endothelial cell models.
Analysis of primary cilia in the developing mouse brain.
Imaging centrosomes and cilia in the mouse kidney.
A Group of ent-Kaurane Diterpenoids Inhibit Hedgehog Signaling and Induce Cilia Elongation.
Cell Cycle-Dependent Localization of Dynactin Subunit p150 glued at Centrosome.
Primary Cilia on Horizontal Basal Cells Regulate Regeneration of the Olfactory Epithelium.
Mutations in TRAF3IP1/IFT54 reveal a new role for IFT proteins in microtubule stabilization.
PDGFRβ and oncogenic mutant PDGFRα D842V promote disassembly of primary cilia through a PLCγ- and AURKA-dependent mechanism.
Conserved Genetic Interactions between Ciliopathy Complexes Cooperatively Support Ciliogenesis and Ciliary Signaling.
The Interaction of CCDC104/BARTL1 with Arl3 and Implications for Ciliary Function.
Loss of primary cilia in melanoma cells is likely independent of proliferation and cell cycle progression.
Myosin heavy chain 10 (MYH10) is required for centriole migration during the biogenesis of primary cilia.
Cell context-specific expression of primary cilia in the human testis and ciliary coordination of Hedgehog signalling in mouse Leydig cells.
Centrin2 regulates CP110 removal in primary cilium formation.
The heterotrimeric kinesin-2 complex interacts with and regulates GLI protein function.
Mutations in DYNC2LI1 disrupt cilia function and cause short rib polydactyly syndrome.
Ablation of retinal ciliopathy protein RPGR results in altered photoreceptor ciliary composition.
Non-Overlapping Distributions and Functions of the VDAC Family in Ciliogenesis.
A function for the Joubert syndrome protein Arl13b in ciliary membrane extension and ciliary length regulation.
Polycystin-1 maturation requires polycystin-2 in a dose-dependent manner.
The Microtubule-Depolymerizing Activity of a Mitotic Kinesin Protein KIF2A Drives Primary Cilia Disassembly Coupled with Cell Proliferation.
Mutations in human homologue of chicken talpid3 gene (KIAA0586) cause a hybrid ciliopathy with overlapping features of Jeune and Joubert syndromes.
The Orphan G Protein-coupled Receptor Gpr175 (Tpra40) Enhances Hedgehog Signaling by Modulating cAMP Levels.
An siRNA-based functional genomics screen for the identification of regulators of ciliogenesis and ciliopathy genes.
A role for Rab23 in the trafficking of Kif17 to the primary cilium.
Accumulation of non-outer segment proteins in the outer segment underlies photoreceptor degeneration in Bardet-Biedl syndrome.
BBS4 is necessary for ciliary localization of TrkB receptor and activation by BDNF.
Centrosome-kinase fusions promote oncogenic signaling and disrupt centrosome function in myeloproliferative neoplasms.
Prostaglandin signalling regulates ciliogenesis by modulating intraflagellar transport.
Centriole maturation requires regulated Plk1 activity during two consecutive cell cycles.
A mutation in the mouse ttc26 gene leads to impaired hedgehog signaling.
Primary cilia in the developing pig testis.
A homozygous missense mutation in the ciliary gene TTC21B causes familial FSGS.
Ciliary membrane proteins traffic through the Golgi via a Rabep1/GGA1/Arl3-dependent mechanism.
RFX7 is required for the formation of cilia in the neural tube.
Hypomorphism for RPGRIP1L, a ciliary gene vicinal to the FTO locus, causes increased adiposity in mice.
Primary cilia disassembly down-regulates mechanosensitive hedgehog signalling: a feedback mechanism controlling ADAMTS-5 expression in chondrocytes.
SIRT2 regulates ciliogenesis and contributes to abnormal centrosome amplification caused by loss of polycystin-1.
Ciliopathy-associated gene Cc2d2a promotes assembly of subdistal appendages on the mother centriole during cilia biogenesis.
ICK is essential for cell type-specific ciliogenesis and the regulation of ciliary transport.
Α-tubulin K40 acetylation is required for contact inhibition of proliferation and cell-substrate adhesion.
Basal body proteins regulate Notch signaling through endosomal trafficking.
Specific polar subpopulations of astral microtubules control spindle orientation and symmetric neural stem cell division.
Arhgap36-dependent activation of Gli transcription factors.
Cep164 triggers ciliogenesis by recruiting Tau tubulin kinase 2 to the mother centriole.
Disruption of the basal body protein POC1B results in autosomal-recessive cone-rod dystrophy.
The master cell cycle regulator APC-Cdc20 regulates ciliary length and disassembly of the primary cilium.
The ciliary protein Ftm is required for ventricular wall and septal development.
Disruption of CEP290 microtubule/membrane-binding domains causes retinal degeneration.
Asymmetric inheritance of centrosome-associated primary cilium membrane directs ciliogenesis after cell division.
Genetically encoded calcium indicator illuminates calcium dynamics in primary cilia.
Defects in the IFT-B component IFT172 cause Jeune and Mainzer-Saldino syndromes in humans.
Reduction of oxidative stress during recovery accelerates normalization of primary cilia length that is altered after ischemic injury in murine kidneys.
Visualizing renal primary cilia.
VDAC3 and Mps1 negatively regulate ciliogenesis.
Prickle1 is expressed in distinct cell populations of the central nervous system and contributes to neuronal morphogenesis.
The ciliary G-protein-coupled receptor Gpr161 negatively regulates the Sonic hedgehog pathway via cAMP signaling.
The microtubule affinity regulating kinase MARK4 promotes axoneme extension during early ciliogenesis.
Mutations in ARL2BP, encoding ADP-ribosylation-factor-like 2 binding protein, cause autosomal-recessive retinitis pigmentosa.
Molecular basis of tubulin transport within the cilium by IFT74 and IFT81.
CCDC41 is required for ciliary vesicle docking to the mother centriole.
ARL13B, PDE6D, and CEP164 form a functional network for INPP5E ciliary targeting.
SUMOylation of the small GTPase ARL-13 promotes ciliary targeting of sensory receptors.
Basolateral rather than apical primary cilia on neuroepithelial cells committed to delamination.
A novel protein LZTFL1 regulates ciliary trafficking of the BBSome and Smoothened.
A ciliopathy complex at the transition zone protects the cilia as a privileged membrane domain.
Guo DF, Williams PA, Laule C, Seaby C, Zhang Q, Sheffield VC, Rahmouni K
Function (Oxford, England) 2024;5(1):zqad070
Function (Oxford, England) 2024;5(1):zqad070
Immunolabel-First-Expand-Later Expansion Microscopy Approach Using Stable STED Dyes.
Kong D, Luvsanjav D, Loncarek J
Methods in molecular biology (Clifton, N.J.) 2024;2725:89-101
Methods in molecular biology (Clifton, N.J.) 2024;2725:89-101
Expansion Microscopy of Ciliary Proteins.
Park S, Shi X
Methods in molecular biology (Clifton, N.J.) 2024;2725:79-88
Methods in molecular biology (Clifton, N.J.) 2024;2725:79-88
A Chemically Inducible Organelle Rerouting Assay to Probe Primary Cilium Assembly, Maintenance, and Disassembly in Cultured Cells.
Turan FB, Ercan ME, Firat-Karalar EN
Methods in molecular biology (Clifton, N.J.) 2024;2725:55-78
Methods in molecular biology (Clifton, N.J.) 2024;2725:55-78
Proximity Mapping of Ciliary Proteins by BioID.
Iazzi M, St-Germain J, Acharya S, Raught B, Gupta GD
Methods in molecular biology (Clifton, N.J.) 2024;2725:181-198
Methods in molecular biology (Clifton, N.J.) 2024;2725:181-198
Ultrastructure expansion microscopy (U-ExM) of mouse and human kidneys for analysis of subcellular structures.
Langner E, Puapatanakul P, Pudlowski R, Alsabbagh DY, Miner JH, Horani A, Dutcher SK, Brody SL, Wang JT, Suleiman HY, Mahjoub MR
Cytoskeleton (Hoboken, N.J.) 2024 Nov;81(11):618-638
Cytoskeleton (Hoboken, N.J.) 2024 Nov;81(11):618-638
Cytoskeletal β-tubulin and cysteine cathepsin L deregulation by SARS-CoV-2 spike protein interaction with the neuronal model cell line SH-SY5Y.
Oliveira BR, Nehlmeier I, Kempf AM, Venugopalan V, Rehders M, Ceniza MEP, Cavalcanti PATPV, Hoffmann M, Pöhlmann S, Brix K
Biochimie 2024 Nov;226:49-61
Biochimie 2024 Nov;226:49-61
Rediscovering the Rete Ovarii: a secreting auxiliary structure to the ovary.
Anbarci DN, McKey J, Levic DS, Bagnat M, Capel B
bioRxiv : the preprint server for biology 2024 Nov 11;
bioRxiv : the preprint server for biology 2024 Nov 11;
Primary Cilia as a Tumor Marker in Pituitary Neuroendocrine Tumors.
Martínez-Hernández R, Serrano-Somavilla A, Fernández-Contreras R, Sanchez-Guerrero C, Sánchez de la Blanca N, Sacristán-Gómez P, Sebastian-Valles F, Sampedro-Núñez M, Fraga J, Calatayud M, Vicente A, García-de-Casasola G, Sanz-García A, Araujo-Castro M, Ruz-Caracuel I, Puig-Domingo M, Marazuela M
Modern pathology : an official journal of the United States and Canadian Academy of Pathology, Inc 2024 May;37(5):100475
Modern pathology : an official journal of the United States and Canadian Academy of Pathology, Inc 2024 May;37(5):100475
Targeting Rab-RILPL interactions as a strategy to downregulate pathogenic LRRK2 in Parkinson's disease.
Alexander KK, Naaldijk Y, Fasiczka R, Brahmia B, Chen T, Hilfiker S, Kennedy EJ
Journal of peptide science : an official publication of the European Peptide Society 2024 May;30(5):e3563
Journal of peptide science : an official publication of the European Peptide Society 2024 May;30(5):e3563
MicroRNA‑155‑5p inhibits trophoblast cell proliferation and invasion by disrupting centrosomal function.
Tsai YC, Kuo TN, Lin RC, Tsai HL, Chao YY, Lee PR, Su PJ, Wang CY
Molecular medicine reports 2024 May;29(5)
Molecular medicine reports 2024 May;29(5)
Engrailed 1 deficiency induces changes in ciliogenesis during human neuronal differentiation.
Hembach S, Schmidt S, Orschmann T, Burtscher I, Lickert H, Giesert F, Weisenhorn DV, Wurst W
Neurobiology of disease 2024 May;194:106474
Neurobiology of disease 2024 May;194:106474
Extracellular matrix hyaluronan modulates fat cell differentiation and primary cilia dynamics.
Drygalski K, Higos R, Merabtene F, Mojsak P, Grubczak K, Ciborowski M, Razak H, Clément K, Dugail I
Biochimica et biophysica acta. Molecular and cell biology of lipids 2024 May;1869(4):159470
Biochimica et biophysica acta. Molecular and cell biology of lipids 2024 May;1869(4):159470
Selective targeting of mu opioid receptors to primary cilia.
Fagan RR, Lee DF, Geron M, Scherrer G, von Zastrow M, Ehrlich AT
Cell reports 2024 May 28;43(5):114164
Cell reports 2024 May 28;43(5):114164
Assessment and Comparison of Early Developmental Toxicity of Six Per- and Polyfluoroalkyl Substances with Human Embryonic Stem Cell Models.
Zhao M, Yin N, Yang R, Li S, Zhang S, Faiola F
Environmental science & technology 2024 May 14;58(19):8215-8227
Environmental science & technology 2024 May 14;58(19):8215-8227
Bridge-like lipid transfer protein family member 2 suppresses ciliogenesis.
Parolek J, Burd CG
Molecular biology of the cell 2024 May 1;35(5):br11
Molecular biology of the cell 2024 May 1;35(5):br11
PI3KCIIα-Dependent Autophagy Program Protects From Endothelial Dysfunction and Atherosclerosis in Response to Low Shear Stress in Mice.
Nasr M, Fay A, Lupieri A, Malet N, Darmon A, Zahreddine R, Swiader A, Wahart A, Viaud J, Nègre-Salvayre A, Hirsch E, Monteyne D, Perez-Morgà D, Dupont N, Codogno P, Ramel D, Morel E, Laffargue M, Gayral S
Arteriosclerosis, thrombosis, and vascular biology 2024 Mar;44(3):620-634
Arteriosclerosis, thrombosis, and vascular biology 2024 Mar;44(3):620-634
Store-operated calcium entry inhibits primary ciliogenesis via the activation of Aurora A.
Lai YS, Chan TW, Nguyen TMH, Lin TC, Chao YY, Wang CY, Hung LY, Tsai SJ, Chiu WT
The FEBS journal 2024 Mar;291(5):1027-1042
The FEBS journal 2024 Mar;291(5):1027-1042
XIAP-mediated degradation of IFT88 disrupts HSC cilia to stimulate HSC activation and liver fibrosis.
Hong R, Tan Y, Tian X, Huang Z, Wang J, Ni H, Yang J, Bu W, Yang S, Li T, Yu F, Zhong W, Sun T, Wang X, Li D, Liu M, Yang Y, Zhou J
EMBO reports 2024 Mar;25(3):1055-1074
EMBO reports 2024 Mar;25(3):1055-1074
Bioengineered Tubular Biliary Organoids.
Elci BS, Nikolaev M, Rezakhani S, Lutolf MP
Advanced healthcare materials 2024 Mar;13(8):e2302912
Advanced healthcare materials 2024 Mar;13(8):e2302912
Primary Cilium in Neural Crest Cells Crucial for Anterior Segment Development and Corneal Avascularity.
Seo S, Sonn SK, Kweon HY, Jin J, Kume T, Ko JY, Park JH, Oh GT
Investigative ophthalmology & visual science 2024 Mar 5;65(3):30
Investigative ophthalmology & visual science 2024 Mar 5;65(3):30
Unraveling cellular complexity with transient adapters in highly multiplexed super-resolution imaging.
Schueder F, Rivera-Molina F, Su M, Marin Z, Kidd P, Rothman JE, Toomre D, Bewersdorf J
Cell 2024 Mar 28;187(7):1769-1784.e18
Cell 2024 Mar 28;187(7):1769-1784.e18
ESCRT-I protein UBAP1 controls ventricular expansion and cortical neurogenesis via modulating adherens junctions of radial glial cells.
Lu D, Zhi Y, Su H, Lin X, Lin J, Shi Y, Yi W, Hong C, Zhang T, Fu Z, Chen LY, Zhao Z, Li R, Xu Z, Chen W, Wang N, Xu D
Cell reports 2024 Mar 26;43(3):113818
Cell reports 2024 Mar 26;43(3):113818
Molecular basis promoting centriole triplet microtubule assembly.
Takeda Y, Chinen T, Honda S, Takatori S, Okuda S, Yamamoto S, Fukuyama M, Takeuchi K, Tomita T, Hata S, Kitagawa D
Nature communications 2024 Mar 22;15(1):2216
Nature communications 2024 Mar 22;15(1):2216
Mechanisms underlying morphological and functional changes of cilia in fibroblasts derived from patients bearing ARL3(T31A) and ARL3(T31A/C118F) mutations.
Zhang X, Yao S, Zhang L, Yang L, Yang M, Guo Q, Li Y, Wang Z, Lei B, Jin X
FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2024 Mar 15;38(5):e23519
FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2024 Mar 15;38(5):e23519
Diesel exhaust particle exposure exacerbates ciliary and epithelial barrier dysfunction in the multiciliated bronchial epithelium models.
Park E, Kim BY, Lee S, Son KH, Bang J, Hong SH, Lee JW, Uhm KO, Kwak HJ, Lim HJ
Ecotoxicology and environmental safety 2024 Mar 15;273:116090
Ecotoxicology and environmental safety 2024 Mar 15;273:116090
KIF3A tail domain phosphorylation is not required for ciliogenesis in mouse embryonic fibroblasts.
Fasawe AS, Adams JM, Engelke MF
iScience 2024 Mar 15;27(3):109149
iScience 2024 Mar 15;27(3):109149
The CPLANE protein Fuzzy regulates ciliogenesis by suppressing actin polymerization at the base of the primary cilium via p190A RhoGAP.
Sharma R, Kalot R, Levin Y, Babayeva S, Kachurina N, Chung CF, Liu KJ, Bouchard M, Torban E
Development (Cambridge, England) 2024 Mar 15;151(6)
Development (Cambridge, England) 2024 Mar 15;151(6)
DLG1 functions upstream of SDCCAG3 and IFT20 to control ciliary targeting of polycystin-2.
Rezi CK, Aslanyan MG, Diwan GD, Cheng T, Chamlali M, Junger K, Anvarian Z, Lorentzen E, Pauly KB, Afshar-Bahadori Y, Fernandes EFA, Qian F, Tosi S, Christensen ST, Pedersen SF, Strømgaard K, Russell RB, Miner JH, Mahjoub MR, Boldt K, Roepman R, Pedersen LB
bioRxiv : the preprint server for biology 2024 Mar 14;
bioRxiv : the preprint server for biology 2024 Mar 14;
In vivo CRISPR screening directly targeting testicular cells.
Noguchi Y, Onodera Y, Miyamoto T, Maruoka M, Kosako H, Suzuki J
Cell genomics 2024 Mar 13;4(3):100510
Cell genomics 2024 Mar 13;4(3):100510
The ARPKD Protein DZIP1L Regulates Ciliary Protein Entry by Modulating the Architecture and Function of Ciliary Transition Fibers.
Chen H, Wu Z, Yan Z, Chen C, Zhang Y, Wang Q, Gao Y, Ling K, Hu J, Wei Q
Advanced science (Weinheim, Baden-Wurttemberg, Germany) 2024 Jun;11(24):e2308820
Advanced science (Weinheim, Baden-Wurttemberg, Germany) 2024 Jun;11(24):e2308820
The Immune Checkpoint Protein PD-L1 Regulates Ciliogenesis and Hedgehog Signaling.
Agborbesong E, Li X
Cells 2024 Jun 8;13(12)
Cells 2024 Jun 8;13(12)
Aurora kinase A inhibition plus Tumor Treating Fields suppress glioma cell proliferation in a cilium-independent manner.
Tian J, Mallinger JC, Shi P, Ling D, Deleyrolle LP, Lin M, Khoshbouei H, Sarkisian MR
Translational oncology 2024 Jul;45:101956
Translational oncology 2024 Jul;45:101956
A sterol analog inhibits hedgehog pathway by blocking cholesterylation of smoothened.
Liu YB, He LM, Sun M, Luo WJ, Lin ZC, Qiu ZP, Zhang YL, Hu A, Luo J, Qiu WW, Song BL
Cell chemical biology 2024 Jul 18;31(7):1264-1276.e7
Cell chemical biology 2024 Jul 18;31(7):1264-1276.e7
Bi-allelic variants in CEP295 cause Seckel-like syndrome presenting with primary microcephaly, developmental delay, intellectual disability, short stature, craniofacial and digital abnormalities.
Li N, Xu Y, Chen H, Lin J, AlAbdi L, Bekheirnia MR, Li G, Gofin Y, Bekheirnia N, Faqeih E, Chen L, Chang G, Tang J, Yao R, Yu T, Wang X, Fu W, Fu Q, Shen Y, Alkuraya FS, Machol K, Wang J
EBioMedicine 2024 Jan;99:104940
EBioMedicine 2024 Jan;99:104940
Racgap1 knockdown results in cells with multiple cilia due to cytokinesis failure.
Basu B, Lake AVR, China B, Szymanska K, Wheway G, Bell S, Morrison E, Bond J, Johnson CA
Annals of human genetics 2024 Jan;88(1):45-57
Annals of human genetics 2024 Jan;88(1):45-57
Human Kidney Proximal Tubule-Microvascular Model Facilitates High-Throughput Analyses of Structural and Functional Effects of Ischemia-Reperfusion Injury.
Shaughnessey EM, Kann SH, Charest JL, Vedula EM
Advanced biology 2024 Jan;8(1):e2300127
Advanced biology 2024 Jan;8(1):e2300127
Interactome Analysis Reveals a Link of the Novel ALMS1-CEP70 Complex to Centrosomal Clusters.
Woerz F, Hoffmann F, Antony S, Bolz S, Jarboui MA, Junger K, Klose F, Stehle IF, Boldt K, Ueffing M, Beyer T
Molecular & cellular proteomics : MCP 2024 Jan;23(1):100701
Molecular & cellular proteomics : MCP 2024 Jan;23(1):100701
Phosphodiesterase 4 activity uniquely regulates ciliary cAMP-dependent 3T3-L1 adipogenesis.
Erdelsky MR, Groves SA, Shah C, Delios SB, Umana MB, Maurice DH
Cellular signalling 2024 Jan;113:110981
Cellular signalling 2024 Jan;113:110981
Upregulation of miR-20b-5p inhibits trophoblast invasion by blocking autophagy in recurrent miscarriage.
Lin RC, Chao YY, Su MT, Tsai HL, Tsai PY, Wang CY
Cellular signalling 2024 Jan;113:110934
Cellular signalling 2024 Jan;113:110934
Variants in the WDR44 WD40-repeat domain cause a spectrum of ciliopathy by impairing ciliogenesis initiation.
Accogli A, Shakya S, Yang T, Insinna C, Kim SY, Bell D, Butov KR, Severino M, Niceta M, Scala M, Lee HS, Yoo T, Stauffer J, Zhao H, Fiorillo C, Pedemonte M, Diana MC, Baldassari S, Zakharova V, Shcherbina A, Rodina Y, Fagerberg C, Roos LS, Wierzba J, Dobosz A, Gerard A, Potocki L, Rosenfeld JA, Lalani SR, Scott TM, Scott D, Azamian MS, Louie R, Moore HW, Champaigne NL, Hollingsworth G, Torella A, Nigro V, Ploski R, Salpietro V, Zara F, Pizzi S, Chillemi G, Ognibene M, Cooney E, Do J, Linnemann A, Larsen MJ, Specht S, Walters KJ, Choi HJ, Choi M, Tartaglia M, Youkharibache P, Chae JH, Capra V, Park SG, Westlake CJ
Nature communications 2024 Jan 8;15(1):365
Nature communications 2024 Jan 8;15(1):365
TMEM216 promotes primary ciliogenesis and Hedgehog signaling through the SUFU-GLI2/GLI3 axis.
Wang Y, Yao H, Zhang Y, Mu N, Lu T, Du Z, Wu Y, Li X, Su M, Shao M, Sun X, Su L, Liu X
Science signaling 2024 Jan 23;17(820):eabo0465
Science signaling 2024 Jan 23;17(820):eabo0465
Cytoneme signaling provides essential contributions to mammalian tissue patterning.
Hall ET, Dillard ME, Cleverdon ER, Zhang Y, Daly CA, Ansari SS, Wakefield R, Stewart DP, Pruett-Miller SM, Lavado A, Carisey AF, Johnson A, Wang YD, Selner E, Tanes M, Ryu YS, Robinson CG, Steinberg J, Ogden SK
Cell 2024 Jan 18;187(2):276-293.e23
Cell 2024 Jan 18;187(2):276-293.e23
Hedgehog signalling is involved in acquired resistance to KRAS(G12C) inhibitors in lung cancer cells.
Lee C, Yi J, Park J, Ahn B, Won YW, Jeon J, Lee BJ, Cho WJ, Park JW
Cell death & disease 2024 Jan 16;15(1):56
Cell death & disease 2024 Jan 16;15(1):56
The novel linkage between Fuz and Gpr161 genes regulates sonic hedgehog signaling during mouse embryonic development.
Kim SE, Kim HY, Wlodarczyk BJ, Finnell RH
bioRxiv : the preprint server for biology 2024 Jan 12;
bioRxiv : the preprint server for biology 2024 Jan 12;
The Fuzzy planar cell polarity protein (FUZ), necessary for primary cilium formation, is essential for pituitary development.
Lodge EJ, Barrell WB, Liu KJ, Andoniadou CL
Journal of anatomy 2024 Feb;244(2):358-367
Journal of anatomy 2024 Feb;244(2):358-367
Primary cilium participates in radiation-induced bystander effects through TGF-β1 signaling.
Qu P, Shao Z, Zhang Y, He J, Lu D, Wei W, Hua J, Wang W, Wang J, Ding N
Journal of cellular physiology 2024 Feb;239(2):e31163
Journal of cellular physiology 2024 Feb;239(2):e31163
The miR-669a-5p/G3BP/HDAC6/AKAP12 Axis Regulates Primary Cilia Length.
Wang W, Dai X, Li Y, Li M, Chi Z, Hu X, Wang Z
Advanced science (Weinheim, Baden-Wurttemberg, Germany) 2024 Feb;11(6):e2305068
Advanced science (Weinheim, Baden-Wurttemberg, Germany) 2024 Feb;11(6):e2305068
DPCD is a regulator of R2TP in ciliogenesis initiation through Akt signaling.
Mao YQ, Seraphim TV, Wan Y, Wu R, Coyaud E, Bin Munim M, Mollica A, Laurent E, Babu M, Mennella V, Raught B, Houry WA
Cell reports 2024 Feb 27;43(2):113713
Cell reports 2024 Feb 27;43(2):113713
Primary cilia promote the differentiation of human neurons through the WNT signaling pathway.
Coschiera A, Yoshihara M, Lauter G, Ezer S, Pucci M, Li H, Kavšek A, Riedel CG, Kere J, Swoboda P
BMC biology 2024 Feb 27;22(1):48
BMC biology 2024 Feb 27;22(1):48
Mouse SAS-6 is required for centriole formation in embryos and integrity in embryonic stem cells.
Grzonka M, Bazzi H
eLife 2024 Feb 26;13
eLife 2024 Feb 26;13
The Primary Cilium and its Hedgehog Signaling in Nociceptors Contribute to Inflammatory and Neuropathic Pain.
Fitzsimons LA, Staurengo-Ferrari L, Bogen O, Araldi D, Bonet IJM, Jordan EE, Levine JD, Tucker KL
Research square 2024 Feb 26;
Research square 2024 Feb 26;
Understanding the effects of per- and polyfluoroalkyl substances on early skin development: Role of ciliogenesis inhibition and altered microtubule dynamics.
Zhao M, Yin N, Yang R, Li S, Zhang S, Faiola F
The Science of the total environment 2024 Feb 25;913:169702
The Science of the total environment 2024 Feb 25;913:169702
Collagen VI Deficiency Impairs Tendon Fibroblasts Mechanoresponse in Ullrich Congenital Muscular Dystrophy.
Cenni V, Sabatelli P, Di Martino A, Merlini L, Antoniel M, Squarzoni S, Neri S, Santi S, Metti S, Bonaldo P, Faldini C
Cells 2024 Feb 22;13(5)
Cells 2024 Feb 22;13(5)
The exocyst complex and intracellular vesicles mediate soluble protein trafficking to the primary cilium.
Niedziółka SM, Datta S, Uśpieński T, Baran B, Skarżyńska W, Humke EW, Rohatgi R, Niewiadomski P
Communications biology 2024 Feb 21;7(1):213
Communications biology 2024 Feb 21;7(1):213
Early insights into the role of Exoc6B associated with spondyloepimetaphyseal dysplasia with joint laxity type 3 in primary ciliogenesis and chondrogenic differentiation in vitro.
Guleria VS, Quadri N, Prasad K, Das R, Upadhyai P
Molecular biology reports 2024 Feb 2;51(1):274
Molecular biology reports 2024 Feb 2;51(1):274
Ultrastructure expansion microscopy (U-ExM) of mouse and human kidneys for analysis of subcellular structures.
Langner E, Puapatanakul P, Pudlowski R, Alsabbagh DY, Miner JH, Horani A, Dutcher SK, Brody SL, Wang JT, Suleiman HY, Mahjoub MR
bioRxiv : the preprint server for biology 2024 Feb 17;
bioRxiv : the preprint server for biology 2024 Feb 17;
Immuno-Scanning Electron Microscopy of Islet Primary Cilia.
Sviben S, Polino AJ, Melena I, Hughes JW
bioRxiv : the preprint server for biology 2024 Feb 16;
bioRxiv : the preprint server for biology 2024 Feb 16;
Motor protein Kif6 regulates cilia motility and polarity in brain ependymal cells.
Takagishi M, Yue Y, Gray RS, Verhey KJ, Wallingford JB
Disease models & mechanisms 2024 Feb 1;17(2)
Disease models & mechanisms 2024 Feb 1;17(2)
Kidney ischemia/reperfusion injury causes cholangiocytes primary cilia disruption and abnormal bile secretion.
Han YK, Lim HJ, Jang G, Jang SY, Park KM
Biochimica et biophysica acta. Molecular basis of disease 2024 Aug;1870(6):167225
Biochimica et biophysica acta. Molecular basis of disease 2024 Aug;1870(6):167225
Absence of the RING domain in MID1 results in patterning defects in the developing human brain.
Frank S, Gabassi E, Käseberg S, Bertin M, Zografidou L, Pfeiffer D, Brennenstuhl H, Falk S, Karow M, Schweiger S
Life science alliance 2024 Apr;7(4)
Life science alliance 2024 Apr;7(4)
INPP5E Regulates the Distribution of Phospholipids on Cilia in RPE1 Cells.
Zhai D, Li L, Chen C, Wang X, Liu R, Shan Y
Journal of clinical laboratory analysis 2024 Apr;38(7):e25031
Journal of clinical laboratory analysis 2024 Apr;38(7):e25031
Differential alternative splicing analysis links variation in ZRSR2 to a novel type of oral-facial-digital syndrome.
Hannes L, Atzori M, Goldenberg A, Argente J, Attie-Bitach T, Amiel J, Attanasio C, Braslavsky DG, Bruel AL, Castanet M, Dubourg C, Jacobs A, Lyonnet S, Martinez-Mayer J, Pérez Millán MI, Pezzella N, Pelgrims E, Aerden M, Bauters M, Rochtus A, Scaglia P, Swillen A, Sifrim A, Tammaro R, Mau-Them FT, Odent S, Thauvin-Robinet C, Franco B, Breckpot J
Genetics in medicine : official journal of the American College of Medical Genetics 2024 Apr;26(4):101059
Genetics in medicine : official journal of the American College of Medical Genetics 2024 Apr;26(4):101059
A disease-associated PPP2R3C-MAP3K1 phospho-regulatory module controls centrosome function.
Ganga AK, Sweeney LK, Ramos AR, Bishop CS, Hamel V, Guichard P, Breslow DK
bioRxiv : the preprint server for biology 2024 Apr 3;
bioRxiv : the preprint server for biology 2024 Apr 3;
Roles for CEP170 in cilia function and dynein-2 assembly.
Weijman JF, Vuolo L, Shak C, Pugnetti A, Mukhopadhyay AG, Hodgson LR, Heesom KJ, Roberts AJ, Stephens DJ
Journal of cell science 2024 Apr 15;137(8)
Journal of cell science 2024 Apr 15;137(8)
The Scaffold Protein KATNIP Enhances CILK1 Control of Primary Cilia.
Turner JS, McCabe EA, Kuang KW, Gailey CD, Brautigan DL, Limerick A, Wang EX, Fu Z
Molecular and cellular biology 2023;43(9):472-480
Molecular and cellular biology 2023;43(9):472-480
Characterization of Primary Cilia Formation in Human ESC-Derived Retinal Organoids.
Ning K, Luo Z, Kowal TJ, Tran M, Majumder R, Jarin TM, Wu AY, Goldberg JL, Sun Y
Stem cells international 2023;2023:6494486
Stem cells international 2023;2023:6494486
In vivo Polycystin-1 interactome using a novel Pkd1 knock-in mouse model.
Lin CC, Menezes LF, Qiu J, Pearson E, Zhou F, Ishimoto Y, Anderson DE, Germino GG
PloS one 2023;18(8):e0289778
PloS one 2023;18(8):e0289778
Human LUHMES and NES cells as models for studying primary cilia in neurons.
Coschiera A, Watts ME, Kere J, Tammimies K, Swoboda P
Methods in cell biology 2023;176:27-41
Methods in cell biology 2023;176:27-41
Identification of novel TMEM231 gene splice variants and pathological findings in a fetus with Meckel Syndrome.
Zhang Q, Yang S, Chen X, Wang H, Li K, Zhang C, Liao S, Qin L, Hou Q
Frontiers in genetics 2023;14:1252873
Frontiers in genetics 2023;14:1252873
Morphological changes and two Nodal paralogs drive left-right asymmetry in the squamate veiled chameleon (C. calyptratus).
Shylo NA, Smith SE, Price AJ, Guo F, McClain M, Trainor PA
Frontiers in cell and developmental biology 2023;11:1132166
Frontiers in cell and developmental biology 2023;11:1132166
A targeted multi-proteomics approach generates a blueprint of the ciliary ubiquitinome.
Aslanyan MG, Doornbos C, Diwan GD, Anvarian Z, Beyer T, Junger K, van Beersum SEC, Russell RB, Ueffing M, Ludwig A, Boldt K, Pedersen LB, Roepman R
Frontiers in cell and developmental biology 2023;11:1113656
Frontiers in cell and developmental biology 2023;11:1113656
Ciliary phenotyping in renal epithelial cells in a cranioectodermal dysplasia patient with WDR35 variants.
Walczak-Sztulpa J, Wawrocka A, Kuszel Ł, Pietras P, Leśniczak-Staszak M, Andrusiewicz M, Krawczyński MR, Latos-Bieleńska A, Pawlak M, Grenda R, Materna-Kiryluk A, Oud MM, Szaflarski W
Frontiers in molecular biosciences 2023;10:1285790
Frontiers in molecular biosciences 2023;10:1285790
Brain microvascular endothelial cells possess a second cilium that arises from the daughter centriole.
Thirugnanam K, Gupta A, Nunez F, Prabhudesai S, Pan AY, Nauli SM, Ramchandran R
Frontiers in molecular biosciences 2023;10:1250016
Frontiers in molecular biosciences 2023;10:1250016
INPP5E regulates CD3ζ enrichment at the immune synapse by phosphoinositide distribution control.
Chiu TY, Lo CH, Lin YH, Lai YD, Lin SS, Fang YT, Huang WS, Huang SY, Tsai PY, Yang FH, Chong WM, Wu YC, Tsai HC, Liu YW, Hsu CL, Liao JC, Wang WJ
Communications biology 2023 Sep 5;6(1):911
Communications biology 2023 Sep 5;6(1):911
Pathogenic RAB34 variants impair primary cilium assembly and cause a novel oral-facial-digital syndrome.
Bruel AL, Ganga AK, Nosková L, Valenzuela I, Martinovic J, Duffourd Y, Zikánová M, Majer F, Kmoch S, Mohler M, Sun J, Sweeney LK, Martínez-Gil N, Thauvin-Robinet C, Breslow DK
Human molecular genetics 2023 Sep 5;32(18):2822-2831
Human molecular genetics 2023 Sep 5;32(18):2822-2831
Increasing Ciliary ARL13B Expression Drives Active and Inhibitor-Resistant Smoothened and GLI into Glioma Primary Cilia.
Shi P, Tian J, Mallinger JC, Ling D, Deleyrolle LP, McIntyre JC, Caspary T, Breunig JJ, Sarkisian MR
Cells 2023 Sep 26;12(19)
Cells 2023 Sep 26;12(19)
The IFT81-IFT74 complex acts as an unconventional RabL2 GTPase-activating protein during intraflagellar transport.
Boegholm N, Petriman NA, Loureiro-López M, Wang J, Vela MIS, Liu B, Kanie T, Ng R, Jackson PK, Andersen JS, Lorentzen E
The EMBO journal 2023 Sep 18;42(18):e111807
The EMBO journal 2023 Sep 18;42(18):e111807
Dnajb11-Kidney Disease Develops from Reduced Polycystin-1 Dosage but not Unfolded Protein Response in Mice.
Ghosh Roy S, Li Z, Guo Z, Long KT, Rehrl S, Tian X, Dong K, Besse W
Journal of the American Society of Nephrology : JASN 2023 Sep 1;34(9):1521-1534
Journal of the American Society of Nephrology : JASN 2023 Sep 1;34(9):1521-1534
ODF2 Negatively Regulates CP110 Levels at the Centrioles/Basal Bodies to Control the Biogenesis of Primary Cilia.
Otto M, Hoyer-Fender S
Cells 2023 Sep 1;12(17)
Cells 2023 Sep 1;12(17)
Primary cilia control oligodendrocyte precursor cell proliferation in white matter injury via Hedgehog-independent CREB signaling.
Hoi KK, Xia W, Wei MM, Ulloa Navas MJ, Garcia Verdugo JM, Nachury MV, Reiter JF, Fancy SPJ
Cell reports 2023 Oct 31;42(10):113272
Cell reports 2023 Oct 31;42(10):113272
PATJ inhibits histone deacetylase 7 to control tight junction formation and cell polarity.
Fiedler J, Moennig T, Hinrichs JH, Weber A, Wagner T, Hemmer T, Schröter R, Weide T, Epting D, Bergmann C, Nedvetsky P, Krahn MP
Cellular and molecular life sciences : CMLS 2023 Oct 25;80(11):333
Cellular and molecular life sciences : CMLS 2023 Oct 25;80(11):333
Parkinsonism Sac domain mutation in Synaptojanin-1 affects ciliary properties in iPSC-derived dopaminergic neurons.
Rafiq NM, Fujise K, Rosenfeld MS, Xu P, Wu Y, De Camilli P
bioRxiv : the preprint server for biology 2023 Oct 13;
bioRxiv : the preprint server for biology 2023 Oct 13;
Primary cilium-mediated signaling cascade suppresses age-related biliary fibrosis.
Hong R, Tian X, Ma H, Ni H, Yang J, Bu W, Li T, Yang S, Li D, Liu M, Tan Y
Journal of cellular physiology 2023 Nov;238(11):2600-2611
Journal of cellular physiology 2023 Nov;238(11):2600-2611
Certain heterozygous variants in the kinase domain of the serine/threonine kinase NEK8 can cause an autosomal dominant form of polycystic kidney disease.
Claus LR, Chen C, Stallworth J, Turner JL, Slaats GG, Hawks AL, Mabillard H, Senum SR, Srikanth S, Flanagan-Steet H, Louie RJ, Silver J, Lerner-Ellis J, Morel C, Mighton C, Sleutels F, van Slegtenhorst M, van Ham T, Brooks AS, Dorresteijn EM, Barakat TS, Dahan K, Demoulin N, Goffin EJ, Olinger E, Genomics England Research Consortium, Larsen M, Hertz JM, Lilien MR, Obeidová L, Seeman T, Stone HK, Kerecuk L, Gurgu M, Yousef Yengej FA, Ammerlaan CME, Rookmaaker MB, Hanna C, Rogers RC, Duran K, Peters E, Sayer JA, van Haaften G, Harris PC, Ling K, Mason JM, van Eerde AM, Steet R
Kidney international 2023 Nov;104(5):995-1007
Kidney international 2023 Nov;104(5):995-1007
Proteomic profiling of centrosomes across multiple mammalian cell and tissue types by an affinity capture method.
Carden S, Vitiello E, Rosa E Silva I, Holder J, Quarantotti V, Kishore K, Roamio Franklin VN, D'Santos C, Ochi T, van Breugel M, Gergely F
Developmental cell 2023 Nov 6;58(21):2393-2410.e9
Developmental cell 2023 Nov 6;58(21):2393-2410.e9
Oxidative Stress Induces Skin Pigmentation in Melasma by Inhibiting Hedgehog Signaling.
Kim NH, Lee AY
Antioxidants (Basel, Switzerland) 2023 Nov 6;12(11)
Antioxidants (Basel, Switzerland) 2023 Nov 6;12(11)
Absence of the primary cilia formation gene Talpid3 impairs muscle stem cell function.
Martinez-Heredia V, Blackwell D, Sebastian S, Pearson T, Mok GF, Mincarelli L, Utting C, Folkes L, Poeschl E, Macaulay I, Mayer U, Münsterberg A
Communications biology 2023 Nov 4;6(1):1121
Communications biology 2023 Nov 4;6(1):1121
MiniBAR/GARRE1 is a dual Rac and Rab effector required for ciliogenesis.
Serres MP, Shaughnessy R, Escot S, Hammich H, Cuvelier F, Salles A, Rocancourt M, Verdon Q, Gaffuri AL, Sourigues Y, Malherbe G, Velikovsky L, Chardon F, Sassoon N, Tinevez JY, Callebaut I, Formstecher E, Houdusse A, David NB, Pylypenko O, Echard A
Developmental cell 2023 Nov 20;58(22):2477-2494.e8
Developmental cell 2023 Nov 20;58(22):2477-2494.e8
Thalamic Neuron Resilience during Osmotic Demyelination Syndrome (ODS) Is Revealed by Primary Cilium Outgrowth and ADP-ribosylation factor-like protein 13B Labeling in Axon Initial Segment.
Gilloteaux J, De Swert K, Suain V, Nicaise C
International journal of molecular sciences 2023 Nov 17;24(22)
International journal of molecular sciences 2023 Nov 17;24(22)
Proximity Labeling Expansion Microscopy (PL-ExM) resolves structure of the interactome.
Park S, Wang X, Li X, Huang X, Fong KC, Yu C, Tran AA, Scipioni L, Dai Z, Huang L, Shi X
bioRxiv : the preprint server for biology 2023 Nov 13;
bioRxiv : the preprint server for biology 2023 Nov 13;
Polycystin-2-dependent transcriptome reveals early response of autosomal dominant polycystic kidney disease.
Jung HJ, Dixon EE, Coleman R, Watnick T, Reiter JF, Outeda P, Cebotaru V, Woodward OM, Welling PA
Physiological genomics 2023 Nov 1;55(11):565-577
Physiological genomics 2023 Nov 1;55(11):565-577
LRP2 contributes to planar cell polarity-dependent coordination of motile cilia function.
Bunatyan L, Margineanu A, Boutin C, Montcouquiol M, Bachmann S, Ilsø Christensen E, Willnow TE, Christ A
Cell and tissue research 2023 May;392(2):535-551
Cell and tissue research 2023 May;392(2):535-551
PDLIM3 supports hedgehog signaling in medulloblastoma by facilitating cilia formation.
Zhang J, Yang Y, Li X, Li G, Mizukami T, Liu Y, Wang Y, Xu G, Roder H, Zhang L, Yang ZJ
Cell death and differentiation 2023 May;30(5):1198-1210
Cell death and differentiation 2023 May;30(5):1198-1210
Rab8 and TNPO1 are ciliary transport adaptors for GTPase Arl13b by interacting with its RVEP motif containing ciliary targeting sequence.
Mahajan D, Madugula V, Lu L
The Journal of biological chemistry 2023 May;299(5):104604
The Journal of biological chemistry 2023 May;299(5):104604
Scanning electron microscopy of human islet cilia.
Polino AJ, Sviben S, Melena I, Piston DW, Hughes JW
Proceedings of the National Academy of Sciences of the United States of America 2023 May 30;120(22):e2302624120
Proceedings of the National Academy of Sciences of the United States of America 2023 May 30;120(22):e2302624120
Extracellular vesicle-mediated delivery of anti-miR-106b inhibits morphine-induced primary ciliogenesis in the brain.
Ma R, Kutchy NA, Wang Z, Hu G
Molecular therapy : the journal of the American Society of Gene Therapy 2023 May 3;31(5):1332-1345
Molecular therapy : the journal of the American Society of Gene Therapy 2023 May 3;31(5):1332-1345
Investigations on Primary Cilia of Nthy-ori 3-1 Cells upon Cysteine Cathepsin Inhibition or Thyrotropin Stimulation.
Doğru AG, Rehders M, Brix K
International journal of molecular sciences 2023 May 26;24(11)
International journal of molecular sciences 2023 May 26;24(11)
CCP5 and CCP6 retain CP110 and negatively regulate ciliogenesis.
Wang Y, Zhang Y, Guo X, Zheng Y, Zhang X, Feng S, Wu HY
BMC biology 2023 May 24;21(1):124
BMC biology 2023 May 24;21(1):124
Ciliary ARL13B inhibits developmental kidney cystogenesis in mouse.
Van Sciver RE, Long AB, Katz HG, Gigante ED, Caspary T
bioRxiv : the preprint server for biology 2023 May 15;
bioRxiv : the preprint server for biology 2023 May 15;
Enhanced primary ciliogenesis via mitochondrial oxidative stress activates AKT to prevent neurotoxicity in HSPA9/mortalin-depleted SH-SY5Y cells.
Bae JE, Jang S, Kim JB, Hyung H, Park NY, Kim YH, Kim SH, Kim SH, Ha JM, Oh GS, Park K, Jeong K, Jang JS, Jo DS, Kim P, Lee HS, Ryoo ZY, Cho DH
Molecular brain 2023 May 11;16(1):41
Molecular brain 2023 May 11;16(1):41
Primary cilia sense glutamine availability and respond via asparagine synthetase.
Steidl ME, Nigro EA, Nielsen AK, Pagliarini R, Cassina L, Lampis M, Podrini C, Chiaravalli M, Mannella V, Distefano G, Yang M, Aslanyan M, Musco G, Roepman R, Frezza C, Boletta A
Nature metabolism 2023 Mar;5(3):385-397
Nature metabolism 2023 Mar;5(3):385-397
The Rab GTPase-binding protein EHBP1L1 and its interactors CD2AP/CIN85 negatively regulate the length of primary cilia via actin remodeling.
Iwano T, Sobajima T, Takeda S, Harada A, Yoshimura SI
The Journal of biological chemistry 2023 Mar;299(3):102985
The Journal of biological chemistry 2023 Mar;299(3):102985
Primary Cilium Identifies a Quiescent Cell Population in the Human Intestinal Crypt.
Sénicourt B, Cloutier G, Basora N, Fallah S, Laniel A, Lavoie C, Beaulieu JF
Cells 2023 Mar 31;12(7)
Cells 2023 Mar 31;12(7)
Reserpine maintains photoreceptor survival in retinal ciliopathy by resolving proteostasis imbalance and ciliogenesis defects.
Chen HY, Swaroop M, Papal S, Mondal AK, Song HB, Campello L, Tawa GJ, Regent F, Shimada H, Nagashima K, de Val N, Jacobson SG, Zheng W, Swaroop A
eLife 2023 Mar 28;12
eLife 2023 Mar 28;12
An actin filament branching surveillance system regulates cell cycle progression, cytokinesis and primary ciliogenesis.
Cao M, Zou X, Li C, Lin Z, Wang N, Zou Z, Ye Y, Seemann J, Levine B, Tang Z, Zhong Q
Nature communications 2023 Mar 27;14(1):1687
Nature communications 2023 Mar 27;14(1):1687
The developmental basis of fingerprint pattern formation and variation.
Glover JD, Sudderick ZR, Shih BB, Batho-Samblas C, Charlton L, Krause AL, Anderson C, Riddell J, Balic A, Li J, Klika V, Woolley TE, Gaffney EA, Corsinotti A, Anderson RA, Johnston LJ, Brown SJ, Wang S, Chen Y, Crichton ML, Headon DJ
Cell 2023 Mar 2;186(5):940-956.e20
Cell 2023 Mar 2;186(5):940-956.e20
Primary cilia control cellular patterning of Meibomian glands during morphogenesis but not lipid composition.
Portal C, Lin Y, Rastogi V, Peterson C, Yiu SC, Foster JW, Wilkerson A, Butovich IA, Iomini C
Communications biology 2023 Mar 17;6(1):282
Communications biology 2023 Mar 17;6(1):282
Inactivation of Invs/Nphp2 in renal epithelial cells drives infantile nephronophthisis like phenotypes in mouse.
Li Y, Xu W, Makova S, Brueckner M, Sun Z
eLife 2023 Mar 15;12
eLife 2023 Mar 15;12
Interactions between TULP3 tubby domain and ARL13B amphipathic helix promote lipidated protein transport to cilia.
Palicharla VR, Hwang SH, Somatilaka BN, Legué E, Shimada IS, Familiari NE, Tran VM, Woodruff JB, Liem KF Jr, Mukhopadhyay S
Molecular biology of the cell 2023 Mar 1;34(3):ar18
Molecular biology of the cell 2023 Mar 1;34(3):ar18
Canonical Wnt signaling regulates soft palate development by mediating ciliary homeostasis.
Janečková E, Feng J, Guo T, Han X, Ghobadi A, Araujo-Villalba A, Rahman MS, Ziaei H, Ho TV, Pareek S, Alvarez J, Chai Y
Development (Cambridge, England) 2023 Mar 1;150(5)
Development (Cambridge, England) 2023 Mar 1;150(5)
Disease-associated mutations in WDR34 lead to diverse impacts on the assembly and function of dynein-2.
Shak C, Vuolo L, Uddin B, Katoh Y, Brown T, Mukhopadhyay AG, Heesom K, Roberts AJ, Stevenson N, Nakayama K, Stephens DJ
Journal of cell science 2023 Mar 1;136(5)
Journal of cell science 2023 Mar 1;136(5)
Multiple interactions of the dynein-2 complex with the IFT-B complex are required for effective intraflagellar transport.
Hiyamizu S, Qiu H, Vuolo L, Stevenson NL, Shak C, Heesom KJ, Hamada Y, Tsurumi Y, Chiba S, Katoh Y, Stephens DJ, Nakayama K
Journal of cell science 2023 Mar 1;136(5)
Journal of cell science 2023 Mar 1;136(5)
Arl13b promotes the proliferation, migration, osteogenesis, and mechanosensation of osteoblasts.
Lin T, Sun Y
Tissue & cell 2023 Jun;82:102088
Tissue & cell 2023 Jun;82:102088
Generation of iPSC-derived human forebrain organoids assembling bilateral eye primordia.
Gabriel E, Albanna W, Pasquini G, Ramani A, Josipovic N, Mariappan A, Riparbelli MG, Callaini G, Karch CM, Goureau O, Papantonis A, Busskamp V, Schneider T, Gopalakrishnan J
Nature protocols 2023 Jun;18(6):1893-1929
Nature protocols 2023 Jun;18(6):1893-1929
Eupatilin Improves Cilia Defects in Human CEP290 Ciliopathy Models.
Corral-Serrano JC, Sladen PE, Ottaviani D, Rezek OF, Athanasiou D, Jovanovic K, van der Spuy J, Mansfield BC, Cheetham ME
Cells 2023 Jun 7;12(12)
Cells 2023 Jun 7;12(12)
Polycomb Ezh1 maintains murine muscle stem cell quiescence through non-canonical regulation of Notch signaling.
Feng X, Wang AH, Juan AH, Ko KD, Jiang K, Riparini G, Ciuffoli V, Kaba A, Lopez C, Naz F, Jarnik M, Aliberti E, Hu S, Segalés J, Khateb M, Acevedo-Luna N, Randazzo D, Cheung TH, Muñoz-Cánoves P, Dell'Orso S, Sartorelli V
Developmental cell 2023 Jun 19;58(12):1052-1070.e10
Developmental cell 2023 Jun 19;58(12):1052-1070.e10
Single-cell transcriptomics reveals ependymal subtypes related to cytoskeleton dynamics as the core driver of syringomyelia pathological development.
Lu C, Wu X, Wang X, Xiao Z, Ma L, Dai J, Jian F
iScience 2023 Jun 16;26(6):106850
iScience 2023 Jun 16;26(6):106850
ULK4 and Fused/STK36 interact to mediate assembly of a motile flagellum.
McCoy CJ, Paupelin-Vaucelle H, Gorilak P, Beneke T, Varga V, Gluenz E
Molecular biology of the cell 2023 Jun 1;34(7):ar66
Molecular biology of the cell 2023 Jun 1;34(7):ar66
Functional characterization of C21ORF2 association with the NEK1 kinase mutated in human in diseases.
Gregorczyk M, Pastore G, Muñoz I, Carroll T, Streubel J, Munro M, Lis P, Lange S, Lamoliatte F, Macartney T, Toth R, Brown F, Hastie J, Pereira G, Durocher D, Rouse J
Life science alliance 2023 Jul;6(7)
Life science alliance 2023 Jul;6(7)
Impairment of Connexin 43 may initiate cilia decline in syringomyelia.
Wang X, Jiang C, Lu C, Ma L, Feng Y, Cui S, Li Q, Li K, Wang X, Jian F
Experimental neurology 2023 Jul;365:114430
Experimental neurology 2023 Jul;365:114430
MRCKα/β positively regulates Gli protein activity.
Baran B, Kosieradzka K, Skarzynska W, Niewiadomski P
Cellular signalling 2023 Jul;107:110666
Cellular signalling 2023 Jul;107:110666
Coordination of canonical and noncanonical Hedgehog signalling pathways mediated by WDR11 during primordial germ cell development.
Lee J, Kim Y, Ataliotis P, Kim HG, Kim DW, Bennett DC, Brown NA, Layman LC, Kim SH
Scientific reports 2023 Jul 29;13(1):12309
Scientific reports 2023 Jul 29;13(1):12309
CEP164-GLI2 association ensures the hedgehog signaling in pancreatic cancer cells.
Fushimi T, Kobayashi T, Itoh H
Biochemical and biophysical research communications 2023 Jul 23;666:179-185
Biochemical and biophysical research communications 2023 Jul 23;666:179-185
Dynein-2-driven intraciliary retrograde trafficking indirectly requires multiple interactions of IFT54 in the IFT-B complex with the dynein-2 complex.
Hiyamizu S, Qiu H, Tsurumi Y, Hamada Y, Katoh Y, Nakayama K
Biology open 2023 Jul 15;12(7)
Biology open 2023 Jul 15;12(7)
Atp7b-dependent choroid plexus dysfunction causes transient copper deficit and metabolic changes in the developing mouse brain.
Washington-Hughes CL, Roy S, Seneviratne HK, Karuppagounder SS, Morel Y, Jones JW, Zak A, Xiao T, Boronina TN, Cole RN, Bumpus NN, Chang CJ, Dawson TM, Lutsenko S
PLoS genetics 2023 Jan;19(1):e1010558
PLoS genetics 2023 Jan;19(1):e1010558
PDGF-AA activates AKT and ERK signaling for testicular interstitial Leydig cell growth via primary cilia.
Tsai YC, Kuo TN, Chao YY, Lee PR, Lin RC, Xiao XY, Huang BM, Wang CY
Journal of cellular biochemistry 2023 Jan;124(1):89-102
Journal of cellular biochemistry 2023 Jan;124(1):89-102
CROCCP2 acts as a human-specific modifier of cilia dynamics and mTOR signaling to promote expansion of cortical progenitors.
Van Heurck R, Bonnefont J, Wojno M, Suzuki IK, Velez-Bravo FD, Erkol E, Nguyen DT, Herpoel A, Bilheu A, Beckers S, Ledent C, Vanderhaeghen P
Neuron 2023 Jan 4;111(1):65-80.e6
Neuron 2023 Jan 4;111(1):65-80.e6
Primary cilia are WNT-transducing organelles whose biogenesis is controlled by a WNT-PP1 axis.
Zhang K, Da Silva F, Seidl C, Wilsch-Bräuninger M, Herbst J, Huttner WB, Niehrs C
Developmental cell 2023 Jan 23;58(2):139-154.e8
Developmental cell 2023 Jan 23;58(2):139-154.e8
Superenhancer activation of KLHDC8A drives glioma ciliation and hedgehog signaling.
Lee D, Gimple RC, Wu X, Prager BC, Qiu Z, Wu Q, Daggubati V, Mariappan A, Gopalakrishnan J, Sarkisian MR, Raleigh DR, Rich JN
The Journal of clinical investigation 2023 Jan 17;133(2)
The Journal of clinical investigation 2023 Jan 17;133(2)
Diverse but unique astrocytic phenotypes during embryonic stem cell differentiation, culturing and development.
Freitag K, Eede P, Ivanov A, Sterczyk N, Schneeberger S, Borodina T, Sauer S, Beule D, Heppner FL
Communications biology 2023 Jan 13;6(1):40
Communications biology 2023 Jan 13;6(1):40
A hierarchical pathway for assembly of the distal appendages that organize primary cilia.
Kanie T, Love JF, Fisher SD, Gustavsson AK, Jackson PK
bioRxiv : the preprint server for biology 2023 Jan 10;
bioRxiv : the preprint server for biology 2023 Jan 10;
Myristoylated Neuronal Calcium Sensor-1 captures the ciliary vesicle at distal appendages.
Kanie T, Ng R, Abbott KL, Pongs O, Jackson PK
bioRxiv : the preprint server for biology 2023 Jan 10;
bioRxiv : the preprint server for biology 2023 Jan 10;
TTBK2 controls cilium stability by regulating distinct modules of centrosomal proteins.
Nguyen A, Goetz SC
Molecular biology of the cell 2023 Jan 1;34(1):ar8
Molecular biology of the cell 2023 Jan 1;34(1):ar8
XBP1 Activation Reduces Severity of Polycystic Kidney Disease due to a Nontruncating Polycystin-1 Mutation in Mice.
Krappitz M, Bhardwaj R, Dong K, Staudner T, Yilmaz DE, Pioppini C, Westergerling P, Ruemmele D, Hollmann T, Nguyen TA, Cai Y, Gallagher AR, Somlo S, Fedeles S
Journal of the American Society of Nephrology : JASN 2023 Jan 1;34(1):110-121
Journal of the American Society of Nephrology : JASN 2023 Jan 1;34(1):110-121
A SEC61A1 variant is associated with autosomal dominant polycystic liver disease.
Schlevogt B, Schlieper V, Krader J, Schröter R, Wagner T, Weiand M, Zibert A, Schmidt HH, Bergmann C, Nedvetsky PI, Krahn MP
Liver international : official journal of the International Association for the Study of the Liver 2023 Feb;43(2):401-412
Liver international : official journal of the International Association for the Study of the Liver 2023 Feb;43(2):401-412
Reduced SPAG17 Expression in Systemic Sclerosis Triggers Myofibroblast Transition and Drives Fibrosis.
Sapao P, Roberson EDO, Shi B, Assassi S, Skaug B, Lee F, Naba A, Perez White BE, Córdova-Fletes C, Tsou PS, Sawalha AH, Gudjonsson JE, Ma F, Verma P, Bhattacharyya D, Carns M, Strauss JF 3rd, Sicard D, Tschumperlin DJ, Champer MI, Campagnola PJ, Teves ME, Varga J
The Journal of investigative dermatology 2023 Feb;143(2):284-293
The Journal of investigative dermatology 2023 Feb;143(2):284-293
Evaluation of Epithelial-Mesenchymal Transition Markers in Autoimmune Thyroid Diseases.
Sacristán-Gómez P, Serrano-Somavilla A, Castro-Espadas L, Sánchez de la Blanca Carrero N, Sampedro-Núñez M, Muñoz-De-Nova JL, Molina-Jiménez F, Rosell A, Marazuela M, Martínez-Hernández R
International journal of molecular sciences 2023 Feb 8;24(4)
International journal of molecular sciences 2023 Feb 8;24(4)
IHH, SHH, and primary cilia mediate epithelial-stromal cross-talk during decidualization in mice.
Li B, Yan YP, He YY, Liang C, Li MY, Wang Y, Yang ZM
Science signaling 2023 Feb 28;16(774):eadd0645
Science signaling 2023 Feb 28;16(774):eadd0645
Deficiency of the minor spliceosome component U4atac snRNA secondarily results in ciliary defects in human and zebrafish.
Khatri D, Putoux A, Cologne A, Kaltenbach S, Besson A, Bertiaux E, Guguin J, Fendler A, Dupont MA, Benoit-Pilven C, Qebibo L, Ahmed-Elie S, Audebert-Bellanger S, Blanc P, Rambaud T, Castelle M, Cornen G, Grotto S, Guët A, Guibaud L, Michot C, Odent S, Ruaud L, Sacaze E, Hamel V, Bordonné R, Leutenegger AL, Edery P, Burglen L, Attié-Bitach T, Mazoyer S, Delous M
Proceedings of the National Academy of Sciences of the United States of America 2023 Feb 28;120(9):e2102569120
Proceedings of the National Academy of Sciences of the United States of America 2023 Feb 28;120(9):e2102569120
Kif6 regulates cilia motility and polarity in brain ependymal cells.
Takagishi M, Yue Y, Gray RS, Verhey KJ, Wallingford JB
bioRxiv : the preprint server for biology 2023 Feb 16;
bioRxiv : the preprint server for biology 2023 Feb 16;
Centriolar satellites expedite mother centriole remodeling to promote ciliogenesis.
Hall EA, Kumar D, Prosser SL, Yeyati PL, Herranz-Pérez V, García-Verdugo JM, Rose L, McKie L, Dodd DO, Tennant PA, Megaw R, Murphy LC, Ferreira MF, Grimes G, Williams L, Quidwai T, Pelletier L, Reiter JF, Mill P
eLife 2023 Feb 15;12
eLife 2023 Feb 15;12
Scanning electron microscopy of human islet cilia.
Polino AJ, Sviben S, Melena I, Piston DW, Hughes J
bioRxiv : the preprint server for biology 2023 Feb 15;
bioRxiv : the preprint server for biology 2023 Feb 15;
GRAMD1/ASTER-mediated cholesterol transport promotes Smoothened cholesterylation at the endoplasmic reticulum.
Qiu ZP, Lin ZC, Hu A, Liu YB, Zeng WE, Zhao X, Shi XJ, Luo J, Song BL
The EMBO journal 2023 Feb 1;42(3):e111513
The EMBO journal 2023 Feb 1;42(3):e111513
TTBK2 mutations associated with spinocerebellar ataxia type 11 disrupt peroxisome dynamics and ciliary localization of SHH signaling proteins.
Muñoz-Estrada J, Nguyen AV, Goetz SC
bioRxiv : the preprint server for biology 2023 Feb 1;
bioRxiv : the preprint server for biology 2023 Feb 1;
Deletion of IFT20 exclusively in the RPE ablates primary cilia and leads to retinal degeneration.
Kretschmer V, Schneider S, Matthiessen PA, Reichert D, Hotaling N, Glasßer G, Lieberwirth I, Bharti K, De Cegli R, Conte I, Nandrot EF, May-Simera HL
PLoS biology 2023 Dec;21(12):e3002402
PLoS biology 2023 Dec;21(12):e3002402
CXCL12 targets the primary cilium cAMP/cGMP ratio to regulate cell polarity during migration.
Atkins M, Wurmser M, Darmon M, Roche F, Nicol X, Métin C
Nature communications 2023 Dec 4;14(1):8003
Nature communications 2023 Dec 4;14(1):8003
Beta cell primary cilia mediate somatostatin responsiveness via SSTR3.
Adamson SE, Li ZA, Hughes JW
Islets 2023 Dec 31;15(1):2252855
Islets 2023 Dec 31;15(1):2252855
The Primary Cilium and its Hedgehog Signaling in Nociceptors Contribute to Inflammatory and Neuropathic Pain.
Fitzsimons LA, Staurengo-Ferrari L, Bogen O, Araldi D, Bonet IJM, Jordan EE, Levine JD, Tucker KL
bioRxiv : the preprint server for biology 2023 Dec 27;
bioRxiv : the preprint server for biology 2023 Dec 27;
Human iPSC-derived renal collecting duct organoid model cystogenesis in ADPKD.
Mae SI, Hattanda F, Morita H, Nozaki A, Katagiri N, Ogawa H, Teranaka K, Nishimura Y, Kudoh A, Yamanaka S, Matsuse K, Ryosaka M, Watanabe A, Soga T, Nishio S, Osafune K
Cell reports 2023 Dec 26;42(12):113431
Cell reports 2023 Dec 26;42(12):113431
HDAC6 inhibition regulates substrate stiffness-mediated inflammation signaling in chondrocytes.
Zhang Y, Tawiah GK, Zhang Y, Wang X, Wei X, Chen W, Qiao X, Zhang Q
Acta biochimica et biophysica Sinica 2023 Dec 25;55(12):1987-1998
Acta biochimica et biophysica Sinica 2023 Dec 25;55(12):1987-1998
Identification of new ciliary signaling pathways in the brain and insights into neurological disorders.
Loukil A, Ebright E, Uezu A, Gao Y, Soderling SH, Goetz SC
bioRxiv : the preprint server for biology 2023 Dec 21;
bioRxiv : the preprint server for biology 2023 Dec 21;
WDR31 displays functional redundancy with GTPase-activating proteins (GAPs) ELMOD and RP2 in regulating IFT complex and recruiting the BBSome to cilium.
Cevik S, Peng X, Beyer T, Pir MS, Yenisert F, Woerz F, Hoffmann F, Altunkaynak B, Pir B, Boldt K, Karaman A, Cakiroglu M, Oner SS, Cao Y, Ueffing M, Kaplan OI
Life science alliance 2023 Aug;6(8)
Life science alliance 2023 Aug;6(8)
Ciliary ARL13B inhibits developmental kidney cystogenesis in mouse.
Van Sciver RE, Long AB, Katz HG, Gigante ED, Caspary T
Developmental biology 2023 Aug;500:1-9
Developmental biology 2023 Aug;500:1-9
A Novel Genetic Variant in MBD5 Associated with Severe Epilepsy and Intellectual Disability: Potential Implications on Neural Primary Cilia.
Martins M, Oliveira AR, Martins S, Vieira JP, Perdigão P, Fernandes AR, de Almeida LP, Palma PJ, Sequeira DB, Santos JMM, Duque F, Oliveira G, Cardoso AL, Peça J, Seabra CM
International journal of molecular sciences 2023 Aug 9;24(16)
International journal of molecular sciences 2023 Aug 9;24(16)
Tau tubulin kinase 1 and 2 regulate ciliogenesis and human pluripotent stem cells-derived neural rosettes.
Binó L, Čajánek L
Scientific reports 2023 Aug 9;13(1):12884
Scientific reports 2023 Aug 9;13(1):12884
DOT1L activity affects neural stem cell division mode and reduces differentiation and ASNS expression.
Appiah B, Fullio CL, Ossola C, Bertani I, Restelli E, Cheffer A, Polenghi M, Haffner C, Garcia-Miralles M, Zeis P, Treppner M, Bovio P, Schlichtholz L, Mas-Sanchez A, Zografidou L, Winter J, Binder H, Grün D, Kalebic N, Taverna E, Vogel T
EMBO reports 2023 Aug 3;24(8):e56233
EMBO reports 2023 Aug 3;24(8):e56233
Hedgehog target genes regulate lipid metabolism to drive basal cell carcinoma and medulloblastoma.
Daggubati V, Vykunta A, Choudhury A, Qadeer Z, Mirchia K, Saulnier O, Zakimi N, Hines K, Paul M, Wang L, Jura N, Xu L, Reiter J, Taylor M, Weiss W, Raleigh D
Research square 2023 Aug 3;
Research square 2023 Aug 3;
PCDH12 loss results in premature neuronal differentiation and impeded migration in a cortical organoid model.
Rakotomamonjy J, Rylaarsdam L, Fares-Taie L, McDermott S, Davies D, Yang G, Fagbemi F, Epstein M, Fairbanks-Santana M, Rozet JM, Guemez-Gamboa A
Cell reports 2023 Aug 29;42(8):112845
Cell reports 2023 Aug 29;42(8):112845
Mutational burden of XPNPEP3 leads to defects in mitochondrial complex I and cilia in NPHPL1.
Tong L, Rao J, Yang C, Xu J, Lu Y, Zhang Y, Cang X, Xie S, Mao J, Jiang P
iScience 2023 Aug 18;26(8):107446
iScience 2023 Aug 18;26(8):107446
Increase in primary cilia number and length upon VDAC1 depletion contributes to attenuated proliferation of cancer cells.
Dutta A, Halder P, Gayen A, Mukherjee A, Mukherjee C, Majumder S
Experimental cell research 2023 Aug 15;429(2):113671
Experimental cell research 2023 Aug 15;429(2):113671
Ubiquitylation of BBSome is required for ciliary assembly and signaling.
Chiuso F, Delle Donne R, Giamundo G, Rinaldi L, Borzacchiello D, Moraca F, Intartaglia D, Iannucci R, Senatore E, Lignitto L, Garbi C, Conflitti P, Catalanotti B, Conte I, Feliciello A
EMBO reports 2023 Apr 5;24(4):e55571
EMBO reports 2023 Apr 5;24(4):e55571
Septin-mediated RhoA activation engages the exocyst complex to recruit the cilium transition zone.
Safavian D, Kim MS, Xie H, El-Zeiry M, Palander O, Dai L, Collins RF, Froese C, Shannon R, Nagata KI, Trimble WS
The Journal of cell biology 2023 Apr 3;222(4)
The Journal of cell biology 2023 Apr 3;222(4)
A stress-induced cilium-to-PML-NB route drives senescence initiation.
Ma X, Zhang Y, Zhang Y, Zhang X, Huang Y, He K, Chen C, Hao J, Zhao D, LeBrasseur NK, Kirkland JL, Chini EN, Wei Q, Ling K, Hu J
Nature communications 2023 Apr 3;14(1):1840
Nature communications 2023 Apr 3;14(1):1840
Identification of a heterogeneous and dynamic ciliome during embryonic development and cell differentiation.
Elliott KH, Balchand SK, Bonatto Paese CL, Chang CF, Yang Y, Brown KM, Rasicci DT, He H, Thorner K, Chaturvedi P, Murray SA, Chen J, Porollo A, Peterson KA, Brugmann SA
Development (Cambridge, England) 2023 Apr 15;150(8)
Development (Cambridge, England) 2023 Apr 15;150(8)
Eupatilin improves cilia defects in human CEP290 ciliopathy models.
Corral-Serrano JC, Sladen PE, Ottaviani D, Rezek FO, Jovanovic K, Athanasiou D, van der Spuy J, Mansfield BC, Cheetham ME
bioRxiv : the preprint server for biology 2023 Apr 12;
bioRxiv : the preprint server for biology 2023 Apr 12;
Pyrogallol-Phloroglucinol-6, 6-Bieckol Restored Primary Cilia Length, Which Was Decreased by High-Fat Diet in Visceral Adipose Tissue, and Decreased Adipogenesis.
Oh S, Son M, Jang JT, Park CH, Son KH, Byun K
International journal of endocrinology 2022;2022:8486965
International journal of endocrinology 2022;2022:8486965
Systematic analysis of cilia characteristics and Hedgehog signaling in five immortal cell lines.
Gómez AE, Christman AK, Van De Weghe JC, Finn M, Doherty D
PloS one 2022;17(12):e0266433
PloS one 2022;17(12):e0266433
Physical Exercise-Induced Astrocytic Neuroprotection and Cognitive Improvement Through Primary Cilia and Mitogen-Activated Protein Kinases Pathway in Rats With Chronic Cerebral Hypoperfusion.
Cao W, Lin J, Xiang W, Liu J, Wang B, Liao W, Jiang T
Frontiers in aging neuroscience 2022;14:866336
Frontiers in aging neuroscience 2022;14:866336
Renal primary cilia lengthen in the progression of diabetic kidney disease.
Bai Y, Li P, Liu J, Zhang L, Cui S, Wei C, Fu B, Sun X, Cai G, Hong Q, Chen X
Frontiers in endocrinology 2022;13:984452
Frontiers in endocrinology 2022;13:984452
Microtubule modification defects underlie cilium degeneration in cell models of retinitis pigmentosa associated with pre-mRNA splicing factor mutations.
Nazlamova L, Villa Vasquez SS, Lord J, Karthik V, Cheung MK, Lakowski J, Wheway G
Frontiers in genetics 2022;13:1009430
Frontiers in genetics 2022;13:1009430
Fluorescence imaging of beta cell primary cilia.
Li ZA, Cho JH, Woodhams LG, Hughes JW
Frontiers in endocrinology 2022;13:1004136
Frontiers in endocrinology 2022;13:1004136
Hedgehog signaling is controlled by Rac1 activity.
Tang C, Wu X, Ren Q, Yao M, Xu S, Yan Z
Theranostics 2022;12(3):1303-1320
Theranostics 2022;12(3):1303-1320
Concurrent Germline and Somatic Mutations in FLCN and Preliminary Exploration of Its Function: A Case Report.
Wang T, Yang Y, Feng H, Cui B, Lv Z, Zhao W, Zhang X, Ma X
Frontiers in oncology 2022;12:877470
Frontiers in oncology 2022;12:877470
Human Organotypic Airway and Lung Organoid Cells of Bronchiolar and Alveolar Differentiation Are Permissive to Infection by Influenza and SARS-CoV-2 Respiratory Virus.
Ekanger CT, Zhou F, Bohan D, Lotsberg ML, Ramnefjell M, Hoareau L, Røsland GV, Lu N, Aanerud M, Gärtner F, Salminen PR, Bentsen M, Halvorsen T, Ræder H, Akslen LA, Langeland N, Cox R, Maury W, Stuhr LEB, Lorens JB, Engelsen AST
Frontiers in cellular and infection microbiology 2022;12:841447
Frontiers in cellular and infection microbiology 2022;12:841447
Modeling Human Primary Microcephaly With hiPSC-Derived Brain Organoids Carrying CPAP-E1235V Disease-Associated Mutant Protein.
An HL, Kuo HC, Tang TK
Frontiers in cell and developmental biology 2022;10:830432
Frontiers in cell and developmental biology 2022;10:830432
Biallelic KIF24 Variants Are Responsible for a Spectrum of Skeletal Disorders Ranging From Lethal Skeletal Ciliopathy to Severe Acromesomelic Dysplasia.
Reilly ML, Ain NU, Muurinen M, Tata A, Huber C, Simon M, Ishaq T, Shaw N, Rusanen S, Pekkinen M, Högler W, Knapen MFCM, van den Born M, Saunier S, Naz S, Cormier-Daire V, Benmerah A, Makitie O
Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research 2022 Sep;37(9):1642-1652
Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research 2022 Sep;37(9):1642-1652
KLC3 Regulates Ciliary Trafficking and Cyst Progression in CILK1 Deficiency-Related Polycystic Kidney Disease.
Rah G, Cha H, Kim J, Song J, Kim H, Oh YK, Ahn C, Kang M, Kim J, Yoo KH, Kim MJ, Ko HW, Ko JY, Park JH
Journal of the American Society of Nephrology : JASN 2022 Sep;33(9):1726-1741
Journal of the American Society of Nephrology : JASN 2022 Sep;33(9):1726-1741
Deletion of CEP164 in mouse photoreceptors post-ciliogenesis interrupts ciliary intraflagellar transport (IFT).
Reed M, Takemaru KI, Ying G, Frederick JM, Baehr W
PLoS genetics 2022 Sep;18(9):e1010154
PLoS genetics 2022 Sep;18(9):e1010154
Ttc21b deficiency attenuates autosomal dominant polycystic kidney disease in a kidney tubular- and maturation-dependent manner.
Wang W, Silva LM, Wang HH, Kavanaugh MA, Pottorf TS, Allard BA, Jacobs DT, Dong R, Cornelius JT, Chaturvedi A, Swenson-Fields KI, Fields TA, Pritchard MT, Sharma M, Slawson C, Wallace DP, Calvet JP, Tran PV
Kidney international 2022 Sep;102(3):577-591
Kidney international 2022 Sep;102(3):577-591
Plexins promote Hedgehog signaling through their cytoplasmic GAP activity.
Pinskey JM, Hoard TM, Zhao XF, Franks NE, Frank ZC, McMellen AN, Giger RJ, Allen BL
eLife 2022 Sep 28;11
eLife 2022 Sep 28;11
Islet primary cilia motility controls insulin secretion.
Cho JH, Li ZA, Zhu L, Muegge BD, Roseman HF, Lee EY, Utterback T, Woodhams LG, Bayly PV, Hughes JW
Science advances 2022 Sep 23;8(38):eabq8486
Science advances 2022 Sep 23;8(38):eabq8486
AMBRA1 p.Gln30Arg Mutation, Identified in a Cowden Syndrome Family, Exhibits Hyperproliferative Potential in hTERT-RPE1 Cells.
Revathidevi S, Hosomichi K, Natsume T, Nakaoka H, Fujito NT, Akatsuka H, Sato T, Munirajan AK, Inoue I
International journal of molecular sciences 2022 Sep 22;23(19)
International journal of molecular sciences 2022 Sep 22;23(19)
The Nicotinic Receptor Polymorphism rs16969968 Is Associated with Airway Remodeling and Inflammatory Dysregulation in COPD Patients.
Saber Cherif L, Diabasana Z, Perotin JM, Ancel J, Petit LMG, Devilliers MA, Bonnomet A, Lalun N, Delepine G, Maskos U, Gosset P, Polette M, Muggeo A, Guillard T, Deslée G, Dormoy V
Cells 2022 Sep 20;11(19)
Cells 2022 Sep 20;11(19)
The intraflagellar transport protein IFT52 associated with short-rib thoracic dysplasia is essential for ciliary function in osteogenic differentiation in vitro and for sensory perception in Drosophila.
Guleria VS, Parit R, Quadri N, Das R, Upadhyai P
Experimental cell research 2022 Sep 15;418(2):113273
Experimental cell research 2022 Sep 15;418(2):113273
Carnitine Protects against MPP(+)-Induced Neurotoxicity and Inflammation by Promoting Primary Ciliogenesis in SH-SY5Y Cells.
Bae JE, Kim JB, Jo DS, Park NY, Kim YH, Lee HJ, Kim SH, Kim SH, Son M, Kim P, Ryu HY, Lee WH, Ryoo ZY, Lee HS, Jung YK, Cho DH
Cells 2022 Sep 1;11(17)
Cells 2022 Sep 1;11(17)
A PKA inhibitor motif within SMOOTHENED controls Hedgehog signal transduction.
Happ JT, Arveseth CD, Bruystens J, Bertinetti D, Nelson IB, Olivieri C, Zhang J, Hedeen DS, Zhu JF, Capener JL, Bröckel JW, Vu L, King CC, Ruiz-Perez VL, Ge X, Veglia G, Herberg FW, Taylor SS, Myers BR
Nature structural & molecular biology 2022 Oct;29(10):990-999
Nature structural & molecular biology 2022 Oct;29(10):990-999
Heterozygous variants in MYH10 associated with neurodevelopmental disorders and congenital anomalies with evidence for primary cilia-dependent defects in Hedgehog signaling.
Holtz AM, VanCoillie R, Vansickle EA, Carere DA, Withrow K, Torti E, Juusola J, Millan F, Person R, Guillen Sacoto MJ, Si Y, Wentzensen IM, Pugh J, Vasileiou G, Rieger M, Reis A, Argilli E, Sherr EH, Aldinger KA, Dobyns WB, Brunet T, Hoefele J, Wagner M, Haber B, Kotzaeridou U, Keren B, Heron D, Mignot C, Heide S, Courtin T, Buratti J, Murugasen S, Donald KA, O'Heir E, Moody S, Kim KH, Burton BK, Yoon G, Campo MD, Masser-Frye D, Kozenko M, Parkinson C, Sell SL, Gordon PL, Prokop JW, Karaa A, Bupp C, Raby BA
Genetics in medicine : official journal of the American College of Medical Genetics 2022 Oct;24(10):2065-2078
Genetics in medicine : official journal of the American College of Medical Genetics 2022 Oct;24(10):2065-2078
The Primary Ciliary Deficits in Cerebellar Bergmann Glia of the Mouse Model of Fragile X Syndrome.
Lee B, Beuhler L, Lee HY
Cerebellum (London, England) 2022 Oct;21(5):801-813
Cerebellum (London, England) 2022 Oct;21(5):801-813
Endothelial β-arrestins regulate mechanotransduction by the type II bone morphogenetic protein receptor in primary cilia.
Park S, Ma Z, Zarkada G, Papangeli I, Paluri S, Nazo N, Rivera-Molina F, Toomre D, Rajagopal S, Chun HJ
Pulmonary circulation 2022 Oct;12(4):e12167
Pulmonary circulation 2022 Oct;12(4):e12167
Mutations in SCNM1 cause orofaciodigital syndrome due to minor intron splicing defects affecting primary cilia.
Iturrate A, Rivera-Barahona A, Flores CL, Otaify GA, Elhossini R, Perez-Sanz ML, Nevado J, Tenorio-Castano J, Triviño JC, Garcia-Gonzalo FR, Piceci-Sparascio F, De Luca A, Martínez L, Kalaycı T, Lapunzina P, Altunoglu U, Aglan M, Abdalla E, Ruiz-Perez VL
American journal of human genetics 2022 Oct 6;109(10):1828-1849
American journal of human genetics 2022 Oct 6;109(10):1828-1849
Analysis of Primary Cilium Expression and Hedgehog Pathway Activation in Mesothelioma Throws Back Its Complex Biology.
Barbarino M, Bottaro M, Spagnoletti L, de Santi MM, Guazzo R, Defraia C, Custoza C, Serio G, Iannelli F, Pesetti M, Aiello R, Rosati D, Zanfrini E, Luzzi L, Bellan C, Giordano A
Cancers 2022 Oct 25;14(21)
Cancers 2022 Oct 25;14(21)
KIF24 depletion induces clustering of supernumerary centrosomes in PDAC cells.
Mashima Y, Nohira H, Sugihara H, Dynlacht BD, Kobayashi T, Itoh H
Life science alliance 2022 Nov;5(11)
Life science alliance 2022 Nov;5(11)
Nuclear factor (erythroid-derived 2)-like 2 counter-regulates thymosin beta-4 expression and primary cilium formation for HeLa cervical cancer cell survival.
Lee JW, Thuy PX, Koo JH, Moon EY
Scientific reports 2022 Nov 23;12(1):20170
Scientific reports 2022 Nov 23;12(1):20170
HeLa Cervical Cancer Cells Are Maintained by Nephronophthisis 3-Associated Primary Cilium Formation via ROS-Induced ERK and HIF-1α Activation under Serum-Deprived Normoxic Condition.
Lee JW, Cho JY, Thuy PX, Moon EY
International journal of molecular sciences 2022 Nov 22;23(23)
International journal of molecular sciences 2022 Nov 22;23(23)
A Potent Antagonist of Smoothened in Hedgehog Signaling for Epilepsy.
Fan J, Zhao Z, Liu R, Li H, He W, Wu J, Wang Y, Chen W
International journal of molecular sciences 2022 Nov 22;23(23)
International journal of molecular sciences 2022 Nov 22;23(23)
A high-throughput electron tomography workflow reveals over-elongated centrioles in relapsed/refractory multiple myeloma.
Dittrich T, Köhrer S, Schorb M, Haberbosch I, Börmel M, Goldschmidt H, Pajor G, Müller-Tidow C, Raab MS, Hegenbart U, Schönland SO, Schwab Y, Krämer A
Cell reports methods 2022 Nov 21;2(11):100322
Cell reports methods 2022 Nov 21;2(11):100322
Metabolomic fingerprinting of renal disease progression in Bardet-Biedl syndrome reveals mitochondrial dysfunction in kidney tubular cells.
Marchese E, Caterino M, Viggiano D, Cevenini A, Tolone S, Docimo L, Di Iorio V, Del Vecchio Blanco F, Fedele R, Simonelli F, Perna A, Nigro V, Capasso G, Ruoppolo M, Zacchia M
iScience 2022 Nov 18;25(11):105230
iScience 2022 Nov 18;25(11):105230
Cilia-Mediated Insulin/Akt and ST2/JNK Signaling Pathways Regulate the Recovery of Muscle Injury.
Yamakawa D, Tsuboi J, Kasahara K, Matsuda C, Nishimura Y, Kodama T, Katayama N, Watanabe M, Inagaki M
Advanced science (Weinheim, Baden-Wurttemberg, Germany) 2022 Nov 14;10(1):e2202632
Advanced science (Weinheim, Baden-Wurttemberg, Germany) 2022 Nov 14;10(1):e2202632
LRRK1-mediated NDEL1 phosphorylation promotes cilia disassembly via dynein-2-driven retrograde intraflagellar transport.
Hanafusa H, Kedashiro S, Gotoh M, Saitoh KH, Inaba H, Nishioka T, Kaibuchi K, Inagaki M, Hisamoto N, Matsumoto K
Journal of cell science 2022 Nov 1;135(21)
Journal of cell science 2022 Nov 1;135(21)
Midbody plays an active role in fibroblast-myofibroblast transition by mediating TGF-β signaling.
Jung SW, Kim SM, Kim A, Park SH, Moon JY, Lee SH
FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2022 May;36(5):e22272
FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2022 May;36(5):e22272
Agonists of prostaglandin E(2) receptors as potential first in class treatment for nephronophthisis and related ciliopathies.
Garcia H, Serafin AS, Silbermann F, Porée E, Viau A, Mahaut C, Billot K, Birgy É, Garfa-Traore M, Roy S, Ceccarelli S, Mehraz M, Rodriguez PC, Deleglise B, Furio L, Jabot-Hanin F, Cagnard N, Del Nery E, Fila M, Sin-Monnot S, Antignac C, Lyonnet S, Krug P, Salomon R, Annereau JP, Benmerah A, Delous M, Briseño-Roa L, Saunier S
Proceedings of the National Academy of Sciences of the United States of America 2022 May 3;119(18):e2115960119
Proceedings of the National Academy of Sciences of the United States of America 2022 May 3;119(18):e2115960119
Inhibition of Ciliogenesis Enhances the Cellular Sensitivity to Temozolomide and Ionizing Radiation in Human Glioblastoma Cells.
Wei L, Ma W, Cai H, Peng SP, Tian HB, Wang JF, Gao L, He JP
Biomedical and environmental sciences : BES 2022 May 20;35(5):419-436
Biomedical and environmental sciences : BES 2022 May 20;35(5):419-436
The ciliary gene INPP5E confers dorsal telencephalic identity to human cortical organoids by negatively regulating Sonic hedgehog signaling.
Schembs L, Willems A, Hasenpusch-Theil K, Cooper JD, Whiting K, Burr K, Bøstrand SMK, Selvaraj BT, Chandran S, Theil T
Cell reports 2022 May 17;39(7):110811
Cell reports 2022 May 17;39(7):110811
Cholesterylation of Smoothened is a calcium-accelerated autoreaction involving an intramolecular ester intermediate.
Hu A, Zhang JZ, Wang J, Li CC, Yuan M, Deng G, Lin ZC, Qiu ZP, Liu HY, Wang XW, Wei PC, He X, Zhao X, Qiu WW, Song BL
Cell research 2022 Mar;32(3):288-301
Cell research 2022 Mar;32(3):288-301
Identification of a novel variant of the ciliopathic gene FUZZY associated with craniosynostosis.
Barrell WB, Adel Al-Lami H, Goos JAC, Swagemakers SMA, van Dooren M, Torban E, van der Spek PJ, Mathijssen IMJ, Liu KJ
European journal of human genetics : EJHG 2022 Mar;30(3):282-290
European journal of human genetics : EJHG 2022 Mar;30(3):282-290
The carboxyl-terminal region of SDCCAG8 comprises a functional module essential for cilia formation as well as organ development and homeostasis.
Tsutsumi R, Chaya T, Tsujii T, Furukawa T
The Journal of biological chemistry 2022 Mar;298(3):101686
The Journal of biological chemistry 2022 Mar;298(3):101686
TMED2 binding restricts SMO to the ER and Golgi compartments.
Di Minin G, Holzner M, Grison A, Dumeau CE, Chan W, Monfort A, Jerome-Majewska LA, Roelink H, Wutz A
PLoS biology 2022 Mar;20(3):e3001596
PLoS biology 2022 Mar;20(3):e3001596
Biallelic variants in CENPF causing a phenotype distinct from Strømme syndrome.
Cappuccio G, Brillante S, Tammaro R, Pinelli M, De Bernardi ML, Gensini MG, Bijlsma EK, Koopmann TT, Hoffer MJV, McDonald K, Hendon LG, Douzgou S, Deshpande C, D'Arrigo S, Torella A, Nigro V, Franco B, Brunetti-Pierri N
American journal of medical genetics. Part C, Seminars in medical genetics 2022 Mar;190(1):102-108
American journal of medical genetics. Part C, Seminars in medical genetics 2022 Mar;190(1):102-108
Evolutionary conservation of centriole rotational asymmetry in the human centrosome.
Gaudin N, Martin Gil P, Boumendjel M, Ershov D, Pioche-Durieu C, Bouix M, Delobelle Q, Maniscalco L, Phan TBN, Heyer V, Reina-San-Martin B, Azimzadeh J
eLife 2022 Mar 23;11
eLife 2022 Mar 23;11
Cilia proteins are biomarkers of altered flow in the vasculature.
Gupta A, Thirugnanam K, Thamilarasan M, Mohieldin AM, Zedan HT, Prabhudesai S, Griffin MR, Spearman AD, Pan A, Palecek SP, Yalcin HC, Nauli SM, Rarick KR, Zennadi R, Ramchandran R
JCI insight 2022 Mar 22;7(6)
JCI insight 2022 Mar 22;7(6)
3D-Structured Illumination Microscopy of Centrosomes in Human Cell Lines.
Frikstad KM, Schink KO, Gilani S, Pedersen LB, Patzke S
Bio-protocol 2022 Mar 20;12(6):e4360
Bio-protocol 2022 Mar 20;12(6):e4360
A protocol for generation and live-cell imaging analysis of primary cilia reporter cell lines.
Binó L, Mikulenková E, Štepánek L, Bernatík O, Vysloužil D, Pejšková P, Gorilák P, Huranová M, Varga V, Čajánek L
STAR protocols 2022 Mar 18;3(1):101199
STAR protocols 2022 Mar 18;3(1):101199
ALKBH3-dependent m(1)A demethylation of Aurora A mRNA inhibits ciliogenesis.
Kuang W, Jin H, Yang F, Chen X, Liu J, Li T, Chang Y, Liu M, Xu Z, Huo C, Yan X, Yang Y, Liu W, Shu Q, Xie S, Zhou T
Cell discovery 2022 Mar 11;8(1):25
Cell discovery 2022 Mar 11;8(1):25
Primary Cilium by Polyinosinic:Polycytidylic Acid Regulates the Regenerative Migration of Beas-2B Bronchial Epithelial Cells.
Gweon B, Jang TK, Thuy PX, Moon EY
Biomolecules & therapeutics 2022 Mar 1;30(2):170-178
Biomolecules & therapeutics 2022 Mar 1;30(2):170-178
Ciliary IFT88 Protects Coordinated Adolescent Growth Plate Ossification From Disruptive Physiological Mechanical Forces.
Coveney CR, Samvelyan HJ, Miotla-Zarebska J, Carnegie J, Chang E, Corrin CJ, Coveney T, Stott B, Parisi I, Duarte C, Vincent TL, Staines KA, Wann AK
Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research 2022 Jun;37(6):1081-1096
Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research 2022 Jun;37(6):1081-1096
Preliminary results on a proposed histopathological assessment of predictive factors for basal cell carcinoma recurrence after primary free margin excision.
Jacquet A, Dormoy V, Lorenzato M, Durlach A
Skin health and disease 2022 Jun;2(2):e88
Skin health and disease 2022 Jun;2(2):e88
Ciliary Proteins Repurposed by the Synaptic Ribbon: Trafficking Myristoylated Proteins at Rod Photoreceptor Synapses.
Suiwal S, Dembla M, Schwarz K, Katiyar R, Jung M, Carius Y, Maxeiner S, Lauterbach MA, Lancaster CRD, Schmitz F
International journal of molecular sciences 2022 Jun 27;23(13)
International journal of molecular sciences 2022 Jun 27;23(13)
Control of meiotic chromosomal bouquet and germ cell morphogenesis by the zygotene cilium.
Mytlis A, Kumar V, Qiu T, Deis R, Hart N, Levy K, Masek M, Shawahny A, Ahmad A, Eitan H, Nather F, Adar-Levor S, Birnbaum RY, Elia N, Bachmann-Gagescu R, Roy S, Elkouby YM
Science (New York, N.Y.) 2022 Jun 17;376(6599):eabh3104
Science (New York, N.Y.) 2022 Jun 17;376(6599):eabh3104
Pathogenic LRRK2 regulates centrosome cohesion via Rab10/RILPL1-mediated CDK5RAP2 displacement.
Fdez E, Madero-Pérez J, Lara Ordóñez AJ, Naaldijk Y, Fasiczka R, Aiastui A, Ruiz-Martínez J, López de Munain A, Cowley SA, Wade-Martins R, Hilfiker S
iScience 2022 Jun 17;25(6):104476
iScience 2022 Jun 17;25(6):104476
Angiomotin isoform 2 promotes binding of PALS1 to KIF13B at primary cilia and regulates ciliary length and signaling.
Morthorst SK, Nielsen C, Farinelli P, Anvarian Z, Rasmussen CBR, Serra-Marques A, Grigoriev I, Altelaar M, Fürstenberg N, Ludwig A, Akhmanova A, Christensen ST, Pedersen LB
Journal of cell science 2022 Jun 15;135(12)
Journal of cell science 2022 Jun 15;135(12)
The developmental stage of the medulloblastoma cell-of-origin restricts Sonic hedgehog pathway usage and drug sensitivity.
Smit MJ, Martini TEI, Armandari I, Bočkaj I, Zomerman WW, de Camargo Magalhães ES, Siragna Z, Meeuwsen TGJ, Scherpen FJG, Schoots MH, Ritsema M, den Dunnen WFA, Hoving EW, Paridaen JTML, de Haan G, Guryev V, Bruggeman SWM
Journal of cell science 2022 Jun 1;135(11)
Journal of cell science 2022 Jun 1;135(11)
Soft, Dynamic Hydrogel Confinement Improves Kidney Organoid Lumen Morphology and Reduces Epithelial-Mesenchymal Transition in Culture.
Ruiter FAA, Morgan FLC, Roumans N, Schumacher A, Slaats GG, Moroni L, LaPointe VLS, Baker MB
Advanced science (Weinheim, Baden-Wurttemberg, Germany) 2022 Jul;9(20):e2200543
Advanced science (Weinheim, Baden-Wurttemberg, Germany) 2022 Jul;9(20):e2200543
Differential requirements for the Eps15 homology domain proteins EHD4 and EHD2 in the regulation of mammalian ciliogenesis.
Jones T, Naslavsky N, Caplan S
Traffic (Copenhagen, Denmark) 2022 Jul;23(7):360-373
Traffic (Copenhagen, Denmark) 2022 Jul;23(7):360-373
A pair of primers facing at the double-strand break site enables to detect NHEJ-mediated indel mutations at a 1-bp resolution.
Ijaz F, Nakazato R, Setou M, Ikegami K
Scientific reports 2022 Jul 8;12(1):11681
Scientific reports 2022 Jul 8;12(1):11681
Palmitic acid control of ciliogenesis modulates insulin signaling in hypothalamic neurons through an autophagy-dependent mechanism.
Ávalos Y, Hernández-Cáceres MP, Lagos P, Pinto-Nuñez D, Rivera P, Burgos P, Díaz-Castro F, Joy-Immediato M, Venegas-Zamora L, Lopez-Gallardo E, Kretschmar C, Batista-Gonzalez A, Cifuentes-Araneda F, Toledo-Valenzuela L, Rodriguez-Peña M, Espinoza-Caicedo J, Perez-Leighton C, Bertocchi C, Cerda M, Troncoso R, Parra V, Budini M, Burgos PV, Criollo A, Morselli E
Cell death & disease 2022 Jul 28;13(7):659
Cell death & disease 2022 Jul 28;13(7):659
Modulation of Primary Cilia by Alvocidib Inhibition of CILK1.
Wang EX, Turner JS, Brautigan DL, Fu Z
International journal of molecular sciences 2022 Jul 23;23(15)
International journal of molecular sciences 2022 Jul 23;23(15)
Chondrocyte-specific response to stiffness-mediated primary cilia formation and centriole positioning.
Williantarra I, Leung S, Choi YS, Chhana A, McGlashan SR
American journal of physiology. Cell physiology 2022 Jul 1;323(1):C236-C247
American journal of physiology. Cell physiology 2022 Jul 1;323(1):C236-C247
Porcine choroid plexus-Riems cell line demonstrates altered polarization of transport proteins compared with the native epithelium.
Hochstetler A, Hulme L, Delpire E, Schwerk C, Schroten H, Preston D, Simpson S, Blazer-Yost BL
American journal of physiology. Cell physiology 2022 Jul 1;323(1):C1-C13
American journal of physiology. Cell physiology 2022 Jul 1;323(1):C1-C13
TTC30A and TTC30B Redundancy Protects IFT Complex B Integrity and Its Pivotal Role in Ciliogenesis.
Hoffmann F, Bolz S, Junger K, Klose F, Schubert T, Woerz F, Boldt K, Ueffing M, Beyer T
Genes 2022 Jul 1;13(7)
Genes 2022 Jul 1;13(7)
Ciliary Type III Adenylyl Cyclase in the VMH Is Crucial for High-Fat Diet-Induced Obesity Mediated by Autophagy.
Yang D, Wu X, Wang W, Zhou Y, Wang Z
Advanced science (Weinheim, Baden-Wurttemberg, Germany) 2022 Jan;9(3):e2102568
Advanced science (Weinheim, Baden-Wurttemberg, Germany) 2022 Jan;9(3):e2102568
Vangl2 participates in the primary ciliary assembly under low fluid shear stress in hUVECs.
Sheng X, Gao S, Sheng Y, Xie X, Wang J, He Y
Cell and tissue research 2022 Jan;387(1):95-109
Cell and tissue research 2022 Jan;387(1):95-109
The N-terminal Leu-Pro-Gln sequence of Rab34 is required for ciliogenesis in hTERT-RPE1 cells.
Oguchi ME, Homma Y, Fukuda M
Small GTPases 2022 Jan;13(1):77-83
Small GTPases 2022 Jan;13(1):77-83
Molecular mechanisms underlying the role of the centriolar CEP164-TTBK2 complex in ciliopathies.
Rosa E Silva I, Binó L, Johnson CM, Rutherford TJ, Neuhaus D, Andreeva A, Čajánek L, van Breugel M
Structure (London, England : 1993) 2022 Jan 6;30(1):114-128.e9
Structure (London, England : 1993) 2022 Jan 6;30(1):114-128.e9
Genome-wide screening in human kidney organoids identifies developmental and disease-related aspects of nephrogenesis.
Ungricht R, Guibbal L, Lasbennes MC, Orsini V, Beibel M, Waldt A, Cuttat R, Carbone W, Basler A, Roma G, Nigsch F, Tchorz JS, Hoepfner D, Hoppe PS
Cell stem cell 2022 Jan 6;29(1):160-175.e7
Cell stem cell 2022 Jan 6;29(1):160-175.e7
Evolutionarily conserved genetic interactions between nphp-4 and bbs-5 mutations exacerbate ciliopathy phenotypes.
Bentley-Ford MR, LaBonty M, Thomas HR, Haycraft CJ, Scott M, LaFayette C, Croyle MJ, Andersen RS, Parant JM, Yoder BK
Genetics 2022 Jan 4;220(1)
Genetics 2022 Jan 4;220(1)
LUBAC regulates ciliogenesis by promoting CP110 removal from the mother centriole.
Shen XL, Yuan JF, Qin XH, Song GP, Hu HB, Tu HQ, Song ZQ, Li PY, Xu YL, Li S, Jian XX, Li JN, He CY, Yu XP, Liang LY, Wu M, Han QY, Wang K, Li AL, Zhou T, Zhang YC, Wang N, Li HY
The Journal of cell biology 2022 Jan 3;221(1)
The Journal of cell biology 2022 Jan 3;221(1)
Theobroma cacao improves bone growth by modulating defective ciliogenesis in a mouse model of achondroplasia.
Martin L, Kaci N, Benoist-Lasselin C, Mondoloni M, Decaudaveine S, Estibals V, Cornille M, Loisay L, Flipo J, Demuynck B, de la Luz Cádiz-Gurrea M, Barbault F, Fernández-Arroyo S, Schibler L, Segura-Carretero A, Dambroise E, Legeai-Mallet L
Bone research 2022 Jan 25;10(1):8
Bone research 2022 Jan 25;10(1):8
C2cd6-encoded CatSperτ targets sperm calcium channel to Ca(2+) signaling domains in the flagellar membrane.
Hwang JY, Wang H, Lu Y, Ikawa M, Chung JJ
Cell reports 2022 Jan 18;38(3):110226
Cell reports 2022 Jan 18;38(3):110226
KIAA0319 influences cilia length, cell migration and mechanical cell-substrate interaction.
Diaz R, Kronenberg NM, Martinelli A, Liehm P, Riches AC, Gather MC, Paracchini S
Scientific reports 2022 Jan 14;12(1):722
Scientific reports 2022 Jan 14;12(1):722
Hedgehog-induced ciliary trafficking of kinesin-4 motor KIF7 requires intraflagellar transport but not KIF7's microtubule binding.
Yue Y, Engelke MF, Blasius TL, Verhey KJ
Molecular biology of the cell 2022 Jan 1;33(1):br1
Molecular biology of the cell 2022 Jan 1;33(1):br1
Regulators of tubulin polyglutamylation control nuclear shape and cilium disassembly by balancing microtubule and actin assembly.
Wang L, Paudyal SC, Kang Y, Owa M, Liang FX, Spektor A, Knaut H, Sánchez I, Dynlacht BD
Cell research 2022 Feb;32(2):190-209
Cell research 2022 Feb;32(2):190-209
Mutations in OSBPL2 cause hearing loss associated with primary cilia defects via sonic hedgehog signaling.
Shi H, Wang H, Zhang C, Lu Y, Yao J, Chen Z, Xing G, Wei Q, Cao X
JCI insight 2022 Feb 22;7(4)
JCI insight 2022 Feb 22;7(4)
TP53 promotes lineage commitment of human embryonic stem cells through ciliogenesis and sonic hedgehog signaling.
Sivakumar S, Qi S, Cheng N, Sathe AA, Kanchwala M, Kumar A, Evers BM, Xing C, Yu H
Cell reports 2022 Feb 15;38(7):110395
Cell reports 2022 Feb 15;38(7):110395
Cytoskeletal Protein 4.1G Is Essential for the Primary Ciliogenesis and Osteoblast Differentiation in Bone Formation.
Saito M, Hirano M, Izumi T, Mori Y, Ito K, Saitoh Y, Terada N, Sato T, Sukegawa J
International journal of molecular sciences 2022 Feb 14;23(4)
International journal of molecular sciences 2022 Feb 14;23(4)
GLI transcriptional repression is inert prior to Hedgehog pathway activation.
Lex RK, Zhou W, Ji Z, Falkenstein KN, Schuler KE, Windsor KE, Kim JD, Ji H, Vokes SA
Nature communications 2022 Feb 10;13(1):808
Nature communications 2022 Feb 10;13(1):808
The ARF GAPs ELMOD1 and ELMOD3 act at the Golgi and cilia to regulate ciliogenesis and ciliary protein traffic.
Turn RE, Hu Y, Dewees SI, Devi N, East MP, Hardin KR, Khatib T, Linnert J, Wolfrum U, Lim MJ, Casanova JE, Caspary T, Kahn RA
Molecular biology of the cell 2022 Feb 1;33(2):ar13
Molecular biology of the cell 2022 Feb 1;33(2):ar13
ExSTED microscopy reveals contrasting functions of dopamine and somatostatin CSF-c neurons along the lamprey central canal.
Jalalvand E, Alvelid J, Coceano G, Edwards S, Robertson B, Grillner S, Testa I
eLife 2022 Feb 1;11
eLife 2022 Feb 1;11
Biallelic loss-of-function variants in EXOC6B are associated with impaired primary ciliogenesis and cause spondylo-epi-metaphyseal dysplasia with joint laxity type 3.
Simsek-Kiper PO, Jacob P, Upadhyai P, Taşkıran ZE, Guleria VS, Karaosmanoglu B, Imren G, Gocmen R, Bhavani GS, Kausthubham N, Shah H, Utine GE, Boduroglu K, Girisha KM
Human mutation 2022 Dec;43(12):2116-2129
Human mutation 2022 Dec;43(12):2116-2129
ATM- and ATR-induced primary ciliogenesis promotes cisplatin resistance in pancreatic ductal adenocarcinoma.
Chao YY, Huang BM, Peng IC, Lee PR, Lai YS, Chiu WT, Lin YS, Lin SC, Chang JH, Chen PS, Tsai SJ, Wang CY
Journal of cellular physiology 2022 Dec;237(12):4487-4503
Journal of cellular physiology 2022 Dec;237(12):4487-4503
Primary Cilia Restrain PI3K-AKT Signaling to Orchestrate Human Decidualization.
Li B, Yan YP, Liang C, He YY, Wang Y, Li MY, Chen ST, Li Y, Liu AX, Yan GJ, Yang ZM
International journal of molecular sciences 2022 Dec 8;23(24)
International journal of molecular sciences 2022 Dec 8;23(24)
IFT-A structure reveals carriages for membrane protein transport into cilia.
Hesketh SJ, Mukhopadhyay AG, Nakamura D, Toropova K, Roberts AJ
Cell 2022 Dec 22;185(26):4971-4985.e16
Cell 2022 Dec 22;185(26):4971-4985.e16
Primary cilia suppress Ripk3-mediated necroptosis.
Kieckhöfer E, Slaats GG, Ebert LK, Albert MC, Dafinger C, Kashkar H, Benzing T, Schermer B
Cell death discovery 2022 Dec 2;8(1):477
Cell death discovery 2022 Dec 2;8(1):477
Non-cell-autonomous activation of hedgehog signaling contributes to disease progression in a mouse model of renal cystic ciliopathy.
Hsieh CL, Jerman SJ, Sun Z
Human molecular genetics 2022 Dec 16;31(24):4228-4240
Human molecular genetics 2022 Dec 16;31(24):4228-4240
FOXA1 is a transcriptional activator of Odf2/Cenexin and regulates primary ciliation.
Czerny CC, Borschel A, Cai M, Otto M, Hoyer-Fender S
Scientific reports 2022 Dec 12;12(1):21468
Scientific reports 2022 Dec 12;12(1):21468
Characterization of TRPV4-mediated signaling pathways in an optimized human choroid plexus epithelial cell line.
Hulme L, Hochstetler A, Schwerk C, Schroten H, Ishikawa H, Tung CY, Perrin B, Blazer-Yost B
American journal of physiology. Cell physiology 2022 Dec 1;323(6):C1823-C1842
American journal of physiology. Cell physiology 2022 Dec 1;323(6):C1823-C1842
Distribution of prototypical primary cilia markers in subtypes of retinal ganglion cells.
Kowal TJ, Dhande OS, Wang B, Wang Q, Ning K, Liu W, Berbari NF, Hu Y, Sun Y
The Journal of comparative neurology 2022 Aug;530(12):2176-2187
The Journal of comparative neurology 2022 Aug;530(12):2176-2187
Disruption of GMNC-MCIDAS multiciliogenesis program is critical in choroid plexus carcinoma development.
Li Q, Han Z, Singh N, Terré B, Fame RM, Arif U, Page TD, Zahran T, Abdeltawab A, Huang Y, Cao P, Wang J, Lu H, Lidov HGW, Surendran K, Wu L, Virga JQ, Zhao YT, Schüller U, Wechsler-Reya RJ, Lehtinen MK, Roy S, Liu Z, Stracker TH, Zhao H
Cell death and differentiation 2022 Aug;29(8):1596-1610
Cell death and differentiation 2022 Aug;29(8):1596-1610
Ciliogenesis mechanisms mediated by PAK2-ARL13B signaling in brain endothelial cells is responsible for vascular stability.
Thirugnanam K, Prabhudesai S, Van Why E, Pan A, Gupta A, Foreman K, Zennadi R, Rarick KR, Nauli SM, Palecek SP, Ramchandran R
Biochemical pharmacology 2022 Aug;202:115143
Biochemical pharmacology 2022 Aug;202:115143
Monoallelic pathogenic ALG5 variants cause atypical polycystic kidney disease and interstitial fibrosis.
Lemoine H, Raud L, Foulquier F, Sayer JA, Lambert B, Olinger E, Lefèvre S, Knebelmann B, Harris PC, Trouvé P, Desprès A, Duneau G, Matignon M, Poyet A, Jourde-Chiche N, Guerrot D, Lemoine S, Seret G, Barroso-Gil M, Bingham C, Gilbert R, Genomics England Research Consortium, Genkyst Study Group, Le Meur Y, Audrézet MP, Cornec-Le Gall E
American journal of human genetics 2022 Aug 4;109(8):1484-1499
American journal of human genetics 2022 Aug 4;109(8):1484-1499
A cAMP signalosome in primary cilia drives gene expression and kidney cyst formation.
Hansen JN, Kaiser F, Leyendecker P, Stüven B, Krause JH, Derakhshandeh F, Irfan J, Sroka TJ, Preval KM, Desai PB, Kraut M, Theis H, Drews AD, De-Domenico E, Händler K, Pazour GJ, Henderson DJP, Mick DU, Wachten D
EMBO reports 2022 Aug 3;23(8):e54315
EMBO reports 2022 Aug 3;23(8):e54315
Heterogeneity of the NIH3T3 Fibroblast Cell Line.
Rahimi AM, Cai M, Hoyer-Fender S
Cells 2022 Aug 28;11(17)
Cells 2022 Aug 28;11(17)
GJA1 depletion causes ciliary defects by affecting Rab11 trafficking to the ciliary base.
Jang DG, Kwon KY, Kweon YC, Kim BG, Myung K, Lee HS, Young Park C, Kwon T, Park TJ
eLife 2022 Aug 25;11
eLife 2022 Aug 25;11
Subtype-Selective Positive Modulation of K(Ca)2.3 Channels Increases Cilia Length.
Nam YW, Pala R, El-Sayed NS, Larin-Henriquez D, Amirrad F, Yang G, Rahman MA, Orfali R, Downey M, Parang K, Nauli SM, Zhang M
ACS chemical biology 2022 Aug 19;17(8):2344-2354
ACS chemical biology 2022 Aug 19;17(8):2344-2354
Primary cilia and SHH signaling impairments in human and mouse models of Parkinson's disease.
Schmidt S, Luecken MD, Trümbach D, Hembach S, Niedermeier KM, Wenck N, Pflügler K, Stautner C, Böttcher A, Lickert H, Ramirez-Suastegui C, Ahmad R, Ziller MJ, Fitzgerald JC, Ruf V, van de Berg WDJ, Jonker AJ, Gasser T, Winner B, Winkler J, Vogt Weisenhorn DM, Giesert F, Theis FJ, Wurst W
Nature communications 2022 Aug 16;13(1):4819
Nature communications 2022 Aug 16;13(1):4819
The LRRK2 signaling network converges on a centriolar phospho-Rab10/RILPL1 complex to cause deficits in centrosome cohesion and cell polarization.
Lara Ordóñez AJ, Fasiczka R, Fernández B, Naaldijk Y, Fdez E, Blanca Ramírez M, Phan S, Boassa D, Hilfiker S
Biology open 2022 Aug 15;11(8)
Biology open 2022 Aug 15;11(8)
Precise control of microtubule disassembly in living cells.
Liu GY, Chen SC, Lee GH, Shaiv K, Chen PY, Cheng H, Hong SR, Yang WT, Huang SH, Chang YC, Wang HC, Kao CL, Sun PC, Chao MH, Lee YY, Tang MJ, Lin YC
The EMBO journal 2022 Aug 1;41(15):e110472
The EMBO journal 2022 Aug 1;41(15):e110472
Modelling ciliopathy phenotypes in human tissues derived from pluripotent stem cells with genetically ablated cilia.
Cruz NM, Reddy R, McFaline-Figueroa JL, Tran C, Fu H, Freedman BS
Nature biomedical engineering 2022 Apr;6(4):463-475
Nature biomedical engineering 2022 Apr;6(4):463-475
CDK7/12/13 inhibition targets an oscillating leukemia stem cell network and synergizes with venetoclax in acute myeloid leukemia.
He L, Arnold C, Thoma J, Rohde C, Kholmatov M, Garg S, Hsiao CC, Viol L, Zhang K, Sun R, Schmidt C, Janssen M, MacRae T, Huber K, Thiede C, Hébert J, Sauvageau G, Spratte J, Fluhr H, Aust G, Müller-Tidow C, Niehrs C, Pereira G, Hamann J, Tanaka M, Zaugg JB, Pabst C
EMBO molecular medicine 2022 Apr 7;14(4):e14990
EMBO molecular medicine 2022 Apr 7;14(4):e14990
Phylogenetic profiling and cellular analyses of ARL16 reveal roles in traffic of IFT140 and INPP5E.
Dewees SI, Vargová R, Hardin KR, Turn RE, Devi S, Linnert J, Wolfrum U, Caspary T, Eliáš M, Kahn RA
Molecular biology of the cell 2022 Apr 1;33(4):ar33
Molecular biology of the cell 2022 Apr 1;33(4):ar33
Mitotic Maturation Compensates for Premature Centrosome Splitting and PCM Loss in Human cep135 Knockout Cells.
Chu Z, Gruss OJ
Cells 2022 Apr 1;11(7)
Cells 2022 Apr 1;11(7)
ATXN10 Is Required for Embryonic Heart Development and Maintenance of Epithelial Cell Phenotypes in the Adult Kidney and Pancreas.
Bentley-Ford MR, Andersen RS, Croyle MJ, Haycraft CJ, Clearman KR, Foote JB, Reiter JF, Yoder BK
Frontiers in cell and developmental biology 2021;9:705182
Frontiers in cell and developmental biology 2021;9:705182
Centriolar Protein C2cd3 Is Required for Craniofacial Development.
Chang CF, Brown KM, Yang Y, Brugmann SA
Frontiers in cell and developmental biology 2021;9:647391
Frontiers in cell and developmental biology 2021;9:647391
Primary Cilia Formation Does Not Rely on WNT/β-Catenin Signaling.
Bernatik O, Paclikova P, Kotrbova A, Bryja V, Cajanek L
Frontiers in cell and developmental biology 2021;9:623753
Frontiers in cell and developmental biology 2021;9:623753
Loss of Ciliary Gene Bbs8 Results in Physiological Defects in the Retinal Pigment Epithelium.
Schneider S, De Cegli R, Nagarajan J, Kretschmer V, Matthiessen PA, Intartaglia D, Hotaling N, Ueffing M, Boldt K, Conte I, May-Simera HL
Frontiers in cell and developmental biology 2021;9:607121
Frontiers in cell and developmental biology 2021;9:607121
Palmitoylation of acetylated tubulin and association with ceramide-rich platforms is critical for ciliogenesis.
Tripathi P, Zhu Z, Qin H, Elsherbini A, Crivelli SM, Roush E, Wang G, Spassieva SD, Bieberich E
Journal of lipid research 2021;62:100021
Journal of lipid research 2021;62:100021
Primary Cilia as a Biomarker in Mesenchymal Stem Cells Senescence: Influencing Osteoblastic Differentiation Potency Associated with Hedgehog Signaling Regulation.
Fu S, Zhang C, Yan X, Li D, Wang Y, Dong C, Cao Z, Ning Y, Shao C, Yang T
Stem cells international 2021;2021:8850114
Stem cells international 2021;2021:8850114
Differential requirement of NPHP1 for compartmentalized protein localization during photoreceptor outer segment development and maintenance.
Datta P, Cribbs JT, Seo S
PloS one 2021;16(5):e0246358
PloS one 2021;16(5):e0246358
TALPID3/KIAA0586 Regulates Multiple Aspects of Neuromuscular Patterning During Gastrointestinal Development in Animal Models and Human.
Delalande JM, Nagy N, McCann CJ, Natarajan D, Cooper JE, Carreno G, Dora D, Campbell A, Laurent N, Kemos P, Thomas S, Alby C, Attié-Bitach T, Lyonnet S, Logan MP, Goldstein AM, Davey MG, Hofstra RMW, Thapar N, Burns AJ
Frontiers in molecular neuroscience 2021;14:757646
Frontiers in molecular neuroscience 2021;14:757646
Primary Cilia Mediate TSH-Regulated Thyroglobulin Endocytic Pathways.
Lee J, Sul HJ, Kim KH, Chang JY, Shong M
Frontiers in endocrinology 2021;12:700083
Frontiers in endocrinology 2021;12:700083
Quantitative lineage analysis identifies a hepato-pancreato-biliary progenitor niche.
Willnow D, Benary U, Margineanu A, Vignola ML, Konrath F, Pongrac IM, Karimaddini Z, Vigilante A, Wolf J, Spagnoli FM
Nature 2021 Sep;597(7874):87-91
Nature 2021 Sep;597(7874):87-91
The type 3 adenylyl cyclase is crucial for intestinal mucosal neural network in the gut lamina propria.
Zhou K, Zhou Y, Yang D, Chen T, Liu X, Li S, Wang Z
Neurogastroenterology and motility 2021 Sep;33(9):e14140
Neurogastroenterology and motility 2021 Sep;33(9):e14140
Recombinant Antithrombin Attenuates Acute Respiratory Distress Syndrome in Experimental Endotoxemia.
Okamoto H, Muraki I, Okada H, Tomita H, Suzuki K, Takada C, Wakayama Y, Kuroda A, Fukuda H, Kawasaki Y, Nishio A, Matsuo M, Tamaoki Y, Inagawa R, Takashima S, Taniguchi T, Suzuki A, Suzuki K, Miyazaki N, Kakino Y, Yasuda R, Fukuta T, Kitagawa Y, Miyake T, Doi T, Yoshida T, Yoshida S, Ogura S
The American journal of pathology 2021 Sep;191(9):1526-1536
The American journal of pathology 2021 Sep;191(9):1526-1536
Cilium induction triggers differentiation of glioma stem cells.
Goranci-Buzhala G, Mariappan A, Ricci-Vitiani L, Josipovic N, Pacioni S, Gottardo M, Ptok J, Schaal H, Callaini G, Rajalingam K, Dynlacht B, Hadian K, Papantonis A, Pallini R, Gopalakrishnan J
Cell reports 2021 Sep 7;36(10):109656
Cell reports 2021 Sep 7;36(10):109656
2-IPMA Ameliorates PM2.5-Induced Inflammation by Promoting Primary Ciliogenesis in RPE Cells.
Choi JY, Bae JE, Kim JB, Jo DS, Park NY, Kim YH, Lee HJ, Kim SH, Kim SH, Jeon HB, Na HW, Choi H, Ryu HY, Ryoo ZY, Lee HS, Cho DH
Molecules (Basel, Switzerland) 2021 Sep 6;26(17)
Molecules (Basel, Switzerland) 2021 Sep 6;26(17)
A ciliopathy complex builds distal appendages to initiate ciliogenesis.
Kumar D, Rains A, Herranz-Pérez V, Lu Q, Shi X, Swaney DL, Stevenson E, Krogan NJ, Huang B, Westlake C, Garcia-Verdugo JM, Yoder BK, Reiter JF
The Journal of cell biology 2021 Sep 6;220(9)
The Journal of cell biology 2021 Sep 6;220(9)
MAPK15 Controls Hedgehog Signaling in Medulloblastoma Cells by Regulating Primary Ciliogenesis.
Pietrobono S, Franci L, Imperatore F, Zanini C, Stecca B, Chiariello M
Cancers 2021 Sep 29;13(19)
Cancers 2021 Sep 29;13(19)
Bardet-Biedl syndrome proteins modulate the release of bioactive extracellular vesicles.
Volz AK, Frei A, Kretschmer V, de Jesus Domingues AM, Ketting RF, Ueffing M, Boldt K, Krämer-Albers EM, May-Simera HL
Nature communications 2021 Sep 27;12(1):5671
Nature communications 2021 Sep 27;12(1):5671
Elucidation of Distinct Modular Assemblies of Smoothened Receptor by Bitopic Ligand Measurement.
Zhao F, Wu Y, Zhou F, Xue D, Zhao S, Lu W, Liu X, Hu T, Qiu Y, Li R, Gu T, Xu Y, Xu F, Zhong G, Jiang Z, Zhao S, Tao H
Journal of medicinal chemistry 2021 Sep 23;64(18):13830-13840
Journal of medicinal chemistry 2021 Sep 23;64(18):13830-13840
Identification of disease-relevant modulators of the SHH pathway in the developing brain.
Mecklenburg N, Kowalczyk I, Witte F, Görne J, Laier A, Mamo TM, Gonschior H, Lehmann M, Richter M, Sporbert A, Purfürst B, Hübner N, Hammes A
Development (Cambridge, England) 2021 Sep 1;148(17)
Development (Cambridge, England) 2021 Sep 1;148(17)
Oxidative stress following acute kidney injury causes disruption of lung cell cilia and their release into the bronchoaveolar lavage fluid and lung injury, which are exacerbated by Idh2 deletion.
Han YK, Kim JS, Lee GB, Lim JH, Park KM
Redox biology 2021 Oct;46:102077
Redox biology 2021 Oct;46:102077
DZIP1 regulates mammalian cardiac valve development through a Cby1-β-catenin mechanism.
Guo L, Beck T, Fulmer D, Ramos-Ortiz S, Glover J, Wang C, Moore K, Gensemer C, Morningstar J, Moore R, Schott JJ, Le Tourneau T, Koren N, Norris RA
Developmental dynamics : an official publication of the American Association of Anatomists 2021 Oct;250(10):1432-1449
Developmental dynamics : an official publication of the American Association of Anatomists 2021 Oct;250(10):1432-1449
Cep55 regulation of PI3K/Akt signaling is required for neocortical development and ciliogenesis.
Rashidieh B, Shohayeb B, Bain AL, Fortuna PRJ, Sinha D, Burgess A, Mills R, Adams RC, Lopez JA, Blumbergs P, Finnie J, Kalimutho M, Piper M, Hudson JE, Ng DCH, Khanna KK
PLoS genetics 2021 Oct;17(10):e1009334
PLoS genetics 2021 Oct;17(10):e1009334
Human brain organoids assemble functionally integrated bilateral optic vesicles.
Gabriel E, Albanna W, Pasquini G, Ramani A, Josipovic N, Mariappan A, Schinzel F, Karch CM, Bao G, Gottardo M, Suren AA, Hescheler J, Nagel-Wolfrum K, Persico V, Rizzoli SO, Altmüller J, Riparbelli MG, Callaini G, Goureau O, Papantonis A, Busskamp V, Schneider T, Gopalakrishnan J
Cell stem cell 2021 Oct 7;28(10):1740-1757.e8
Cell stem cell 2021 Oct 7;28(10):1740-1757.e8
Induction of inverted morphology in brain organoids by vertical-mixing bioreactors.
Suong DNA, Imamura K, Inoue I, Kabai R, Sakamoto S, Okumura T, Kato Y, Kondo T, Yada Y, Klein WL, Watanabe A, Inoue H
Communications biology 2021 Oct 22;4(1):1213
Communications biology 2021 Oct 22;4(1):1213
Ciliary Hedgehog signaling regulates cell survival to build the facial midline.
Abrams SR, Reiter JF
eLife 2021 Oct 21;10
eLife 2021 Oct 21;10
Primary Ciliogenesis by 2-Isopropylmalic Acid Prevents PM2.5-Induced Inflammatory Response and MMP-1 Activation in Human Dermal Fibroblasts and a 3-D-Skin Model.
Bae JE, Min D, Choi JY, Choi H, Kim JB, Park NY, Jo DS, Kim YH, Na HW, Kim YJ, Kim ES, Kim HJ, Cho DH
International journal of molecular sciences 2021 Oct 10;22(20)
International journal of molecular sciences 2021 Oct 10;22(20)
Δ9-Tetrahydrocannabinol inhibits Hedgehog-dependent patterning during development.
Lo HF, Hong M, Szutorisz H, Hurd YL, Krauss RS
Development (Cambridge, England) 2021 Oct 1;148(19)
Development (Cambridge, England) 2021 Oct 1;148(19)
Gene replacement therapy restores RCBTB1 expression and cilium length in patient-derived retinal pigment epithelium.
Huang Z, Zhang D, Chen SC, Jennings L, Carvalho LS, Fletcher S, Chen FK, McLenachan S
Journal of cellular and molecular medicine 2021 Nov;25(21):10020-10027
Journal of cellular and molecular medicine 2021 Nov;25(21):10020-10027
A WDR35-dependent coat protein complex transports ciliary membrane cargo vesicles to cilia.
Quidwai T, Wang J, Hall EA, Petriman NA, Leng W, Kiesel P, Wells JN, Murphy LC, Keighren MA, Marsh JA, Lorentzen E, Pigino G, Mill P
eLife 2021 Nov 4;10
eLife 2021 Nov 4;10
Extracellular LGALS3BP regulates neural progenitor position and relates to human cortical complexity.
Kyrousi C, O'Neill AC, Brazovskaja A, He Z, Kielkowski P, Coquand L, Di Giaimo R, D' Andrea P, Belka A, Forero Echeverry A, Mei D, Lenge M, Cruceanu C, Buchsbaum IY, Khattak S, Fabien G, Binder E, Elmslie F, Guerrini R, Baffet AD, Sieber SA, Treutlein B, Robertson SP, Cappello S
Nature communications 2021 Nov 2;12(1):6298
Nature communications 2021 Nov 2;12(1):6298
Hydrogen sulfide, a gaseous signaling molecule, elongates primary cilia on kidney tubular epithelial cells by activating extracellular signal-regulated kinase.
Han SJ, Kim JI, Lipschutz JH, Park KM
The Korean journal of physiology & pharmacology : official journal of the Korean Physiological Society and the Korean Society of Pharmacology 2021 Nov 1;25(6):593-601
The Korean journal of physiology & pharmacology : official journal of the Korean Physiological Society and the Korean Society of Pharmacology 2021 Nov 1;25(6):593-601
Kif3a deletion prevents primary cilia assembly on oligodendrocyte progenitor cells, reduces oligodendrogenesis and impairs fine motor function.
Cullen CL, O'Rourke M, Beasley SJ, Auderset L, Zhen Y, Pepper RE, Gasperini R, Young KM
Glia 2021 May;69(5):1184-1203
Glia 2021 May;69(5):1184-1203
LRRK2-phosphorylated Rab10 sequesters Myosin Va with RILPL2 during ciliogenesis blockade.
Dhekne HS, Yanatori I, Vides EG, Sobu Y, Diez F, Tonelli F, Pfeffer SR
Life science alliance 2021 May;4(5)
Life science alliance 2021 May;4(5)
Deletion of Sox9 in the liver leads to hepatic cystogenesis in mice by transcriptionally downregulating Sec63.
Xu WP, Cui YL, Chen LL, Ding K, Ding CH, Chen F, Zhang X, Xie WF
The Journal of pathology 2021 May;254(1):57-69
The Journal of pathology 2021 May;254(1):57-69
De novo purine biosynthesis is a major driver of chemoresistance in glioblastoma.
Shireman JM, Atashi F, Lee G, Ali ES, Saathoff MR, Park CH, Savchuk S, Baisiwala S, Miska J, Lesniak MS, James CD, Stupp R, Kumthekar P, Horbinski CM, Ben-Sahra I, Ahmed AU
Brain : a journal of neurology 2021 May 7;144(4):1230-1246
Brain : a journal of neurology 2021 May 7;144(4):1230-1246
Time-resolved proteomics profiling of the ciliary Hedgehog response.
May EA, Kalocsay M, D'Auriac IG, Schuster PS, Gygi SP, Nachury MV, Mick DU
The Journal of cell biology 2021 May 3;220(5)
The Journal of cell biology 2021 May 3;220(5)
The TBC1D31/praja2 complex controls primary ciliogenesis through PKA-directed OFD1 ubiquitylation.
Senatore E, Chiuso F, Rinaldi L, Intartaglia D, Delle Donne R, Pedone E, Catalanotti B, Pirone L, Fiorillo B, Moraca F, Giamundo G, Scala G, Raffeiner A, Torres-Quesada O, Stefan E, Kwiatkowski M, van Pijkeren A, Morleo M, Franco B, Garbi C, Conte I, Feliciello A
The EMBO journal 2021 May 17;40(10):e106503
The EMBO journal 2021 May 17;40(10):e106503
Primary cilia-dependent lipid raft/caveolin dynamics regulate adipogenesis.
Yamakawa D, Katoh D, Kasahara K, Shiromizu T, Matsuyama M, Matsuda C, Maeno Y, Watanabe M, Nishimura Y, Inagaki M
Cell reports 2021 Mar 9;34(10):108817
Cell reports 2021 Mar 9;34(10):108817
CiliaQ: a simple, open-source software for automated quantification of ciliary morphology and fluorescence in 2D, 3D, and 4D images.
Hansen JN, Rassmann S, Stüven B, Jurisch-Yaksi N, Wachten D
The European physical journal. E, Soft matter 2021 Mar 8;44(2):18
The European physical journal. E, Soft matter 2021 Mar 8;44(2):18
Investigating Primary Cilia during Peripheral Nervous System Formation.
Yusifov E, Dumoulin A, Stoeckli ET
International journal of molecular sciences 2021 Mar 20;22(6)
International journal of molecular sciences 2021 Mar 20;22(6)
Identification of a potent antagonist of smoothened in hedgehog signaling.
Fan J, Li H, Kuang L, Zhao Z, He W, Liu C, Wang Y, Cheng SY, Chen W
Cell & bioscience 2021 Mar 2;11(1):46
Cell & bioscience 2021 Mar 2;11(1):46
Sub-toxic levels of cobalt ions impair chondrocyte mechanostranduction via HDAC6-dependent primary cilia shortening.
Wu H, Wang Z, Liu S, Meng H, Liu S, Fu S
Biochemical and biophysical research communications 2021 Mar 12;544:38-43
Biochemical and biophysical research communications 2021 Mar 12;544:38-43
Exovesicular-Shh confers Imatinib resistance by upregulating Bcl2 expression in chronic myeloid leukemia with variant chromosomes.
Anusha, Dalal H, Subramanian S, V P S, Gowda DA, H K, Damodar S, Vyas N
Cell death & disease 2021 Mar 11;12(3):259
Cell death & disease 2021 Mar 11;12(3):259
Exon junction complex dependent mRNA localization is linked to centrosome organization during ciliogenesis.
Kwon OS, Mishra R, Safieddine A, Coleno E, Alasseur Q, Faucourt M, Barbosa I, Bertrand E, Spassky N, Le Hir H
Nature communications 2021 Mar 1;12(1):1351
Nature communications 2021 Mar 1;12(1):1351
Ciliopathy protein HYLS1 coordinates the biogenesis and signaling of primary cilia by activating the ciliary lipid kinase PIPKIγ.
Chen C, Xu Q, Zhang Y, Davies BA, Huang Y, Katzmann DJ, Harris PC, Hu J, Ling K
Science advances 2021 Jun;7(26)
Science advances 2021 Jun;7(26)
Increase in primary cilia in the epidermis of patients with atopic dermatitis and psoriasis.
Rizaldy D, Toriyama M, Kato H, Fukui R, Fujita F, Nakamura M, Okada F, Morita A, Ishii KJ
Experimental dermatology 2021 Jun;30(6):792-803
Experimental dermatology 2021 Jun;30(6):792-803
Genotoxic stress-activated DNA-PK-p53 cascade and autophagy cooperatively induce ciliogenesis to maintain the DNA damage response.
Chen TY, Huang BM, Tang TK, Chao YY, Xiao XY, Lee PR, Yang LY, Wang CY
Cell death and differentiation 2021 Jun;28(6):1865-1879
Cell death and differentiation 2021 Jun;28(6):1865-1879
Disrupted intraflagellar transport due to IFT74 variants causes Joubert syndrome.
Luo M, Lin Z, Zhu T, Jin M, Meng D, He R, Cao Z, Shen Y, Lu C, Cai R, Zhao Y, Wang X, Li H, Wu S, Zou X, Luo G, Cao L, Huang M, Jiao H, Gao H, Sui R, Zhao C, Ma X, Cao M
Genetics in medicine : official journal of the American College of Medical Genetics 2021 Jun;23(6):1041-1049
Genetics in medicine : official journal of the American College of Medical Genetics 2021 Jun;23(6):1041-1049
Ciliogenesis is Not Directly Regulated by LRRK2 Kinase Activity in Neurons.
Kim H, Sim H, Lee JE, Seo MK, Lim J, Bang Y, Nam D, Lee SY, Chung SK, Choi HJ, Park SW, Son I, Kim J, Seol W
Experimental neurobiology 2021 Jun 30;30(3):232-243
Experimental neurobiology 2021 Jun 30;30(3):232-243
Automatic and unbiased segmentation and quantification of myofibers in skeletal muscle.
Waisman A, Norris AM, Elías Costa M, Kopinke D
Scientific reports 2021 Jun 3;11(1):11793
Scientific reports 2021 Jun 3;11(1):11793
Identification of Cilia in Different Mouse Tissues.
Li X, Yang S, Deepak V, Chinipardaz Z, Yang S
Cells 2021 Jun 29;10(7)
Cells 2021 Jun 29;10(7)
Modeling retinitis pigmentosa through patient-derived retinal organoids.
Li YP, Deng WL, Jin ZB
STAR protocols 2021 Jun 18;2(2):100438
STAR protocols 2021 Jun 18;2(2):100438
Comprehensive interactome profiling of the human Hsp70 network highlights functional differentiation of J domains.
Piette BL, Alerasool N, Lin ZY, Lacoste J, Lam MHY, Qian WW, Tran S, Larsen B, Campos E, Peng J, Gingras AC, Taipale M
Molecular cell 2021 Jun 17;81(12):2549-2565.e8
Molecular cell 2021 Jun 17;81(12):2549-2565.e8
Trace Amine-Associated Receptor 1 Trafficking to Cilia of Thyroid Epithelial Cells.
Qatato M, Venugopalan V, Al-Hashimi A, Rehders M, Valentine AD, Hein Z, Dallto U, Springer S, Brix K
Cells 2021 Jun 16;10(6)
Cells 2021 Jun 16;10(6)
TRIM37 prevents formation of condensate-organized ectopic spindle poles to ensure mitotic fidelity.
Meitinger F, Kong D, Ohta M, Desai A, Oegema K, Loncarek J
The Journal of cell biology 2021 Jul 5;220(7)
The Journal of cell biology 2021 Jul 5;220(7)
Myocardin-related transcription factor and serum response factor regulate cilium turnover by both transcriptional and local mechanisms.
Speight P, Rozycki M, Venugopal S, Szászi K, Kofler M, Kapus A
iScience 2021 Jul 23;24(7):102739
iScience 2021 Jul 23;24(7):102739
CEP78 functions downstream of CEP350 to control biogenesis of primary cilia by negatively regulating CP110 levels.
Gonçalves AB, Hasselbalch SK, Joensen BB, Patzke S, Martens P, Ohlsen SK, Quinodoz M, Nikopoulos K, Suleiman R, Damsø Jeppesen MP, Weiss C, Christensen ST, Rivolta C, Andersen JS, Farinelli P, Pedersen LB
eLife 2021 Jul 14;10
eLife 2021 Jul 14;10
Rab34 GTPase mediates ciliary membrane formation in the intracellular ciliogenesis pathway.
Ganga AK, Kennedy MC, Oguchi ME, Gray S, Oliver KE, Knight TA, De La Cruz EM, Homma Y, Fukuda M, Breslow DK
Current biology : CB 2021 Jul 12;31(13):2895-2905.e7
Current biology : CB 2021 Jul 12;31(13):2895-2905.e7
Rab34 is necessary for early stages of intracellular ciliogenesis.
Stuck MW, Chong WM, Liao JC, Pazour GJ
Current biology : CB 2021 Jul 12;31(13):2887-2894.e4
Current biology : CB 2021 Jul 12;31(13):2887-2894.e4
Targeting an anchored phosphatase-deacetylase unit restores renal ciliary homeostasis.
Gopalan J, Omar MH, Roy A, Cruz NM, Falcone J, Jones KN, Forbush KA, Himmelfarb J, Freedman BS, Scott JD
eLife 2021 Jul 12;10
eLife 2021 Jul 12;10
Sequences in the stalk domain regulate auto-inhibition and ciliary tip localization of the immotile kinesin-4 KIF7.
Blasius TL, Yue Y, Prasad R, Liu X, Gennerich A, Verhey KJ
Journal of cell science 2021 Jul 1;134(13)
Journal of cell science 2021 Jul 1;134(13)
TMEM16A/ANO1 calcium-activated chloride channel as a novel target for the treatment of human respiratory syncytial virus infection.
Pearson H, Todd EJAA, Ahrends M, Hover SE, Whitehouse A, Stacey M, Lippiat JD, Wilkens L, Fieguth HG, Danov O, Hesse C, Barr JN, Mankouri J
Thorax 2021 Jan;76(1):64-72
Thorax 2021 Jan;76(1):64-72
DAB2IP modulates primary cilia formation associated with renal tumorigenesis.
Lin CJ, Dang A, Hernandez E, Hsieh JT
Neoplasia (New York, N.Y.) 2021 Jan;23(1):169-180
Neoplasia (New York, N.Y.) 2021 Jan;23(1):169-180
Nonredundant roles of DIAPHs in primary ciliogenesis.
Palander O, Lam A, Collins RF, Moraes TJ, Trimble WS
The Journal of biological chemistry 2021 Jan-Jun;296:100680
The Journal of biological chemistry 2021 Jan-Jun;296:100680
BBS Proteins Affect Ciliogenesis and Are Essential for Hedgehog Signaling, but Not for Formation of iPSC-Derived RPE-65 Expressing RPE-Like Cells.
Hey CAB, Larsen LJ, Tümer Z, Brøndum-Nielsen K, Grønskov K, Hjortshøj TD, Møller LB
International journal of molecular sciences 2021 Jan 29;22(3)
International journal of molecular sciences 2021 Jan 29;22(3)
LPA signaling acts as a cell-extrinsic mechanism to initiate cilia disassembly and promote neurogenesis.
Hu HB, Song ZQ, Song GP, Li S, Tu HQ, Wu M, Zhang YC, Yuan JF, Li TT, Li PY, Xu YL, Shen XL, Han QY, Li AL, Zhou T, Chun J, Zhang XM, Li HY
Nature communications 2021 Jan 28;12(1):662
Nature communications 2021 Jan 28;12(1):662
Human cells lacking CDC14A and CDC14B show differences in ciliogenesis but not in mitotic progression.
Partscht P, Uddin B, Schiebel E
Journal of cell science 2021 Jan 27;134(2)
Journal of cell science 2021 Jan 27;134(2)
Application of green tea catechins, polysaccharides, and flavonol prevent fine dust induced bronchial damage by modulating inflammation and airway cilia.
Kim J, Choi H, Choi DH, Park K, Kim HJ, Park M
Scientific reports 2021 Jan 26;11(1):2232
Scientific reports 2021 Jan 26;11(1):2232
Interaction of INPP5E with ARL13B is essential for its ciliary membrane retention but dispensable for its ciliary entry.
Qiu H, Fujisawa S, Nozaki S, Katoh Y, Nakayama K
Biology open 2021 Jan 25;10(1)
Biology open 2021 Jan 25;10(1)
EFA6A, an exchange factor for Arf6, regulates early steps in ciliogenesis.
Partisani M, Baron CL, Ghossoub R, Fayad R, Pagnotta S, Abélanet S, Macia E, Brau F, Lacas-Gervais S, Benmerah A, Luton F, Franco M
Journal of cell science 2021 Jan 22;134(2)
Journal of cell science 2021 Jan 22;134(2)
Rabl2 GTP hydrolysis licenses BBSome-mediated export to fine-tune ciliary signaling.
Duan S, Li H, Zhang Y, Yang S, Chen Y, Qiu B, Huang C, Wang J, Li J, Zhu X, Yan X
The EMBO journal 2021 Jan 15;40(2):e105499
The EMBO journal 2021 Jan 15;40(2):e105499
Cooperation of the IFT-A complex with the IFT-B complex is required for ciliary retrograde protein trafficking and GPCR import.
Kobayashi T, Ishida Y, Hirano T, Katoh Y, Nakayama K
Molecular biology of the cell 2021 Jan 1;32(1):45-56
Molecular biology of the cell 2021 Jan 1;32(1):45-56
Phosphosite T674A mutation in kinesin family member 3A fails to reproduce tissue and ciliary defects characteristic of CILK1 loss of function.
Gailey CD, Wang EJ, Jin L, Ahmadi S, Brautigan DL, Li X, Xu W, Scott MM, Fu Z
Developmental dynamics : an official publication of the American Association of Anatomists 2021 Feb;250(2):263-273
Developmental dynamics : an official publication of the American Association of Anatomists 2021 Feb;250(2):263-273
Functional inhibition of fatty acid binding protein 4 ameliorates impaired ciliogenesis in GCs.
Jung Y, Cho SM, Kim S, Cheong JH, Kwon HJ
Biochemical and biophysical research communications 2021 Feb 5;539:28-33
Biochemical and biophysical research communications 2021 Feb 5;539:28-33
Loss of primary cilia promotes mitochondria-dependent apoptosis in thyroid cancer.
Lee J, Park KC, Sul HJ, Hong HJ, Kim KH, Kero J, Shong M
Scientific reports 2021 Feb 18;11(1):4181
Scientific reports 2021 Feb 18;11(1):4181
Expression of α-Tubulin Acetyltransferase 1 and Tubulin Acetylation as Selective Forces in Cell Competition.
Rahimi AM, Cai M, Kılıҫ I, Kazerouni ZB, Tapia Contreras C, Hoyer-Fender S
Cells 2021 Feb 14;10(2)
Cells 2021 Feb 14;10(2)
CEP55 promotes cilia disassembly through stabilizing Aurora A kinase.
Zhang YC, Bai YF, Yuan JF, Shen XL, Xu YL, Jian XX, Li S, Song ZQ, Hu HB, Li PY, Tu HQ, Han QY, Wang N, Li AL, Zhang XM, Wu M, Zhou T, Li HY
The Journal of cell biology 2021 Feb 1;220(2)
The Journal of cell biology 2021 Feb 1;220(2)
YAP/TAZ maintain the proliferative capacity and structural organization of radial glial cells during brain development.
Lavado A, Gangwar R, Paré J, Wan S, Fan Y, Cao X
Developmental biology 2021 Dec;480:39-49
Developmental biology 2021 Dec;480:39-49
The 3-beta-hydroxysteroid-Delta(8), Delta(7)-isomerase EBP inhibits cholesterylation of Smoothened.
Qiu ZP, Hu A, Song BL
Biochimica et biophysica acta. Molecular and cell biology of lipids 2021 Dec;1866(12):159041
Biochimica et biophysica acta. Molecular and cell biology of lipids 2021 Dec;1866(12):159041
Cisplatin induces lung cell cilia disruption and lung damage via oxidative stress.
Han YK, Kim JS, Jang G, Park KM
Free radical biology & medicine 2021 Dec;177:270-277
Free radical biology & medicine 2021 Dec;177:270-277
Quantitative Determination of Primary Cilia Protein Distribution Using Immunofluorescence Staining and MATLAB Analysis.
Jenks AD, Fivaz M, Tanos BE
Bio-protocol 2021 Dec 5;11(23):e4248
Bio-protocol 2021 Dec 5;11(23):e4248
Loss of Planar Cell Polarity Effector Fuzzy Causes Renal Hypoplasia by Disrupting Several Signaling Pathways.
Wang IY, Chung CF, Babayeva S, Sogomonian T, Torban E
Journal of developmental biology 2021 Dec 23;10(1)
Journal of developmental biology 2021 Dec 23;10(1)
Mitral Valve Prolapse Induces Regionalized Myocardial Fibrosis.
Morningstar JE, Gensemer C, Moore R, Fulmer D, Beck TC, Wang C, Moore K, Guo L, Sieg F, Nagata Y, Bertrand P, Spampinato RA, Glover J, Poelzing S, Gourdie RG, Watts K, Richardson WJ, Levine RA, Borger MA, Norris RA
Journal of the American Heart Association 2021 Dec 21;10(24):e022332
Journal of the American Heart Association 2021 Dec 21;10(24):e022332
Endoderm development requires centrioles to restrain p53-mediated apoptosis in the absence of ERK activity.
Xie C, Abrams SR, Herranz-Pérez V, García-Verdugo JM, Reiter JF
Developmental cell 2021 Dec 20;56(24):3334-3348.e6
Developmental cell 2021 Dec 20;56(24):3334-3348.e6
Exocyst complex mediates recycling of internal cilia.
Rivera-Molina FE, Xi Z, Reales E, Wang B, Toomre D
Current biology : CB 2021 Dec 20;31(24):5580-5589.e5
Current biology : CB 2021 Dec 20;31(24):5580-5589.e5
Biallelic deep intronic variant c.5457+81T>A in TRIP11 causes loss of function and results in achondrogenesis 1A.
Upadhyai P, Radhakrishnan P, Guleria VS, Kausthubham N, Nayak SS, Superti-Furga A, Girisha KM
Human mutation 2021 Aug;42(8):1005-1014
Human mutation 2021 Aug;42(8):1005-1014
Identification and characterization of primary cilia-positive salivary gland tumours exhibiting basaloid/myoepithelial differentiation.
Shinmura K, Kusafuka K, Kawasaki H, Kato H, Hariyama T, Tsuchiya K, Kawanishi Y, Funai K, Misawa K, Mineta H, Sugimura H
The Journal of pathology 2021 Aug;254(5):519-530
The Journal of pathology 2021 Aug;254(5):519-530
Differential Roles of Tubby Family Proteins in Ciliary Formation and Trafficking.
Hong JJ, Kim KE, Park SY, Bok J, Seo JT, Moon SJ
Molecules and cells 2021 Aug 31;44(8):591-601
Molecules and cells 2021 Aug 31;44(8):591-601
Impaired Ciliary Beat Frequency and Ciliogenesis Alteration during Airway Epithelial Cell Differentiation in COPD.
Ancel J, Belgacemi R, Diabasana Z, Perotin JM, Bonnomet A, Dewolf M, Launois C, Mulette P, Deslée G, Polette M, Dormoy V
Diagnostics (Basel, Switzerland) 2021 Aug 31;11(9)
Diagnostics (Basel, Switzerland) 2021 Aug 31;11(9)
FBF1 deficiency promotes beiging and healthy expansion of white adipose tissue.
Zhang Y, Hao J, Tarrago MG, Warner GM, Giorgadze N, Wei Q, Huang Y, He K, Chen C, Peclat TR, White TA, Ling K, Tchkonia T, Kirkland JL, Chini EN, Hu J
Cell reports 2021 Aug 3;36(5):109481
Cell reports 2021 Aug 3;36(5):109481
Loss of the ciliary protein Chibby1 in mice leads to exocrine pancreatic degeneration and pancreatitis.
Cyge B, Voronina V, Hoque M, Kim EN, Hall J, Bailey-Lundberg JM, Pazour GJ, Crawford HC, Moon RT, Li FQ, Takemaru KI
Scientific reports 2021 Aug 26;11(1):17220
Scientific reports 2021 Aug 26;11(1):17220
A polycystin-2 protein with modified channel properties leads to an increased diameter of renal tubules and to renal cysts.
Grosch M, Brunner K, Ilyaskin AV, Schober M, Staudner T, Schmied D, Stumpp T, Schmidt KN, Madej MG, Pessoa TD, Othmen H, Kubitza M, Osten L, de Vries U, Mair MM, Somlo S, Moser M, Kunzelmann K, Ziegler C, Haerteis S, Korbmacher C, Witzgall R
Journal of cell science 2021 Aug 15;134(16)
Journal of cell science 2021 Aug 15;134(16)
Targeted Ablation of Primary Cilia in Differentiated Dopaminergic Neurons Reduces Striatal Dopamine and Responsiveness to Metabolic Stress.
Mustafa R, Rawas C, Mannal N, Kreiner G, Spittau B, Kamińska K, Yilmaz R, Pötschke C, Kirsch J, Liss B, Tucker KL, Parlato R
Antioxidants (Basel, Switzerland) 2021 Aug 13;10(8)
Antioxidants (Basel, Switzerland) 2021 Aug 13;10(8)
Multifaceted Functions of Rab23 on Primary Cilium-Mediated and Hedgehog Signaling-Mediated Cerebellar Granule Cell Proliferation.
Hor CHH, Lo JCW, Cham ALS, Leong WY, Goh ELK
The Journal of neuroscience : the official journal of the Society for Neuroscience 2021 Aug 11;41(32):6850-6863
The Journal of neuroscience : the official journal of the Society for Neuroscience 2021 Aug 11;41(32):6850-6863
Xpo7 negatively regulates Hedgehog signaling by exporting Gli2 from the nucleus.
Markiewicz Ł, Uśpieński T, Baran B, Niedziółka SM, Niewiadomski P
Cellular signalling 2021 Apr;80:109907
Cellular signalling 2021 Apr;80:109907
A CRISPR and high-content imaging assay compliant with ACMG/AMP guidelines for clinical variant interpretation in ciliopathies.
Nazlamova L, Thomas NS, Cheung MK, Legebeke J, Lord J, Pengelly RJ, Tapper WJ, Wheway G
Human genetics 2021 Apr;140(4):593-607
Human genetics 2021 Apr;140(4):593-607
A mouse model of BBS identifies developmental and homeostatic effects of BBS5 mutation and identifies novel pituitary abnormalities.
Bentley-Ford MR, Engle SE, Clearman KR, Haycraft CJ, Andersen RS, Croyle MJ, Rains AB, Berbari NF, Yoder BK
Human molecular genetics 2021 Apr 26;30(3-4):234-246
Human molecular genetics 2021 Apr 26;30(3-4):234-246
WDR35 is involved in subcellular localization of acetylated tubulin in 293T cells.
Sekiguchi T, Ishii T, Kobayashi H, Furuno N
Biochemical and biophysical research communications 2021 Apr 2;547:169-175
Biochemical and biophysical research communications 2021 Apr 2;547:169-175
Roles for ELMOD2 and Rootletin in ciliogenesis.
Turn RE, Linnert J, Gigante ED, Wolfrum U, Caspary T, Kahn RA
Molecular biology of the cell 2021 Apr 15;32(8):800-822
Molecular biology of the cell 2021 Apr 15;32(8):800-822
Rpgrip1l controls ciliary gating by ensuring the proper amount of Cep290 at the vertebrate transition zone.
Wiegering A, Dildrop R, Vesque C, Khanna H, Schneider-Maunoury S, Gerhardt C
Molecular biology of the cell 2021 Apr 15;32(8):675-689
Molecular biology of the cell 2021 Apr 15;32(8):675-689
A ZFYVE19 gene mutation associated with neonatal cholestasis and cilia dysfunction: case report with a novel pathogenic variant.
Mandato C, Siano MA, Nazzaro L, Gelzo M, Francalanci P, Rizzo F, D'Agostino Y, Morleo M, Brillante S, Weisz A, Franco B, Vajro P
Orphanet journal of rare diseases 2021 Apr 14;16(1):179
Orphanet journal of rare diseases 2021 Apr 14;16(1):179
HDAC6 Signaling at Primary Cilia Promotes Proliferation and Restricts Differentiation of Glioma Cells.
Shi P, Hoang-Minh LB, Tian J, Cheng A, Basrai R, Kalaria N, Lebowitz JJ, Khoshbouei H, Deleyrolle LP, Sarkisian MR
Cancers 2021 Apr 1;13(7)
Cancers 2021 Apr 1;13(7)
Characterization of primary cilia features reveal cell-type specific variability in in vitro models of osteogenic and chondrogenic differentiation.
Upadhyai P, Guleria VS, Udupa P
PeerJ 2020;8:e9799
PeerJ 2020;8:e9799
FOP Negatively Regulates Ciliogenesis and Promotes Cell Cycle Re-entry by Facilitating Primary Cilia Disassembly.
Jiang H, Liu S, Cheung MH, Amin A, Liang C
Frontiers in cell and developmental biology 2020;8:590449
Frontiers in cell and developmental biology 2020;8:590449
Effect of Cell Spreading on Rosette Formation by Human Pluripotent Stem Cell-Derived Neural Progenitor Cells.
Townshend RF, Shao Y, Wang S, Cortez CL, Esfahani SN, Spence JR, O'Shea KS, Fu J, Gumucio DL, Taniguchi K
Frontiers in cell and developmental biology 2020;8:588941
Frontiers in cell and developmental biology 2020;8:588941
CEP164 Deficiency Causes Hyperproliferation of Pancreatic Cancer Cells.
Kobayashi T, Tanaka K, Mashima Y, Shoda A, Tokuda M, Itoh H
Frontiers in cell and developmental biology 2020;8:587691
Frontiers in cell and developmental biology 2020;8:587691
Distinct Roles of TRAPPC8 and TRAPPC12 in Ciliogenesis via Their Interactions With OFD1.
Zhang C, Li C, Siu GKY, Luo X, Yu S
Frontiers in cell and developmental biology 2020;8:148
Frontiers in cell and developmental biology 2020;8:148
Patient-Specific Retinal Organoids Recapitulate Disease Features of Late-Onset Retinitis Pigmentosa.
Gao ML, Lei XL, Han F, He KW, Jin SQ, Zhang YY, Jin ZB
Frontiers in cell and developmental biology 2020;8:128
Frontiers in cell and developmental biology 2020;8:128
Analysis of Caveolin in Primary Cilia.
Morthorst SK, Mogensen JB, Christensen ST, Pedersen LB
Methods in molecular biology (Clifton, N.J.) 2020;2169:27-41
Methods in molecular biology (Clifton, N.J.) 2020;2169:27-41
A Shh/Gli-driven three-node timer motif controls temporal identity and fate of neural stem cells.
Dias JM, Alekseenko Z, Jeggari A, Boareto M, Vollmer J, Kozhevnikova M, Wang H, Matise MP, Alexeyenko A, Iber D, Ericson J
Science advances 2020 Sep;6(38)
Science advances 2020 Sep;6(38)
Loss of polycystins suppresses deciliation via the activation of the centrosomal integrity pathway.
Gerakopoulos V, Ngo P, Tsiokas L
Life science alliance 2020 Sep;3(9)
Life science alliance 2020 Sep;3(9)
Loss of Cilia Does Not Slow Liver Disease Progression in Mouse Models of Autosomal Recessive Polycystic Kidney Disease.
Gallagher AR, Somlo S
Kidney360 2020 Sep 24;1(9):962-968
Kidney360 2020 Sep 24;1(9):962-968
Live imaging of hair bundle polarity acquisition demonstrates a critical timeline for transcription factor Emx2.
Tona Y, Wu DK
eLife 2020 Sep 23;9
eLife 2020 Sep 23;9
Cell Types of the Human Retina and Its Organoids at Single-Cell Resolution.
Cowan CS, Renner M, De Gennaro M, Gross-Scherf B, Goldblum D, Hou Y, Munz M, Rodrigues TM, Krol J, Szikra T, Cuttat R, Waldt A, Papasaikas P, Diggelmann R, Patino-Alvarez CP, Galliker P, Spirig SE, Pavlinic D, Gerber-Hollbach N, Schuierer S, Srdanovic A, Balogh M, Panero R, Kusnyerik A, Szabo A, Stadler MB, Orgül S, Picelli S, Hasler PW, Hierlemann A, Scholl HPN, Roma G, Nigsch F, Roska B
Cell 2020 Sep 17;182(6):1623-1640.e34
Cell 2020 Sep 17;182(6):1623-1640.e34
Mosaic Analysis with Double Markers reveals IGF1R function in granule cell progenitors during cerebellar development.
Terry TT, Cheng T, Mahjoub M, Zong H
Developmental biology 2020 Sep 15;465(2):130-143
Developmental biology 2020 Sep 15;465(2):130-143
Protein phosphatase 1 regulatory subunit 35 is required for ciliogenesis, notochord morphogenesis, and cell-cycle progression during murine development.
Archambault D, Cheong A, Iverson E, Tremblay KD, Mager J
Developmental biology 2020 Sep 1;465(1):1-10
Developmental biology 2020 Sep 1;465(1):1-10
Primary cilia mediate parathyroid hormone receptor type 1 osteogenic actions in osteocytes and osteoblasts via Gli activation.
Martín-Guerrero E, Tirado-Cabrera I, Buendía I, Alonso V, Gortázar AR, Ardura JA
Journal of cellular physiology 2020 Oct;235(10):7356-7369
Journal of cellular physiology 2020 Oct;235(10):7356-7369
Mutations in the exocyst component EXOC2 cause severe defects in human brain development.
Van Bergen NJ, Ahmed SM, Collins F, Cowley M, Vetro A, Dale RC, Hock DH, de Caestecker C, Menezes M, Massey S, Ho G, Pisano T, Glover S, Gusman J, Stroud DA, Dinger M, Guerrini R, Macara IG, Christodoulou J
The Journal of experimental medicine 2020 Oct 5;217(10)
The Journal of experimental medicine 2020 Oct 5;217(10)
A novel hypomorphic allele of Spag17 causes primary ciliary dyskinesia phenotypes in mice.
Abdelhamed Z, Lukacs M, Cindric S, Ali S, Omran H, Stottmann RW
Disease models & mechanisms 2020 Oct 30;13(10)
Disease models & mechanisms 2020 Oct 30;13(10)
Comparative Super-Resolution Mapping of Basal Feet Reveals a Modular but Distinct Architecture in Primary and Motile Cilia.
Nguyen QPH, Liu Z, Albulescu A, Ouyang H, Zlock L, Coyaud E, Laurent E, Finkbeiner W, Moraes TJ, Raught B, Mennella V
Developmental cell 2020 Oct 26;55(2):209-223.e7
Developmental cell 2020 Oct 26;55(2):209-223.e7
Roles of TOG and jelly-roll domains of centrosomal protein CEP104 in its functions in cilium elongation and Hedgehog signaling.
Yamazoe T, Nagai T, Umeda S, Sugaya Y, Mizuno K
The Journal of biological chemistry 2020 Oct 23;295(43):14723-14736
The Journal of biological chemistry 2020 Oct 23;295(43):14723-14736
Use of patient derived urine renal epithelial cells to confirm pathogenicity of PKHD1 alleles.
Molinari E, Srivastava S, Dewhurst RM, Sayer JA
BMC nephrology 2020 Oct 15;21(1):435
BMC nephrology 2020 Oct 15;21(1):435
Differentiation of ciliated human midbrain-derived LUHMES neurons.
Lauter G, Coschiera A, Yoshihara M, Sugiaman-Trapman D, Ezer S, Sethurathinam S, Katayama S, Kere J, Swoboda P
Journal of cell science 2020 Nov 9;133(21)
Journal of cell science 2020 Nov 9;133(21)
Centrosome and ciliary abnormalities in fetal akinesia deformation sequence human fibroblasts.
Jühlen R, Martinelli V, Vinci C, Breckpot J, Fahrenkrog B
Scientific reports 2020 Nov 9;10(1):19301
Scientific reports 2020 Nov 9;10(1):19301
Mutations in GRK2 cause Jeune syndrome by impairing Hedgehog and canonical Wnt signaling.
Bosakova M, Abraham SP, Nita A, Hruba E, Buchtova M, Taylor SP, Duran I, Martin J, Svozilova K, Barta T, Varecha M, Balek L, Kohoutek J, Radaszkiewicz T, Pusapati GV, Bryja V, Rush ET, Thiffault I, Nickerson DA, Bamshad MJ, University of Washington Center for Mendelian Genomics, Rohatgi R, Cohn DH, Krakow D, Krejci P
EMBO molecular medicine 2020 Nov 6;12(11):e11739
EMBO molecular medicine 2020 Nov 6;12(11):e11739
LRP2 controls sonic hedgehog-dependent differentiation of cardiac progenitor cells during outflow tract formation.
Christ A, Marczenke M, Willnow TE
Human molecular genetics 2020 Nov 25;29(19):3183-3196
Human molecular genetics 2020 Nov 25;29(19):3183-3196
Primary cilia mediate early life programming of adiposity through lysosomal regulation in the developing mouse hypothalamus.
Lee CH, Song DK, Park CB, Choi J, Kang GM, Shin SH, Kwon I, Park S, Kim S, Kim JY, Dugu H, Park JW, Choi JH, Min SH, Sohn JW, Kim MS
Nature communications 2020 Nov 13;11(1):5772
Nature communications 2020 Nov 13;11(1):5772
Primary cilium remodeling mediates a cell signaling switch in differentiating neurons.
Toro-Tapia G, Das RM
Science advances 2020 May;6(21):eabb0601
Science advances 2020 May;6(21):eabb0601
Pannexin-1 mediates fluid shear stress-sensitive purinergic signaling and cyst growth in polycystic kidney disease.
Verschuren EHJ, Rigalli JP, Castenmiller C, Rohrbach MU, Bindels RJM, Peters DJM, Arjona FJ, Hoenderop JGJ
FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2020 May;34(5):6382-6398
FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2020 May;34(5):6382-6398
Genetic interaction of mammalian IFT-A paralogs regulates cilia disassembly, ciliary entry of membrane protein, Hedgehog signaling, and embryogenesis.
Wang W, Allard BA, Pottorf TS, Wang HH, Vivian JL, Tran PV
FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2020 May;34(5):6369-6381
FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2020 May;34(5):6369-6381
ECE2 regulates neurogenesis and neuronal migration during human cortical development.
Buchsbaum IY, Kielkowski P, Giorgio G, O'Neill AC, Di Giaimo R, Kyrousi C, Khattak S, Sieber SA, Robertson SP, Cappello S
EMBO reports 2020 May 6;21(5):e48204
EMBO reports 2020 May 6;21(5):e48204
TALPID3 and ANKRD26 selectively orchestrate FBF1 localization and cilia gating.
Yan H, Chen C, Chen H, Hong H, Huang Y, Ling K, Hu J, Wei Q
Nature communications 2020 May 4;11(1):2196
Nature communications 2020 May 4;11(1):2196
Profilin1-Dependent F-Actin Assembly Controls Division of Apical Radial Glia and Neocortex Development.
Kullmann JA, Meyer S, Pipicelli F, Kyrousi C, Schneider F, Bartels N, Cappello S, Rust MB
Cerebral cortex (New York, N.Y. : 1991) 2020 May 18;30(6):3467-3482
Cerebral cortex (New York, N.Y. : 1991) 2020 May 18;30(6):3467-3482
Prominins control ciliary length throughout the animal kingdom: New lessons from human prominin-1 and zebrafish prominin-3.
Jászai J, Thamm K, Karbanová J, Janich P, Fargeas CA, Huttner WB, Corbeil D
The Journal of biological chemistry 2020 May 1;295(18):6007-6022
The Journal of biological chemistry 2020 May 1;295(18):6007-6022
ARL13B regulates Sonic hedgehog signaling from outside primary cilia.
Gigante ED, Taylor MR, Ivanova AA, Kahn RA, Caspary T
eLife 2020 Mar 4;9
eLife 2020 Mar 4;9
Conditional switching of KIF2A mutation provides new insights into cortical malformation pathogeny.
Gilet JG, Ivanova EL, Trofimova D, Rudolf G, Meziane H, Broix L, Drouot N, Courraud J, Skory V, Voulleminot P, Osipenko M, Bahi-Buisson N, Yalcin B, Birling MC, Hinckelmann MV, Kwok BH, Allingham JS, Chelly J
Human molecular genetics 2020 Mar 27;29(5):766-784
Human molecular genetics 2020 Mar 27;29(5):766-784
Nek2 kinase displaces distal appendages from the mother centriole prior to mitosis.
Viol L, Hata S, Pastor-Peidro A, Neuner A, Murke F, Wuchter P, Ho AD, Giebel B, Pereira G
The Journal of cell biology 2020 Mar 2;219(3)
The Journal of cell biology 2020 Mar 2;219(3)
Functional Alterations in Ciliogenesis-Associated Kinase 1 (CILK1) that Result from Mutations Linked to Juvenile Myoclonic Epilepsy.
Wang EJ, Gailey CD, Brautigan DL, Fu Z
Cells 2020 Mar 12;9(3)
Cells 2020 Mar 12;9(3)
Primary cilia mediate Klf2-dependant Notch activation in regenerating heart.
Li X, Lu Q, Peng Y, Geng F, Shao X, Zhou H, Cao Y, Zhang R
Protein & cell 2020 Jun;11(6):433-445
Protein & cell 2020 Jun;11(6):433-445
A novel CDK-independent function of p27(Kip1) in preciliary vesicle trafficking during ciliogenesis.
Yukimoto H, Miyamoto T, Kiyono T, Wang S, Matsuura S, Mizoguchi A, Katayama N, Inagaki M, Kasahara K
Biochemical and biophysical research communications 2020 Jun 30;527(3):716-722
Biochemical and biophysical research communications 2020 Jun 30;527(3):716-722
Epb41l5 interacts with Iqcb1 and regulates ciliary function in zebrafish embryos.
Yu T, Matsuda M
Journal of cell science 2020 Jun 28;133(12)
Journal of cell science 2020 Jun 28;133(12)
Nanobody-directed targeting of optogenetic tools to study signaling in the primary cilium.
Hansen JN, Kaiser F, Klausen C, Stüven B, Chong R, Bönigk W, Mick DU, Möglich A, Jurisch-Yaksi N, Schmidt FI, Wachten D
eLife 2020 Jun 24;9
eLife 2020 Jun 24;9
Loss of CPAP in developing mouse brain and its functional implication for human primary microcephaly.
Lin YN, Lee YS, Li SK, Tang TK
Journal of cell science 2020 Jun 24;133(12)
Journal of cell science 2020 Jun 24;133(12)
mTOR and S6K1 drive polycystic kidney by the control of Afadin-dependent oriented cell division.
Bonucci M, Kuperwasser N, Barbe S, Koka V, de Villeneuve D, Zhang C, Srivastava N, Jia X, Stokes MP, Bienaimé F, Verkarre V, Lopez JB, Jaulin F, Pontoglio M, Terzi F, Delaval B, Piel M, Pende M
Nature communications 2020 Jun 24;11(1):3200
Nature communications 2020 Jun 24;11(1):3200
CCDC61/VFL3 Is a Paralog of SAS6 and Promotes Ciliary Functions.
Ochi T, Quarantotti V, Lin H, Jullien J, Rosa E Silva I, Boselli F, Barnabas DD, Johnson CM, McLaughlin SH, Freund SMV, Blackford AN, Kimata Y, Goldstein RE, Jackson SP, Blundell TL, Dutcher SK, Gergely F, van Breugel M
Structure (London, England : 1993) 2020 Jun 2;28(6):674-689.e11
Structure (London, England : 1993) 2020 Jun 2;28(6):674-689.e11
Insufficiency of ciliary cholesterol in hereditary Zellweger syndrome.
Miyamoto T, Hosoba K, Itabashi T, Iwane AH, Akutsu SN, Ochiai H, Saito Y, Yamamoto T, Matsuura S
The EMBO journal 2020 Jun 17;39(12):e103499
The EMBO journal 2020 Jun 17;39(12):e103499
Robust classification of cell cycle phase and biological feature extraction by image-based deep learning.
Nagao Y, Sakamoto M, Chinen T, Okada Y, Takao D
Molecular biology of the cell 2020 Jun 15;31(13):1346-1354
Molecular biology of the cell 2020 Jun 15;31(13):1346-1354
Intracellular calcium response of primary cilia of tubular cells to modulated shear stress under oxidative stress.
Chikamori M, Kimura H, Inagi R, Zhou J, Nangaku M, Fujii T
Biomicrofluidics 2020 Jul;14(4):044102
Biomicrofluidics 2020 Jul;14(4):044102
Ciliary proteins specify the cell inflammatory response by tuning NFκB signalling, independently of primary cilia.
Mc Fie M, Koneva L, Collins I, Coveney CR, Clube AM, Chanalaris A, Vincent TL, Bezbradica JS, Sansom SN, Wann AKT
Journal of cell science 2020 Jul 8;133(13)
Journal of cell science 2020 Jul 8;133(13)
Ubiquitin links smoothened to intraflagellar transport to regulate Hedgehog signaling.
Desai PB, Stuck MW, Lv B, Pazour GJ
The Journal of cell biology 2020 Jul 6;219(7)
The Journal of cell biology 2020 Jul 6;219(7)
Ciliogenesis and Hedgehog signalling are suppressed downstream of KRAS during acinar-ductal metaplasia in mouse.
Bangs FK, Miller P, O'Neill E
Disease models & mechanisms 2020 Jul 30;13(7)
Disease models & mechanisms 2020 Jul 30;13(7)
Meningioma cells express primary cilia but do not transduce ciliary Hedgehog signals.
Findakly S, Choudhury A, Daggubati V, Pekmezci M, Lang UE, Raleigh DR
Acta neuropathologica communications 2020 Jul 20;8(1):114
Acta neuropathologica communications 2020 Jul 20;8(1):114
Transient Primary Cilia Mediate Robust Hedgehog Pathway-Dependent Cell Cycle Control.
Ho EK, Tsai AE, Stearns T
Current biology : CB 2020 Jul 20;30(14):2829-2835.e5
Current biology : CB 2020 Jul 20;30(14):2829-2835.e5
Desert hedgehog-primary cilia cross talk shapes mitral valve tissue by organizing smooth muscle actin.
Fulmer D, Toomer KA, Glover J, Guo L, Moore K, Moore R, Stairley R, Gensemer C, Abrol S, Rumph MK, Emetu F, Lipschutz JH, McDowell C, Bian J, Wang C, Beck T, Wessels A, Renault MA, Norris RA
Developmental biology 2020 Jul 1;463(1):26-38
Developmental biology 2020 Jul 1;463(1):26-38
Airway epithelial cell differentiation relies on deficient Hedgehog signalling in COPD.
Belgacemi R, Luczka E, Ancel J, Diabasana Z, Perotin JM, Germain A, Lalun N, Birembaut P, Dubernard X, Mérol JC, Delepine G, Polette M, Deslée G, Dormoy V
EBioMedicine 2020 Jan;51:102572
EBioMedicine 2020 Jan;51:102572
Intraflagellar-transport A dysfunction causes hyperphagia-induced systemic insulin resistance in a pre-obese state.
Jacobs DT, Allard BA, Pottorf TS, Silva LM, Wang W, Al-Naamani A, Agborbesong E, Wang T, Carr DA, Tran PV
FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2020 Jan;34(1):148-160
FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2020 Jan;34(1):148-160
ESCRT subunit CHMP4B localizes to primary cilia and is required for the structural integrity of the ciliary membrane.
Jung E, Choi TI, Lee JE, Kim CH, Kim J
FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2020 Jan;34(1):1331-1344
FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2020 Jan;34(1):1331-1344
CENPV Is a CYLD-Interacting Molecule Regulating Ciliary Acetylated α-Tubulin.
Chiticariu E, Regamey A, Huber M, Hohl D
The Journal of investigative dermatology 2020 Jan;140(1):66-74.e4
The Journal of investigative dermatology 2020 Jan;140(1):66-74.e4
hVFL3/CCDC61 is a component of mother centriole subdistal appendages required for centrosome cohesion and positioning.
Pizon V, Gaudin N, Poteau M, Cifuentes-Diaz C, Demdou R, Heyer V, Reina San Martin B, Azimzadeh J
Biology of the cell 2020 Jan;112(1):22-37
Biology of the cell 2020 Jan;112(1):22-37
The novel testicular enrichment protein Cfap58 is required for Notch-associated ciliogenesis.
Li ZZ, Zhao WL, Wang GS, Gu NH, Sun F
Bioscience reports 2020 Jan 31;40(1)
Bioscience reports 2020 Jan 31;40(1)
MetAP2 inhibition reduces food intake and body weight in a ciliopathy mouse model of obesity.
Pottorf TS, Fagan MP, Burkey BF, Cho DJ, Vath JE, Tran PV
JCI insight 2020 Jan 30;5(2)
JCI insight 2020 Jan 30;5(2)
The association of microcephaly protein WDR62 with CPAP/IFT88 is required for cilia formation and neocortical development.
Shohayeb B, Ho U, Yeap YY, Parton RG, Millard SS, Xu Z, Piper M, Ng DCH
Human molecular genetics 2020 Jan 15;29(2):248-263
Human molecular genetics 2020 Jan 15;29(2):248-263
PI3KC2α-dependent and VPS34-independent generation of PI3P controls primary cilium-mediated autophagy in response to shear stress.
Boukhalfa A, Nascimbeni AC, Ramel D, Dupont N, Hirsch E, Gayral S, Laffargue M, Codogno P, Morel E
Nature communications 2020 Jan 15;11(1):294
Nature communications 2020 Jan 15;11(1):294
TTBK2 and primary cilia are essential for the connectivity and survival of cerebellar Purkinje neurons.
Bowie E, Goetz SC
eLife 2020 Jan 14;9
eLife 2020 Jan 14;9
NRF2 negatively regulates primary ciliogenesis and hedgehog signaling.
Liu P, Dodson M, Fang D, Chapman E, Zhang DD
PLoS biology 2020 Feb;18(2):e3000620
PLoS biology 2020 Feb;18(2):e3000620
CEP41-mediated ciliary tubulin glutamylation drives angiogenesis through AURKA-dependent deciliation.
Ki SM, Kim JH, Won SY, Oh SJ, Lee IY, Bae YK, Chung KW, Choi BO, Park B, Choi EJ, Lee JE
EMBO reports 2020 Feb 5;21(2):e48290
EMBO reports 2020 Feb 5;21(2):e48290
Nubp2 is required for cranial neural crest survival in the mouse.
DiStasio A, Paulding D, Chaturvedi P, Stottmann RW
Developmental biology 2020 Feb 15;458(2):189-199
Developmental biology 2020 Feb 15;458(2):189-199
Altered gene regulation as a candidate mechanism by which ciliopathy gene SDCCAG8 contributes to schizophrenia and cognitive function.
Flynn M, Whitton L, Donohoe G, Morrison CG, Morris DW
Human molecular genetics 2020 Feb 1;29(3):407-417
Human molecular genetics 2020 Feb 1;29(3):407-417
DIAPH1 regulates ciliogenesis and trafficking in primary cilia.
Palander O, Trimble WS
FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2020 Dec;34(12):16516-16535
FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2020 Dec;34(12):16516-16535
Serotonin Receptor 2A Activation Promotes Evolutionarily Relevant Basal Progenitor Proliferation in the Developing Neocortex.
Xing L, Kalebic N, Namba T, Vaid S, Wimberger P, Huttner WB
Neuron 2020 Dec 23;108(6):1113-1129.e6
Neuron 2020 Dec 23;108(6):1113-1129.e6
Gpr37l1/prosaposin receptor regulates Ptch1 trafficking, Shh production, and cell proliferation in cerebellar primary astrocytes.
La Sala G, Di Pietro C, Matteoni R, Bolasco G, Marazziti D, Tocchini-Valentini GP
Journal of neuroscience research 2020 Dec 17;
Journal of neuroscience research 2020 Dec 17;
Sonic hedgehog signalling as a potential endobronchial biomarker in COPD.
Ancel J, Belgacemi R, Perotin JM, Diabasana Z, Dury S, Dewolf M, Bonnomet A, Lalun N, Birembaut P, Polette M, Deslée G, Dormoy V
Respiratory research 2020 Aug 7;21(1):207
Respiratory research 2020 Aug 7;21(1):207
The novel ciliogenesis regulator DYRK2 governs Hedgehog signaling during mouse embryogenesis.
Yoshida S, Aoki K, Fujiwara K, Nakakura T, Kawamura A, Yamada K, Ono M, Yogosawa S, Yoshida K
eLife 2020 Aug 6;9
eLife 2020 Aug 6;9
Polycystin-1 regulates ARHGAP35-dependent centrosomal RhoA activation and ROCK signaling.
Streets AJ, Prosseda PP, Ong AC
JCI insight 2020 Aug 20;5(16)
JCI insight 2020 Aug 20;5(16)
An interphase pool of KIF11 localizes at the basal bodies of primary cilia and a reduction in KIF11 expression alters cilia dynamics.
Zalenski AA, Majumder S, De K, Venere M
Scientific reports 2020 Aug 18;10(1):13946
Scientific reports 2020 Aug 18;10(1):13946
Primary Ciliary Deficits in the Dentate Gyrus of Fragile X Syndrome.
Lee B, Panda S, Lee HY
Stem cell reports 2020 Aug 11;15(2):454-466
Stem cell reports 2020 Aug 11;15(2):454-466
An IFT20 mechanotrafficking axis is required for integrin recycling, focal adhesion dynamics, and polarized cell migration.
Su S, Begum S, Ezratty EJ
Molecular biology of the cell 2020 Aug 1;31(17):1917-1930
Molecular biology of the cell 2020 Aug 1;31(17):1917-1930
A reduction of primary cilia but not hedgehog signaling disrupts morphogenesis in testicular organoids.
Goldsmith TM, Sakib S, Webster D, Carlson DF, Van der Hoorn F, Dobrinski I
Cell and tissue research 2020 Apr;380(1):191-200
Cell and tissue research 2020 Apr;380(1):191-200
Ptch2/Gas1 and Ptch1/Boc differentially regulate Hedgehog signalling in murine primordial germ cell migration.
Kim Y, Lee J, Seppala M, Cobourne MT, Kim SH
Nature communications 2020 Apr 24;11(1):1994
Nature communications 2020 Apr 24;11(1):1994
The HOPS complex subunit VPS39 controls ciliogenesis through autophagy.
Iaconis D, Crina C, Brillante S, Indrieri A, Morleo M, Franco B
Human molecular genetics 2020 Apr 15;29(6):1018-1029
Human molecular genetics 2020 Apr 15;29(6):1018-1029
The primary cilium dampens proliferative signaling and represses a G2/M transcriptional network in quiescent myoblasts.
Venugopal N, Ghosh A, Gala H, Aloysius A, Vyas N, Dhawan J
BMC molecular and cell biology 2020 Apr 15;21(1):25
BMC molecular and cell biology 2020 Apr 15;21(1):25
Roles for IFT172 and Primary Cilia in Cell Migration, Cell Division, and Neocortex Development.
Pruski M, Hu L, Yang C, Wang Y, Zhang JB, Zhang L, Huang Y, Rajnicek AM, St Clair D, McCaig CD, Lang B, Ding YQ
Frontiers in cell and developmental biology 2019;7:287
Frontiers in cell and developmental biology 2019;7:287
Ocular Ciliopathies: Genetic and Mechanistic Insights into Developing Therapies.
Shivanna M, Anand M, Chakrabarti S, Khanna H
Current medicinal chemistry 2019;26(17):3120-3131
Current medicinal chemistry 2019;26(17):3120-3131
Pharmacological cholesterol depletion disturbs ciliogenesis and ciliary function in developing zebrafish.
Maerz LD, Burkhalter MD, Schilpp C, Wittekindt OH, Frick M, Philipp M
Communications biology 2019;2:31
Communications biology 2019;2:31
Induction of Neural Crest Stem Cells From Bardet-Biedl Syndrome Patient Derived hiPSCs.
Barrell WB, Griffin JN, Harvey JL, HipSci Consortium, Danovi D, Beales P, Grigoriadis AE, Liu KJ
Frontiers in molecular neuroscience 2019;12:139
Frontiers in molecular neuroscience 2019;12:139
661W Photoreceptor Cell Line as a Cell Model for Studying Retinal Ciliopathies.
Wheway G, Nazlamova L, Turner D, Cross S
Frontiers in genetics 2019;10:308
Frontiers in genetics 2019;10:308
Centrosome-derived microtubule radial array, PCM-1 protein, and primary cilia formation.
Fokin Artem I, Zhapparova Olga N, Burakov Anton V, Nadezhdina Elena S
Protoplasma 2019 Sep;256(5):1361-1373
Protoplasma 2019 Sep;256(5):1361-1373
Patient-derived lung cancer organoids as in vitro cancer models for therapeutic screening.
Kim M, Mun H, Sung CO, Cho EJ, Jeon HJ, Chun SM, Jung DJ, Shin TH, Jeong GS, Kim DK, Choi EK, Jeong SY, Taylor AM, Jain S, Meyerson M, Jang SJ
Nature communications 2019 Sep 5;10(1):3991
Nature communications 2019 Sep 5;10(1):3991
Ahi1 promotes Arl13b ciliary recruitment, regulates Arl13b stability and is required for normal cell migration.
Muñoz-Estrada J, Ferland RJ
Journal of cell science 2019 Sep 4;132(17)
Journal of cell science 2019 Sep 4;132(17)
NRF2-dependent gene expression promotes ciliogenesis and Hedgehog signaling.
Martin-Hurtado A, Martin-Morales R, Robledinos-Antón N, Blanco R, Palacios-Blanco I, Lastres-Becker I, Cuadrado A, Garcia-Gonzalo FR
Scientific reports 2019 Sep 25;9(1):13896
Scientific reports 2019 Sep 25;9(1):13896
Requirement of IFT-B-BBSome complex interaction in export of GPR161 from cilia.
Nozaki S, Castro Araya RF, Katoh Y, Nakayama K
Biology open 2019 Sep 17;8(9)
Biology open 2019 Sep 17;8(9)
Neddylation inhibitor MLN4924 suppresses cilia formation by modulating AKT1.
Mao H, Tang Z, Li H, Sun B, Tan M, Fan S, Zhu Y, Sun Y
Protein & cell 2019 Oct;10(10):726-744
Protein & cell 2019 Oct;10(10):726-744
Planar Cell Polarity Effector Proteins Inturned and Fuzzy Form a Rab23 GEF Complex.
Gerondopoulos A, Strutt H, Stevenson NL, Sobajima T, Levine TP, Stephens DJ, Strutt D, Barr FA
Current biology : CB 2019 Oct 7;29(19):3323-3330.e8
Current biology : CB 2019 Oct 7;29(19):3323-3330.e8
Centrosomal and ciliary targeting of CCDC66 requires cooperative action of centriolar satellites, microtubules and molecular motors.
Conkar D, Bayraktar H, Firat-Karalar EN
Scientific reports 2019 Oct 3;9(1):14250
Scientific reports 2019 Oct 3;9(1):14250
Differential requirements for the EF-hand domains of human centrin 2 in primary ciliogenesis and nucleotide excision repair.
Khouj EM, Prosser SL, Tada H, Chong WM, Liao JC, Sugasawa K, Morrison CG
Journal of cell science 2019 Oct 3;132(19)
Journal of cell science 2019 Oct 3;132(19)
KCTD15 inhibits the Hedgehog pathway in Medulloblastoma cells by increasing protein levels of the oncosuppressor KCASH2.
Spiombi E, Angrisani A, Fonte S, De Feudis G, Fabretti F, Cucchi D, Izzo M, Infante P, Miele E, Po A, Di Magno L, Magliozzi R, Guardavaccaro D, Maroder M, Canettieri G, Giannini G, Ferretti E, Gulino A, Di Marcotullio L, Moretti M, De Smaele E
Oncogenesis 2019 Nov 4;8(11):64
Oncogenesis 2019 Nov 4;8(11):64
Omega-3 Fatty Acids Activate Ciliary FFAR4 to Control Adipogenesis.
Hilgendorf KI, Johnson CT, Mezger A, Rice SL, Norris AM, Demeter J, Greenleaf WJ, Reiter JF, Kopinke D, Jackson PK
Cell 2019 Nov 27;179(6):1289-1305.e21
Cell 2019 Nov 27;179(6):1289-1305.e21
microRNAs Regulating Human and Mouse Naïve Pluripotency.
Wang Y, Hussein AM, Somasundaram L, Sankar R, Detraux D, Mathieu J, Ruohola-Baker H
International journal of molecular sciences 2019 Nov 22;20(23)
International journal of molecular sciences 2019 Nov 22;20(23)
RAB8, RAB10 and RILPL1 contribute to both LRRK2 kinase-mediated centrosomal cohesion and ciliogenesis deficits.
Lara Ordónez AJ, Fernández B, Fdez E, Romo-Lozano M, Madero-Pérez J, Lobbestael E, Baekelandt V, Aiastui A, López de Munaín A, Melrose HL, Civiero L, Hilfiker S
Human molecular genetics 2019 Nov 1;28(21):3552-3568
Human molecular genetics 2019 Nov 1;28(21):3552-3568
A Sexually Dimorphic Role for STAT3 in Sonic Hedgehog Medulloblastoma.
White CL, Jayasekara WSN, Picard D, Chen J, Watkins DN, Cain JE, Remke M, Gough DJ
Cancers 2019 Nov 1;11(11)
Cancers 2019 Nov 1;11(11)
Primary Cilia Exhibit Mechanosensitivity to Cyclic Tensile Strain and Lineage-Dependent Expression in Adipose-Derived Stem Cells.
Bodle J, Hamouda MS, Cai S, Williams RB, Bernacki SH, Loboa EG
Scientific reports 2019 May 29;9(1):8009
Scientific reports 2019 May 29;9(1):8009
The ciliary GTPase Arl3 maintains tissue architecture by directing planar spindle orientation during epidermal morphogenesis.
Bhattarai SR, Begum S, Popow R, Ezratty EJ
Development (Cambridge, England) 2019 May 10;146(9)
Development (Cambridge, England) 2019 May 10;146(9)
Endosomal trafficking defects in patient cells with KIAA1109 biallelic variants.
Kane MS, Diamonstein CJ, Hauser N, Deeken JF, Niederhuber JE, Vilboux T
Genes & diseases 2019 Mar;6(1):56-67
Genes & diseases 2019 Mar;6(1):56-67
Indian Hedgehog links obesity to development of hepatocellular carcinoma.
Chong YC, Lim TE, Fu Y, Shin EM, Tergaonkar V, Han W
Oncogene 2019 Mar;38(12):2206-2222
Oncogene 2019 Mar;38(12):2206-2222
Fine particulate matter (PM2.5) inhibits ciliogenesis by increasing SPRR3 expression via c-Jun activation in RPE cells and skin keratinocytes.
Bae JE, Choi H, Shin DW, Na HW, Park NY, Kim JB, Jo DS, Cho MJ, Lyu JH, Chang JH, Lee EH, Lee TR, Kim HJ, Cho DH
Scientific reports 2019 Mar 8;9(1):3994
Scientific reports 2019 Mar 8;9(1):3994
Fibroblast growth factor receptor influences primary cilium length through an interaction with intestinal cell kinase.
Kunova Bosakova M, Nita A, Gregor T, Varecha M, Gudernova I, Fafilek B, Barta T, Basheer N, Abraham SP, Balek L, Tomanova M, Fialova Kucerova J, Bosak J, Potesil D, Zieba J, Song J, Konik P, Park S, Duran I, Zdrahal Z, Smajs D, Jansen G, Fu Z, Ko HW, Hampl A, Trantirek L, Krakow D, Krejci P
Proceedings of the National Academy of Sciences of the United States of America 2019 Mar 5;116(10):4316-4325
Proceedings of the National Academy of Sciences of the United States of America 2019 Mar 5;116(10):4316-4325
Homeostatic and tumourigenic activity of SOX2+ pituitary stem cells is controlled by the LATS/YAP/TAZ cascade.
Lodge EJ, Santambrogio A, Russell JP, Xekouki P, Jacques TS, Johnson RL, Thavaraj S, Bornstein SR, Andoniadou CL
eLife 2019 Mar 26;8
eLife 2019 Mar 26;8
HDAC1 and HDAC2 Regulate Intermediate Progenitor Positioning to Safeguard Neocortical Development.
Tang T, Zhang Y, Wang Y, Cai Z, Lu Z, Li L, Huang R, Hagelkruys A, Matthias P, Zhang H, Seiser C, Xie Y
Neuron 2019 Mar 20;101(6):1117-1133.e5
Neuron 2019 Mar 20;101(6):1117-1133.e5
Decreased Expression of Cilia Genes in Pancreatic Islets as a Risk Factor for Type 2 Diabetes in Mice and Humans.
Kluth O, Stadion M, Gottmann P, Aga H, Jähnert M, Scherneck S, Vogel H, Krus U, Seelig A, Ling C, Gerdes J, Schürmann A
Cell reports 2019 Mar 12;26(11):3027-3036.e3
Cell reports 2019 Mar 12;26(11):3027-3036.e3
The deubiquitinating enzyme Usp14 controls ciliogenesis and Hedgehog signaling.
Massa F, Tammaro R, Prado MA, Cesana M, Lee BH, Finley D, Franco B, Morleo M
Human molecular genetics 2019 Mar 1;28(5):764-777
Human molecular genetics 2019 Mar 1;28(5):764-777
Elevated miR-200a and miR-141 inhibit endocrine gland-derived vascular endothelial growth factor expression and ciliogenesis in preeclampsia.
Wang CY, Tsai PY, Chen TY, Tsai HL, Kuo PL, Su MT
The Journal of physiology 2019 Jun;597(12):3069-3083
The Journal of physiology 2019 Jun;597(12):3069-3083
Fbxo41 Promotes Disassembly of Neuronal Primary Cilia.
King CR, A A Quadros AR, Chazeau A, Saarloos I, van der Graaf AJ, Verhage M, Toonen RF
Scientific reports 2019 Jun 3;9(1):8179
Scientific reports 2019 Jun 3;9(1):8179
Silencing Y-box binding protein-1 inhibits triple-negative breast cancer cell invasiveness via regulation of MMP1 and beta-catenin expression.
Lim JP, Nair S, Shyamasundar S, Chua PJ, Muniasamy U, Matsumoto K, Gunaratne J, Bay BH
Cancer letters 2019 Jun 28;452:119-131
Cancer letters 2019 Jun 28;452:119-131
Extraciliary roles of the ciliopathy protein JBTS17 in mitosis and neurogenesis.
Hong H, Joo K, Park SM, Seo J, Kim MH, Shin E, Cheong HI, Lee JH, Kim J
Annals of neurology 2019 Jul;86(1):99-115
Annals of neurology 2019 Jul;86(1):99-115
Characterization of Functional Primary Cilia in Human Induced Pluripotent Stem Cell-Derived Neurons.
Miki D, Kobayashi Y, Okada T, Miyamoto T, Takei N, Sekino Y, Koganezawa N, Shirao T, Saito Y
Neurochemical research 2019 Jul;44(7):1736-1744
Neurochemical research 2019 Jul;44(7):1736-1744
Mechanical loading inhibits cartilage inflammatory signalling via an HDAC6 and IFT-dependent mechanism regulating primary cilia elongation.
Fu S, Thompson CL, Ali A, Wang W, Chapple JP, Mitchison HM, Beales PL, Wann AKT, Knight MM
Osteoarthritis and cartilage 2019 Jul;27(7):1064-1074
Osteoarthritis and cartilage 2019 Jul;27(7):1064-1074
Chloroquine inhibits human retina pigmented epithelial cell growth and microtubule nucleation by downregulating p150(glued).
Chen TY, Lien WC, Cheng HL, Kuan TS, Sheu SY, Wang CY
Journal of cellular physiology 2019 Jul;234(7):10445-10457
Journal of cellular physiology 2019 Jul;234(7):10445-10457
Primary cilium loss in mammalian cells occurs predominantly by whole-cilium shedding.
Mirvis M, Siemers KA, Nelson WJ, Stearns TP
PLoS biology 2019 Jul;17(7):e3000381
PLoS biology 2019 Jul;17(7):e3000381
Ciliopathy-Associated Protein Kinase ICK Requires Its Non-Catalytic Carboxyl-Terminal Domain for Regulation of Ciliogenesis.
Oh YS, Wang EJ, Gailey CD, Brautigan DL, Allen BL, Fu Z
Cells 2019 Jul 4;8(7)
Cells 2019 Jul 4;8(7)
Targeted exon skipping rescues ciliary protein composition defects in Joubert syndrome patient fibroblasts.
Molinari E, Ramsbottom SA, Srivastava S, Booth P, Alkanderi S, McLafferty SM, Devlin LA, White K, Gunay-Aygun M, Miles CG, Sayer JA
Scientific reports 2019 Jul 25;9(1):10828
Scientific reports 2019 Jul 25;9(1):10828
Spastin Contributes to Neural Development through the Regulation of Microtubule Dynamics in the Primary Cilia of Neural Stem Cells.
Jeong B, Kim TH, Kim DS, Shin WH, Lee JR, Kim NS, Lee DY
Neuroscience 2019 Jul 15;411:76-85
Neuroscience 2019 Jul 15;411:76-85
Unexpected Activities in Regulating Ciliation Contribute to Off-target Effects of Targeted Drugs.
Kiseleva AA, Korobeynikov VA, Nikonova AS, Zhang P, Makhov P, Deneka AY, Einarson MB, Serebriiskii IG, Liu H, Peterson JR, Golemis EA
Clinical cancer research : an official journal of the American Association for Cancer Research 2019 Jul 1;25(13):4179-4193
Clinical cancer research : an official journal of the American Association for Cancer Research 2019 Jul 1;25(13):4179-4193
CCDC114 is mutated in patient with a complex phenotype combining primary ciliary dyskinesia, sensorineural deafness, and renal disease.
Li P, He Y, Cai G, Xiao F, Yang J, Li Q, Chen X
Journal of human genetics 2019 Jan;64(1):39-48
Journal of human genetics 2019 Jan;64(1):39-48
Fragmentation of kidney epithelial cell primary cilia occurs by cisplatin and these cilia fragments are excreted into the urine.
Kong MJ, Bak SH, Han KH, Kim JI, Park JW, Park KM
Redox biology 2019 Jan;20:38-45
Redox biology 2019 Jan;20:38-45
Reproducibility and staging of 3D human retinal organoids across multiple pluripotent stem cell lines.
Capowski EE, Samimi K, Mayerl SJ, Phillips MJ, Pinilla I, Howden SE, Saha J, Jansen AD, Edwards KL, Jager LD, Barlow K, Valiauga R, Erlichman Z, Hagstrom A, Sinha D, Sluch VM, Chamling X, Zack DJ, Skala MC, Gamm DM
Development (Cambridge, England) 2019 Jan 9;146(1)
Development (Cambridge, England) 2019 Jan 9;146(1)
Hydrocephalus in a rat model of Meckel Gruber syndrome with a TMEM67 mutation.
Shim JW, Territo PR, Simpson S, Watson JC, Jiang L, Riley AA, McCarthy B, Persohn S, Fulkerson D, Blazer-Yost BL
Scientific reports 2019 Jan 31;9(1):1069
Scientific reports 2019 Jan 31;9(1):1069
Prominin-1 controls stem cell activation by orchestrating ciliary dynamics.
Singer D, Thamm K, Zhuang H, Karbanová J, Gao Y, Walker JV, Jin H, Wu X, Coveney CR, Marangoni P, Lu D, Grayson PRC, Gulsen T, Liu KJ, Ardu S, Wann AK, Luo S, Zambon AC, Jetten AM, Tredwin C, Klein OD, Attanasio M, Carmeliet P, Huttner WB, Corbeil D, Hu B
The EMBO journal 2019 Jan 15;38(2)
The EMBO journal 2019 Jan 15;38(2)
Bardet-Biedl Syndrome proteins regulate cilia disassembly during tissue maturation.
Patnaik SR, Kretschmer V, Brücker L, Schneider S, Volz AK, Oancea-Castillo LDR, May-Simera HL
Cellular and molecular life sciences : CMLS 2019 Feb;76(4):757-775
Cellular and molecular life sciences : CMLS 2019 Feb;76(4):757-775
Carbocisteine stimulated an increase in ciliary bend angle via a decrease in [Cl(-)](i) in mouse airway cilia.
Ikeuchi Y, Kogiso H, Hosogi S, Tanaka S, Shimamoto C, Matsumura H, Inui T, Marunaka Y, Nakahari T
Pflugers Archiv : European journal of physiology 2019 Feb;471(2):365-380
Pflugers Archiv : European journal of physiology 2019 Feb;471(2):365-380
Loss of Primary Cilia Results in the Development of Cancer in the Murine Thyroid Gland.
Lee J, Yi S, Chang JY, Kim JT, Sul HJ, Park KC, Zhu X, Cheng SY, Kero J, Kim J, Shong M
Molecules and cells 2019 Feb 28;42(2):113-122
Molecules and cells 2019 Feb 28;42(2):113-122
Recycling endosomal CD133 functions as an inhibitor of autophagy at the pericentrosomal region.
Izumi H, Li Y, Shibaki M, Mori D, Yasunami M, Sato S, Matsunaga H, Mae T, Kodama K, Kamijo T, Kaneko Y, Nakagawara A
Scientific reports 2019 Feb 19;9(1):2236
Scientific reports 2019 Feb 19;9(1):2236
Integrated miRNA and mRNA expression profiling identifies novel targets and pathological mechanisms in autoimmune thyroid diseases.
Martínez-Hernández R, Serrano-Somavilla A, Ramos-Leví A, Sampedro-Nuñez M, Lens-Pardo A, Muñoz De Nova JL, Triviño JC, González MU, Torné L, Casares-Arias J, Martín-Cófreces NB, Sánchez-Madrid F, Marazuela M
EBioMedicine 2019 Dec;50:329-342
EBioMedicine 2019 Dec;50:329-342
Rab7 regulates primary cilia disassembly through cilia excision.
Wang G, Hu HB, Chang Y, Huang Y, Song ZQ, Zhou SB, Chen L, Zhang YC, Wu M, Tu HQ, Yuan JF, Wang N, Pan X, Li AL, Zhou T, Zhang XM, He K, Li HY
The Journal of cell biology 2019 Dec 2;218(12):4030-4041
The Journal of cell biology 2019 Dec 2;218(12):4030-4041
Primary cilia deficiency in neural crest cells models anterior segment dysgenesis in mouse.
Portal C, Rompolas P, Lwigale P, Iomini C
eLife 2019 Dec 17;8
eLife 2019 Dec 17;8
Primary cilia mediate mitochondrial stress responses to promote dopamine neuron survival in a Parkinson's disease model.
Bae JE, Kang GM, Min SH, Jo DS, Jung YK, Kim K, Kim MS, Cho DH
Cell death & disease 2019 Dec 16;10(12):952
Cell death & disease 2019 Dec 16;10(12):952
AAV-Mediated Gene Augmentation Therapy Restores Critical Functions in Mutant PRPF31(+/-) iPSC-Derived RPE Cells.
Brydon EM, Bronstein R, Buskin A, Lako M, Pierce EA, Fernandez-Godino R
Molecular therapy. Methods & clinical development 2019 Dec 13;15:392-402
Molecular therapy. Methods & clinical development 2019 Dec 13;15:392-402
Neutrophil gelatinase-associated lipocalin (NGAL) is localised to the primary cilium in renal tubular epithelial cells - A novel source of urinary biomarkers of renal injury.
Cassidy H, Slyne J, Higgins M, Radford R, Conlon PJ, Watson AJ, Ryan MP, McMorrow T, Slattery C
Biochimica et biophysica acta. Molecular basis of disease 2019 Dec 1;1865(12):165532
Biochimica et biophysica acta. Molecular basis of disease 2019 Dec 1;1865(12):165532
Mutations in ARL2BP, a protein required for ciliary microtubule structure, cause syndromic male infertility in humans and mice.
Moye AR, Bedoni N, Cunningham JG, Sanzhaeva U, Tucker ES, Mathers P, Peter VG, Quinodoz M, Paris LP, Coutinho-Santos L, Camacho P, Purcell MG, Winkelmann AC, Foster JA, Pugacheva EN, Rivolta C, Ramamurthy V
PLoS genetics 2019 Aug;15(8):e1008315
PLoS genetics 2019 Aug;15(8):e1008315
HDAC3 and HDAC8 are required for cilia assembly and elongation.
Park SA, Yoo H, Seol JH, Rhee K
Biology open 2019 Aug 9;8(8)
Biology open 2019 Aug 9;8(8)
Mutations in the Heterotopia Gene Eml1/EML1 Severely Disrupt the Formation of Primary Cilia.
Uzquiano A, Cifuentes-Diaz C, Jabali A, Romero DM, Houllier A, Dingli F, Maillard C, Boland A, Deleuze JF, Loew D, Mancini GMS, Bahi-Buisson N, Ladewig J, Francis F
Cell reports 2019 Aug 6;28(6):1596-1611.e10
Cell reports 2019 Aug 6;28(6):1596-1611.e10
Characterization of an immortalized human small airway basal stem/progenitor cell line with airway region-specific differentiation capacity.
Wang G, Lou HH, Salit J, Leopold PL, Driscoll S, Schymeinsky J, Quast K, Visvanathan S, Fine JS, Thomas MJ, Crystal RG
Respiratory research 2019 Aug 23;20(1):196
Respiratory research 2019 Aug 23;20(1):196
A null allele of Dnaaf2 displays embryonic lethality and mimics human ciliary dyskinesia.
Cheong A, Degani R, Tremblay KD, Mager J
Human molecular genetics 2019 Aug 15;28(16):2775-2784
Human molecular genetics 2019 Aug 15;28(16):2775-2784
Human IFT52 mutations uncover a novel role for the protein in microtubule dynamics and centrosome cohesion.
Dupont MA, Humbert C, Huber C, Siour Q, Guerrera IC, Jung V, Christensen A, Pouliet A, Garfa-Traoré M, Nitschké P, Injeyan M, Millar K, Chitayat D, Shannon P, Girisha KM, Shukla A, Mechler C, Lorentzen E, Benmerah A, Cormier-Daire V, Jeanpierre C, Saunier S, Delous M
Human molecular genetics 2019 Aug 15;28(16):2720-2737
Human molecular genetics 2019 Aug 15;28(16):2720-2737
Restoration of primary cilia in obese adipose-derived mesenchymal stem cells by inhibiting Aurora A or extracellular signal-regulated kinase.
Ritter A, Kreis NN, Roth S, Friemel A, Jennewein L, Eichbaum C, Solbach C, Louwen F, Yuan J
Stem cell research & therapy 2019 Aug 14;10(1):255
Stem cell research & therapy 2019 Aug 14;10(1):255
A CEP104-CSPP1 Complex Is Required for Formation of Primary Cilia Competent in Hedgehog Signaling.
Frikstad KM, Molinari E, Thoresen M, Ramsbottom SA, Hughes F, Letteboer SJF, Gilani S, Schink KO, Stokke T, Geimer S, Pedersen LB, Giles RH, Akhmanova A, Roepman R, Sayer JA, Patzke S
Cell reports 2019 Aug 13;28(7):1907-1922.e6
Cell reports 2019 Aug 13;28(7):1907-1922.e6
Mutagenesis of putative ciliary genes with the CRISPR/Cas9 system in zebrafish identifies genes required for retinal development.
Hu R, Huang W, Liu J, Jin M, Wu Y, Li J, Wang J, Yu Z, Wang H, Cao Y
FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2019 Apr;33(4):5248-5256
FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2019 Apr;33(4):5248-5256
Tubular flow activates magnesium transport in the distal convoluted tubule.
Verschuren EHJ, Hoenderop JGJ, Peters DJM, Arjona FJ, Bindels RJM
FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2019 Apr;33(4):5034-5044
FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2019 Apr;33(4):5034-5044
The RNA-Protein Interactome of Differentiated Kidney Tubular Epithelial Cells.
Ignarski M, Rill C, Kaiser RWJ, Kaldirim M, Neuhaus R, Esmaillie R, Li X, Klein C, Bohl K, Petersen M, Frese CK, Höhne M, Atanassov I, Rinschen MM, Höpker K, Schermer B, Benzing T, Dieterich C, Fabretti F, Müller RU
Journal of the American Society of Nephrology : JASN 2019 Apr;30(4):564-576
Journal of the American Society of Nephrology : JASN 2019 Apr;30(4):564-576
CEP120 interacts with C2CD3 and Talpid3 and is required for centriole appendage assembly and ciliogenesis.
Tsai JJ, Hsu WB, Liu JH, Chang CW, Tang TK
Scientific reports 2019 Apr 15;9(1):6037
Scientific reports 2019 Apr 15;9(1):6037
Acute Inhibition of Heterotrimeric Kinesin-2 Function Reveals Mechanisms of Intraflagellar Transport in Mammalian Cilia.
Engelke MF, Waas B, Kearns SE, Suber A, Boss A, Allen BL, Verhey KJ
Current biology : CB 2019 Apr 1;29(7):1137-1148.e4
Current biology : CB 2019 Apr 1;29(7):1137-1148.e4
Glioma cell proliferation is enhanced in the presence of tumor-derived cilia vesicles.
Hoang-Minh LB, Dutra-Clarke M, Breunig JJ, Sarkisian MR
Cilia 2018;7:6
Cilia 2018;7:6
Adenylyl cyclases and TRPV4 mediate Ca(2+)/cAMP dynamics to enhance fluid flow-induced osteogenesis in osteocytes.
Moore ER, Ryu HS, Zhu YX, Jacobs CR
Journal of molecular biochemistry 2018;7:48-59
Journal of molecular biochemistry 2018;7:48-59
Ciliogenesis is reciprocally regulated by PPARA and NR1H4/FXR through controlling autophagy in vitro and in vivo.
Liu ZQ, Lee JN, Son M, Lim JY, Dutta RK, Maharjan Y, Kwak S, Oh GT, Byun K, Choe SK, Park R
Autophagy 2018;14(6):1011-1027
Autophagy 2018;14(6):1011-1027
Dorsoventral differences in cAMP levels and correlated changes in the subcellular distribution of the PKA catalytic domain, provide further evidence that PKA signaling coordinates dorsoventral patterning of the otocyst.
Ohta S, Schoenwolf GC
Development, growth & differentiation 2018 Sep;60(7):431-441
Development, growth & differentiation 2018 Sep;60(7):431-441
A distal centriolar protein network controls organelle maturation and asymmetry.
Wang L, Failler M, Fu W, Dynlacht BD
Nature communications 2018 Sep 26;9(1):3938
Nature communications 2018 Sep 26;9(1):3938
LRRC45 contributes to early steps of axoneme extension.
Kurtulmus B, Yuan C, Schuy J, Neuner A, Hata S, Kalamakis G, Martin-Villalba A, Pereira G
Journal of cell science 2018 Sep 20;131(18)
Journal of cell science 2018 Sep 20;131(18)
A slow-cycling LGR5 tumour population mediates basal cell carcinoma relapse after therapy.
Sánchez-Danés A, Larsimont JC, Liagre M, Muñoz-Couselo E, Lapouge G, Brisebarre A, Dubois C, Suppa M, Sukumaran V, Del Marmol V, Tabernero J, Blanpain C
Nature 2018 Oct;562(7727):434-438
Nature 2018 Oct;562(7727):434-438
Homozygous variants in KIAA1549, encoding a ciliary protein, are associated with autosomal recessive retinitis pigmentosa.
de Bruijn SE, Verbakel SK, de Vrieze E, Kremer H, Cremers FPM, Hoyng CB, van den Born LI, Roosing S
Journal of medical genetics 2018 Oct;55(10):705-712
Journal of medical genetics 2018 Oct;55(10):705-712
Characterization of Primary Cilia in Normal Fallopian Tube Epithelium and Serous Tubal Intraepithelial Carcinoma.
Abdelhamed ZA, Ryan TA, Fuller M, Coulson-Gilmer C, Abdelmottaleb DI, Wang TL, Kaun JC, Wang P, Hutson R, Wilkinson N, Bell SM, Johnson CA
International journal of gynecological cancer : official journal of the International Gynecological Cancer Society 2018 Oct;28(8):1535-1544
International journal of gynecological cancer : official journal of the International Gynecological Cancer Society 2018 Oct;28(8):1535-1544
The Ciliary Machinery Is Repurposed for T Cell Immune Synapse Trafficking of LCK.
Stephen LA, ElMaghloob Y, McIlwraith MJ, Yelland T, Castro Sanchez P, Roda-Navarro P, Ismail S
Developmental cell 2018 Oct 8;47(1):122-132.e4
Developmental cell 2018 Oct 8;47(1):122-132.e4
ARL3 Mutations Cause Joubert Syndrome by Disrupting Ciliary Protein Composition.
Alkanderi S, Molinari E, Shaheen R, Elmaghloob Y, Stephen LA, Sammut V, Ramsbottom SA, Srivastava S, Cairns G, Edwards N, Rice SJ, Ewida N, Alhashem A, White K, Miles CG, Steel DH, Alkuraya FS, Ismail S, Sayer JA
American journal of human genetics 2018 Oct 4;103(4):612-620
American journal of human genetics 2018 Oct 4;103(4):612-620
M-Phase Phosphoprotein 9 regulates ciliogenesis by modulating CP110-CEP97 complex localization at the mother centriole.
Huang N, Zhang D, Li F, Chai P, Wang S, Teng J, Chen J
Nature communications 2018 Oct 30;9(1):4511
Nature communications 2018 Oct 30;9(1):4511
Dynein-2 intermediate chains play crucial but distinct roles in primary cilia formation and function.
Vuolo L, Stevenson NL, Heesom KJ, Stephens DJ
eLife 2018 Oct 16;7
eLife 2018 Oct 16;7
Disrupted alternative splicing for genes implicated in splicing and ciliogenesis causes PRPF31 retinitis pigmentosa.
Buskin A, Zhu L, Chichagova V, Basu B, Mozaffari-Jovin S, Dolan D, Droop A, Collin J, Bronstein R, Mehrotra S, Farkas M, Hilgen G, White K, Pan KT, Treumann A, Hallam D, Bialas K, Chung G, Mellough C, Ding Y, Krasnogor N, Przyborski S, Zwolinski S, Al-Aama J, Alharthi S, Xu Y, Wheway G, Szymanska K, McKibbin M, Inglehearn CF, Elliott DJ, Lindsay S, Ali RR, Steel DH, Armstrong L, Sernagor E, Urlaub H, Pierce E, Lührmann R, Grellscheid SN, Johnson CA, Lako M
Nature communications 2018 Oct 12;9(1):4234
Nature communications 2018 Oct 12;9(1):4234
Mutation of FOP/FGFR1OP in mice recapitulates human short rib-polydactyly ciliopathy.
Cabaud O, Roubin R, Comte A, Bascunana V, Sergé A, Sedjaï F, Birnbaum D, Rosnet O, Acquaviva C
Human molecular genetics 2018 Oct 1;27(19):3377-3391
Human molecular genetics 2018 Oct 1;27(19):3377-3391
Membrane association and remodeling by intraflagellar transport protein IFT172.
Wang Q, Taschner M, Ganzinger KA, Kelley C, Villasenor A, Heymann M, Schwille P, Lorentzen E, Mizuno N
Nature communications 2018 Nov 8;9(1):4684
Nature communications 2018 Nov 8;9(1):4684
A Cell/Cilia Cycle Biosensor for Single-Cell Kinetics Reveals Persistence of Cilia after G1/S Transition Is a General Property in Cells and Mice.
Ford MJ, Yeyati PL, Mali GR, Keighren MA, Waddell SH, Mjoseng HK, Douglas AT, Hall EA, Sakaue-Sawano A, Miyawaki A, Meehan RR, Boulter L, Jackson IJ, Mill P, Mort RL
Developmental cell 2018 Nov 19;47(4):509-523.e5
Developmental cell 2018 Nov 19;47(4):509-523.e5
Oncoprotein CIP2A promotes the disassembly of primary cilia and inhibits glycolytic metabolism.
Jeong AL, Ka HI, Han S, Lee S, Lee EW, Soh SJ, Joo HJ, Sumiyasuren B, Park JY, Lim JS, Park JH, Lee MS, Yang Y
EMBO reports 2018 May;19(5)
EMBO reports 2018 May;19(5)
Primary cilia sensitize endothelial cells to BMP and prevent excessive vascular regression.
Vion AC, Alt S, Klaus-Bergmann A, Szymborska A, Zheng T, Perovic T, Hammoutene A, Oliveira MB, Bartels-Klein E, Hollfinger I, Rautou PE, Bernabeu MO, Gerhardt H
The Journal of cell biology 2018 May 7;217(5):1651-1665
The Journal of cell biology 2018 May 7;217(5):1651-1665
The Joubert syndrome protein ARL13B binds tubulin to maintain uniform distribution of proteins along the ciliary membrane.
Revenkova E, Liu Q, Gusella GL, Iomini C
Journal of cell science 2018 May 4;131(9)
Journal of cell science 2018 May 4;131(9)
Regulator of calcineurin-2 is a centriolar protein with a role in cilia length control.
Stevenson NL, Bergen DJM, Xu A, Wyatt E, Henry F, McCaughey J, Vuolo L, Hammond CL, Stephens DJ
Journal of cell science 2018 May 4;131(9)
Journal of cell science 2018 May 4;131(9)
Cell type-specific regulation of ciliary transition zone assembly in vertebrates.
Wiegering A, Dildrop R, Kalfhues L, Spychala A, Kuschel S, Lier JM, Zobel T, Dahmen S, Leu T, Struchtrup A, Legendre F, Vesque C, Schneider-Maunoury S, Saunier S, Rüther U, Gerhardt C
The EMBO journal 2018 May 15;37(10)
The EMBO journal 2018 May 15;37(10)
Loss of Tctn3 causes neuronal apoptosis and neural tube defects in mice.
Wang B, Zhang Y, Dong H, Gong S, Wei B, Luo M, Wang H, Wu X, Liu W, Xu X, Zheng Y, Sun M
Cell death & disease 2018 May 1;9(5):520
Cell death & disease 2018 May 1;9(5):520
Quantitative Comparison of Primary Cilia Marker Expression and Length in the Mouse Brain.
Sipos É, Komoly S, Ács P
Journal of molecular neuroscience : MN 2018 Mar;64(3):397-409
Journal of molecular neuroscience : MN 2018 Mar;64(3):397-409
The actin-MRTF-SRF transcriptional circuit controls tubulin acetylation via α-TAT1 gene expression.
Fernández-Barrera J, Bernabé-Rubio M, Casares-Arias J, Rangel L, Fernández-Martín L, Correas I, Alonso MA
The Journal of cell biology 2018 Mar 5;217(3):929-944
The Journal of cell biology 2018 Mar 5;217(3):929-944
Insm1 Induces Neural Progenitor Delamination in Developing Neocortex via Downregulation of the Adherens Junction Belt-Specific Protein Plekha7.
Tavano S, Taverna E, Kalebic N, Haffner C, Namba T, Dahl A, Wilsch-Bräuninger M, Paridaen JTML, Huttner WB
Neuron 2018 Mar 21;97(6):1299-1314.e8
Neuron 2018 Mar 21;97(6):1299-1314.e8
Regulation of ciliary function by fibroblast growth factor signaling identifies FGFR3-related disorders achondroplasia and thanatophoric dysplasia as ciliopathies.
Kunova Bosakova M, Varecha M, Hampl M, Duran I, Nita A, Buchtova M, Dosedelova H, Machat R, Xie Y, Ni Z, Martin JH, Chen L, Jansen G, Krakow D, Krejci P
Human molecular genetics 2018 Mar 15;27(6):1093-1105
Human molecular genetics 2018 Mar 15;27(6):1093-1105
Mice Expressing Myc in Neural Precursors Develop Choroid Plexus and Ciliary Body Tumors.
Shannon ML, Fame RM, Chau KF, Dani N, Calicchio ML, Géléoc GS, Lidov HGW, Alexandrescu S, Lehtinen MK
The American journal of pathology 2018 Jun;188(6):1334-1344
The American journal of pathology 2018 Jun;188(6):1334-1344
Primary Cilia Mediate Diverse Kinase Inhibitor Resistance Mechanisms in Cancer.
Jenks AD, Vyse S, Wong JP, Kostaras E, Keller D, Burgoyne T, Shoemark A, Tsalikis A, de la Roche M, Michaelis M, Cinatl J Jr, Huang PH, Tanos BE
Cell reports 2018 Jun 5;23(10):3042-3055
Cell reports 2018 Jun 5;23(10):3042-3055
Targeted deletion of the AAA-ATPase Ruvbl1 in mice disrupts ciliary integrity and causes renal disease and hydrocephalus.
Dafinger C, Rinschen MM, Borgal L, Ehrenberg C, Basten SG, Franke M, Höhne M, Rauh M, Göbel H, Bloch W, Wunderlich FT, Peters DJM, Tasche D, Mishra T, Habbig S, Dötsch J, Müller RU, Brüning JC, Persigehl T, Giles RH, Benzing T, Schermer B, Liebau MC
Experimental & molecular medicine 2018 Jun 28;50(6):1-17
Experimental & molecular medicine 2018 Jun 28;50(6):1-17
Autophagy alteration prevents primary cilium disassembly in RPE1 cells.
Maharjan Y, Lee JN, Kwak S, Lim H, Dutta RK, Liu ZQ, So HS, Park R
Biochemical and biophysical research communications 2018 Jun 2;500(2):242-248
Biochemical and biophysical research communications 2018 Jun 2;500(2):242-248
Cilia protein IFT88 regulates extracellular protease activity by optimizing LRP-1-mediated endocytosis.
Coveney CR, Collins I, Mc Fie M, Chanalaris A, Yamamoto K, Wann AKT
FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2018 Jun 19;32(12):fj201800334
FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2018 Jun 19;32(12):fj201800334
Primary cilia-regulated transcriptome in the renal collecting duct.
Mohammed SG, Arjona FJ, Verschuren EHJ, Bakey Z, Alkema W, van Hijum S, Schmidts M, Bindels RJM, Hoenderop JGJ
FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2018 Jul;32(7):3653-3668
FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2018 Jul;32(7):3653-3668
CRISPR/Cas9-mediated Genomic Editing of Cluap1/IFT38 Reveals a New Role in Actin Arrangement.
Beyer T, Bolz S, Junger K, Horn N, Moniruzzaman M, Wissinger Y, Ueffing M, Boldt K
Molecular & cellular proteomics : MCP 2018 Jul;17(7):1285-1294
Molecular & cellular proteomics : MCP 2018 Jul;17(7):1285-1294
EZH2-Mediated Primary Cilium Deconstruction Drives Metastatic Melanoma Formation.
Zingg D, Debbache J, Peña-Hernández R, Antunes AT, Schaefer SM, Cheng PF, Zimmerli D, Haeusel J, Calçada RR, Tuncer E, Zhang Y, Bossart R, Wong KK, Basler K, Dummer R, Santoro R, Levesque MP, Sommer L
Cancer cell 2018 Jul 9;34(1):69-84.e14
Cancer cell 2018 Jul 9;34(1):69-84.e14
Brain Somatic Mutations in MTOR Disrupt Neuronal Ciliogenesis, Leading to Focal Cortical Dyslamination.
Park SM, Lim JS, Ramakrishina S, Kim SH, Kim WK, Lee J, Kang HC, Reiter JF, Kim DS, Kim HH, Lee JH
Neuron 2018 Jul 11;99(1):83-97.e7
Neuron 2018 Jul 11;99(1):83-97.e7
Glucose deprivation induces primary cilium formation through mTORC1 inactivation.
Takahashi K, Nagai T, Chiba S, Nakayama K, Mizuno K
Journal of cell science 2018 Jan 8;131(1)
Journal of cell science 2018 Jan 8;131(1)
CDKL Family Kinases Have Evolved Distinct Structural Features and Ciliary Function.
Canning P, Park K, Gonçalves J, Li C, Howard CJ, Sharpe TD, Holt LJ, Pelletier L, Bullock AN, Leroux MR
Cell reports 2018 Jan 23;22(4):885-894
Cell reports 2018 Jan 23;22(4):885-894
Primary Cilium-Mediated Retinal Pigment Epithelium Maturation Is Disrupted in Ciliopathy Patient Cells.
May-Simera HL, Wan Q, Jha BS, Hartford J, Khristov V, Dejene R, Chang J, Patnaik S, Lu Q, Banerjee P, Silver J, Insinna-Kettenhofen C, Patel D, Lotfi M, Malicdan M, Hotaling N, Maminishkis A, Sridharan R, Brooks B, Miyagishima K, Gunay-Aygun M, Pal R, Westlake C, Miller S, Sharma R, Bharti K
Cell reports 2018 Jan 2;22(1):189-205
Cell reports 2018 Jan 2;22(1):189-205
Functional characterization of tektin-1 in motile cilia and evidence for TEKT1 as a new candidate gene for motile ciliopathies.
Ryan R, Failler M, Reilly ML, Garfa-Traore M, Delous M, Filhol E, Reboul T, Bole-Feysot C, Nitschké P, Baudouin V, Amselem S, Escudier E, Legendre M, Benmerah A, Saunier S
Human molecular genetics 2018 Jan 15;27(2):266-282
Human molecular genetics 2018 Jan 15;27(2):266-282
Ciliogenesis and cell cycle alterations contribute to KIF2A-related malformations of cortical development.
Broix L, Asselin L, Silva CG, Ivanova EL, Tilly P, Gilet JG, Lebrun N, Jagline H, Muraca G, Saillour Y, Drouot N, Reilly ML, Francis F, Benmerah A, Bahi-Buisson N, Belvindrah R, Nguyen L, Godin JD, Chelly J, Hinckelmann MV
Human molecular genetics 2018 Jan 15;27(2):224-238
Human molecular genetics 2018 Jan 15;27(2):224-238
Constitutively-active FGFR3 disrupts primary cilium length and IFT20 trafficking in various chondrocyte models of achondroplasia.
Martin L, Kaci N, Estibals V, Goudin N, Garfa-Traore M, Benoist-Lasselin C, Dambroise E, Legeai-Mallet L
Human molecular genetics 2018 Jan 1;27(1):1-13
Human molecular genetics 2018 Jan 1;27(1):1-13
Adenylyl cyclase 5 deficiency reduces renal cyclic AMP and cyst growth in an orthologous mouse model of polycystic kidney disease.
Wang Q, Cobo-Stark P, Patel V, Somlo S, Han PL, Igarashi P
Kidney international 2018 Feb;93(2):403-415
Kidney international 2018 Feb;93(2):403-415
Export of membrane proteins from the Golgi complex to the primary cilium requires the kinesin motor, KIFC1.
Lee SH, Joo K, Jung EJ, Hong H, Seo J, Kim J
FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2018 Feb;32(2):957-968
FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2018 Feb;32(2):957-968
WDR11-mediated Hedgehog signalling defects underlie a new ciliopathy related to Kallmann syndrome.
Kim YJ, Osborn DP, Lee JY, Araki M, Araki K, Mohun T, Känsäkoski J, Brandstack N, Kim HT, Miralles F, Kim CH, Brown NA, Kim HG, Martinez-Barbera JP, Ataliotis P, Raivio T, Layman LC, Kim SH
EMBO reports 2018 Feb;19(2):269-289
EMBO reports 2018 Feb;19(2):269-289
Deficiency of primary cilia in kidney epithelial cells induces epithelial to mesenchymal transition.
Han SJ, Jung JK, Im SS, Lee SR, Jang BC, Park KM, Kim JI
Biochemical and biophysical research communications 2018 Feb 5;496(2):450-454
Biochemical and biophysical research communications 2018 Feb 5;496(2):450-454
DNAAF1 links heart laterality with the AAA+ ATPase RUVBL1 and ciliary intraflagellar transport.
Hartill VL, van de Hoek G, Patel MP, Little R, Watson CM, Berry IR, Shoemark A, Abdelmottaleb D, Parkes E, Bacchelli C, Szymanska K, Knoers NV, Scambler PJ, Ueffing M, Boldt K, Yates R, Winyard PJ, Adler B, Moya E, Hattingh L, Shenoy A, Hogg C, Sheridan E, Roepman R, Norris D, Mitchison HM, Giles RH, Johnson CA
Human molecular genetics 2018 Feb 1;27(3):529-545
Human molecular genetics 2018 Feb 1;27(3):529-545
Truncated SALL1 Impedes Primary Cilia Function in Townes-Brocks Syndrome.
Bozal-Basterra L, Martín-Ruíz I, Pirone L, Liang Y, Sigurðsson JO, Gonzalez-Santamarta M, Giordano I, Gabicagogeascoa E, de Luca A, Rodríguez JA, Wilkie AOM, Kohlhase J, Eastwood D, Yale C, Olsen JV, Rauchman M, Anderson KV, Sutherland JD, Barrio R
American journal of human genetics 2018 Feb 1;102(2):249-265
American journal of human genetics 2018 Feb 1;102(2):249-265
Reconstructing the Human Renal Vascular-Tubular Unit In Vitro.
Rayner SG, Phong KT, Xue J, Lih D, Shankland SJ, Kelly EJ, Himmelfarb J, Zheng Y
Advanced healthcare materials 2018 Dec;7(23):e1801120
Advanced healthcare materials 2018 Dec;7(23):e1801120
Targeted exon skipping of a CEP290 mutation rescues Joubert syndrome phenotypes in vitro and in a murine model.
Ramsbottom SA, Molinari E, Srivastava S, Silberman F, Henry C, Alkanderi S, Devlin LA, White K, Steel DH, Saunier S, Miles CG, Sayer JA
Proceedings of the National Academy of Sciences of the United States of America 2018 Dec 4;115(49):12489-12494
Proceedings of the National Academy of Sciences of the United States of America 2018 Dec 4;115(49):12489-12494
Kinesin-2 and IFT-A act as a complex promoting nuclear localization of β-catenin during Wnt signalling.
Vuong LT, Iomini C, Balmer S, Esposito D, Aaronson SA, Mlodzik M
Nature communications 2018 Dec 13;9(1):5304
Nature communications 2018 Dec 13;9(1):5304
Cullin-3-KCTD10-mediated CEP97 degradation promotes primary cilium formation.
Nagai T, Mukoyama S, Kagiwada H, Goshima N, Mizuno K
Journal of cell science 2018 Dec 12;131(24)
Journal of cell science 2018 Dec 12;131(24)
Microtubule asters anchored by FSD1 control axoneme assembly and ciliogenesis.
Tu HQ, Qin XH, Liu ZB, Song ZQ, Hu HB, Zhang YC, Chang Y, Wu M, Huang Y, Bai YF, Wang G, Han QY, Li AL, Zhou T, Liu F, Zhang XM, Li HY
Nature communications 2018 Dec 11;9(1):5277
Nature communications 2018 Dec 11;9(1):5277
Loss-of-function of IFT88 determines metabolic phenotypes in thyroid cancer.
Lee J, Yi S, Won M, Song YS, Yi HS, Park YJ, Park KC, Kim JT, Chang JY, Lee MJ, Sul HJ, Choi JE, Kim KS, Kero J, Kim J, Shong M
Oncogene 2018 Aug;37(32):4455-4474
Oncogene 2018 Aug;37(32):4455-4474
Axoneme polyglutamylation regulated by Joubert syndrome protein ARL13B controls ciliary targeting of signaling molecules.
He K, Ma X, Xu T, Li Y, Hodge A, Zhang Q, Torline J, Huang Y, Zhao J, Ling K, Hu J
Nature communications 2018 Aug 17;9(1):3310
Nature communications 2018 Aug 17;9(1):3310
Basal exon skipping and nonsense-associated altered splicing allows bypassing complete CEP290 loss-of-function in individuals with unusually mild retinal disease.
Barny I, Perrault I, Michel C, Soussan M, Goudin N, Rio M, Thomas S, Attié-Bitach T, Hamel C, Dollfus H, Kaplan J, Rozet JM, Gerard X
Human molecular genetics 2018 Aug 1;27(15):2689-2702
Human molecular genetics 2018 Aug 1;27(15):2689-2702
Aspm knockout ferret reveals an evolutionary mechanism governing cerebral cortical size.
Johnson MB, Sun X, Kodani A, Borges-Monroy R, Girskis KM, Ryu SC, Wang PP, Patel K, Gonzalez DM, Woo YM, Yan Z, Liang B, Smith RS, Chatterjee M, Coman D, Papademetris X, Staib LH, Hyder F, Mandeville JB, Grant PE, Im K, Kwak H, Engelhardt JF, Walsh CA, Bae BI
Nature 2018 Apr;556(7701):370-375
Nature 2018 Apr;556(7701):370-375
Mapping GPR88-Venus illuminates a novel role for GPR88 in sensory processing.
Ehrlich AT, Semache M, Bailly J, Wojcik S, Arefin TM, Colley C, Le Gouill C, Gross F, Lukasheva V, Hogue M, Darcq E, Harsan LA, Bouvier M, Kieffer BL
Brain structure & function 2018 Apr;223(3):1275-1296
Brain structure & function 2018 Apr;223(3):1275-1296
Robust interaction of IFT70 with IFT52-IFT88 in the IFT-B complex is required for ciliogenesis.
Takei R, Katoh Y, Nakayama K
Biology open 2018 Apr 30;7(5)
Biology open 2018 Apr 30;7(5)
Centrobin controls primary ciliogenesis in vertebrates.
Ogungbenro YA, Tena TC, Gaboriau D, Lalor P, Dockery P, Philipp M, Morrison CG
The Journal of cell biology 2018 Apr 2;217(4):1205-1215
The Journal of cell biology 2018 Apr 2;217(4):1205-1215
Mutations in IFT-A satellite core component genes IFT43 and IFT121 produce short rib polydactyly syndrome with distinctive campomelia.
Duran I, Taylor SP, Zhang W, Martin J, Qureshi F, Jacques SM, Wallerstein R, Lachman RS, Nickerson DA, Bamshad M, Cohn DH, Krakow D
Cilia 2017;6:7
Cilia 2017;6:7
Abnormal glycosylation in Joubert syndrome type 10.
Kane MS, Davids M, Bond MR, Adams CJ, Grout ME, Phelps IG, O'Day DR, Dempsey JC, Li X, Golas G, Vezina G, Gunay-Aygun M, Hanover JA, Doherty D, He M, Malicdan MCV, Gahl WA, Boerkoel CF
Cilia 2017;6:2
Cilia 2017;6:2
Jasplakinolide induces primary cilium formation through cell rounding and YAP inactivation.
Nagai T, Mizuno K
PloS one 2017;12(8):e0183030
PloS one 2017;12(8):e0183030
Widespread GLI expression but limited canonical hedgehog signaling restricted to the ductular reaction in human chronic liver disease.
Grzelak CA, Sigglekow ND, Tirnitz-Parker JE, Hamson EJ, Warren A, Maneck B, Chen J, Patkunanathan B, Boland J, Cheng R, Shackel NA, Seth D, Bowen DG, Martelotto LG, Watkins DN, McCaughan GW
PloS one 2017;12(2):e0171480
PloS one 2017;12(2):e0171480
Missense mutations in the WD40 domain of AHI1 cause non-syndromic retinitis pigmentosa.
Nguyen TT, Hull S, Roepman R, van den Born LI, Oud MM, de Vrieze E, Hetterschijt L, Letteboer SJF, van Beersum SEC, Blokland EA, Yntema HG, Cremers FPM, van der Zwaag PA, Arno G, van Wijk E, Webster AR, Haer-Wigman L
Journal of medical genetics 2017 Sep;54(9):624-632
Journal of medical genetics 2017 Sep;54(9):624-632
Reduced primary cilia length and altered Arl13b expression are associated with deregulated chondrocyte Hedgehog signaling in alkaptonuria.
Thorpe SD, Gambassi S, Thompson CL, Chandrakumar C, Santucci A, Knight MM
Journal of cellular physiology 2017 Sep;232(9):2407-2417
Journal of cellular physiology 2017 Sep;232(9):2407-2417
The ciliary membrane-associated proteome reveals actin-binding proteins as key components of cilia.
Kohli P, Höhne M, Jüngst C, Bertsch S, Ebert LK, Schauss AC, Benzing T, Rinschen MM, Schermer B
EMBO reports 2017 Sep;18(9):1521-1535
EMBO reports 2017 Sep;18(9):1521-1535
CDK10 Mutations in Humans and Mice Cause Severe Growth Retardation, Spine Malformations, and Developmental Delays.
Windpassinger C, Piard J, Bonnard C, Alfadhel M, Lim S, Bisteau X, Blouin S, Ali NB, Ng AYJ, Lu H, Tohari S, Talib SZA, van Hul N, Caldez MJ, Van Maldergem L, Yigit G, Kayserili H, Youssef SA, Coppola V, de Bruin A, Tessarollo L, Choi H, Rupp V, Roetzer K, Roschger P, Klaushofer K, Altmüller J, Roy S, Venkatesh B, Ganger R, Grill F, Ben Chehida F, Wollnik B, Altunoglu U, Al Kaissi A, Reversade B, Kaldis P
American journal of human genetics 2017 Sep 7;101(3):391-403
American journal of human genetics 2017 Sep 7;101(3):391-403
Chondrocyte expansion is associated with loss of primary cilia and disrupted hedgehog signalling.
Thompson CL, Plant JC, Wann AK, Bishop CL, Novak P, Mitchison HM, Beales PL, Chapple JP, Knight MM
European cells & materials 2017 Sep 20;34:128-141
European cells & materials 2017 Sep 20;34:128-141
Primary cilia modulate TLR4-mediated inflammatory responses in hippocampal neurons.
Baek H, Shin HJ, Kim JJ, Shin N, Kim S, Yi MH, Zhang E, Hong J, Kang JW, Kim Y, Kim CS, Kim DW
Journal of neuroinflammation 2017 Sep 19;14(1):189
Journal of neuroinflammation 2017 Sep 19;14(1):189
GPR37L1 modulates seizure susceptibility: Evidence from mouse studies and analyses of a human GPR37L1 variant.
Giddens MM, Wong JC, Schroeder JP, Farrow EG, Smith BM, Owino S, Soden SE, Meyer RC, Saunders C, LePichon JB, Weinshenker D, Escayg A, Hall RA
Neurobiology of disease 2017 Oct;106:181-190
Neurobiology of disease 2017 Oct;106:181-190
Palmitoylation of the ciliary GTPase ARL13b is necessary for its stability and its role in cilia formation.
Roy K, Jerman S, Jozsef L, McNamara T, Onyekaba G, Sun Z, Marin EP
The Journal of biological chemistry 2017 Oct 27;292(43):17703-17717
The Journal of biological chemistry 2017 Oct 27;292(43):17703-17717
Loss of OCRL increases ciliary PI(4,5)P(2) in Lowe oculocerebrorenal syndrome.
Prosseda PP, Luo N, Wang B, Alvarado JA, Hu Y, Sun Y
Journal of cell science 2017 Oct 15;130(20):3447-3454
Journal of cell science 2017 Oct 15;130(20):3447-3454
A novel Cre-inducible knock-in ARL13B-tRFP fusion cilium reporter.
Schmitz F, Burtscher I, Stauber M, Gossler A, Lickert H
Genesis (New York, N.Y. : 2000) 2017 Nov;55(11)
Genesis (New York, N.Y. : 2000) 2017 Nov;55(11)
Neural crest cells utilize primary cilia to regulate ventral forebrain morphogenesis via Hedgehog-dependent regulation of oriented cell division.
Schock EN, Brugmann SA
Developmental biology 2017 Nov 15;431(2):168-178
Developmental biology 2017 Nov 15;431(2):168-178
Human Embryonic Stem Cell-Derived Cardiomyocytes Self-Arrange with Areas of Different Subtypes During Differentiation.
Vestergaard ML, Grubb S, Koefoed K, Anderson-Jenkins Z, Grunnet-Lauridsen K, Calloe K, Clausen C, Christensen ST, Møllgård K, Andersen CY
Stem cells and development 2017 Nov 1;26(21):1566-1577
Stem cells and development 2017 Nov 1;26(21):1566-1577
An ovine hepatorenal fibrocystic model of a Meckel-like syndrome associated with dysmorphic primary cilia and TMEM67 mutations.
Stayner C, Poole CA, McGlashan SR, Pilanthananond M, Brauning R, Markie D, Lett B, Slobbe L, Chae A, Johnstone AC, Jensen CG, McEwan JC, Dittmer K, Parker K, Wiles A, Blackburne W, Leichter A, Leask M, Pinnapureddy A, Jennings M, Horsfield JA, Walker RJ, Eccles MR
Scientific reports 2017 May 9;7(1):1601
Scientific reports 2017 May 9;7(1):1601
Afadin controls cell polarization and mitotic spindle orientation in developing cortical radial glia.
Rakotomamonjy J, Brunner M, Jüschke C, Zang K, Huang EJ, Reichardt LF, Chenn A
Neural development 2017 May 8;12(1):7
Neural development 2017 May 8;12(1):7
Recent Zika Virus Isolates Induce Premature Differentiation of Neural Progenitors in Human Brain Organoids.
Gabriel E, Ramani A, Karow U, Gottardo M, Natarajan K, Gooi LM, Goranci-Buzhala G, Krut O, Peters F, Nikolic M, Kuivanen S, Korhonen E, Smura T, Vapalahti O, Papantonis A, Schmidt-Chanasit J, Riparbelli M, Callaini G, Krönke M, Utermöhlen O, Gopalakrishnan J
Cell stem cell 2017 Mar 2;20(3):397-406.e5
Cell stem cell 2017 Mar 2;20(3):397-406.e5
Centriole splitting caused by loss of the centrosomal linker protein C-NAP1 reduces centriolar satellite density and impedes centrosome amplification.
Flanagan AM, Stavenschi E, Basavaraju S, Gaboriau D, Hoey DA, Morrison CG
Molecular biology of the cell 2017 Mar 15;28(6):736-745
Molecular biology of the cell 2017 Mar 15;28(6):736-745
SOX4 regulates gonad morphogenesis and promotes male germ cell differentiation in mice.
Zhao L, Arsenault M, Ng ET, Longmuss E, Chau TC, Hartwig S, Koopman P
Developmental biology 2017 Mar 1;423(1):46-56
Developmental biology 2017 Mar 1;423(1):46-56
Ciliary entry of KIF17 is dependent on its binding to the IFT-B complex via IFT46-IFT56 as well as on its nuclear localization signal.
Funabashi T, Katoh Y, Michisaka S, Terada M, Sugawa M, Nakayama K
Molecular biology of the cell 2017 Mar 1;28(5):624-633
Molecular biology of the cell 2017 Mar 1;28(5):624-633
Primary Cilium-Regulated EG-VEGF Signaling Facilitates Trophoblast Invasion.
Wang CY, Tsai HL, Syu JS, Chen TY, Su MT
Journal of cellular physiology 2017 Jun;232(6):1467-1477
Journal of cellular physiology 2017 Jun;232(6):1467-1477
Mutations in DZIP1L, which encodes a ciliary-transition-zone protein, cause autosomal recessive polycystic kidney disease.
Lu H, Galeano MCR, Ott E, Kaeslin G, Kausalya PJ, Kramer C, Ortiz-Brüchle N, Hilger N, Metzis V, Hiersche M, Tay SY, Tunningley R, Vij S, Courtney AD, Whittle B, Wühl E, Vester U, Hartleben B, Neuber S, Frank V, Little MH, Epting D, Papathanasiou P, Perkins AC, Wright GD, Hunziker W, Gee HY, Otto EA, Zerres K, Hildebrandt F, Roy S, Wicking C, Bergmann C
Nature genetics 2017 Jul;49(7):1025-1034
Nature genetics 2017 Jul;49(7):1025-1034
Guided self-organization and cortical plate formation in human brain organoids.
Lancaster MA, Corsini NS, Wolfinger S, Gustafson EH, Phillips AW, Burkard TR, Otani T, Livesey FJ, Knoblich JA
Nature biotechnology 2017 Jul;35(7):659-666
Nature biotechnology 2017 Jul;35(7):659-666
Ciliary Hedgehog Signaling Restricts Injury-Induced Adipogenesis.
Kopinke D, Roberson EC, Reiter JF
Cell 2017 Jul 13;170(2):340-351.e12
Cell 2017 Jul 13;170(2):340-351.e12
In Vitro Modeling Using Ciliopathy-Patient-Derived Cells Reveals Distinct Cilia Dysfunctions Caused by CEP290 Mutations.
Shimada H, Lu Q, Insinna-Kettenhofen C, Nagashima K, English MA, Semler EM, Mahgerefteh J, Cideciyan AV, Li T, Brooks BP, Gunay-Aygun M, Jacobson SG, Cogliati T, Westlake CJ, Swaroop A
Cell reports 2017 Jul 11;20(2):384-396
Cell reports 2017 Jul 11;20(2):384-396
The CEP19-RABL2 GTPase Complex Binds IFT-B to Initiate Intraflagellar Transport at the Ciliary Base.
Kanie T, Abbott KL, Mooney NA, Plowey ED, Demeter J, Jackson PK
Developmental cell 2017 Jul 10;42(1):22-36.e12
Developmental cell 2017 Jul 10;42(1):22-36.e12
Arl3 and RP2 regulate the trafficking of ciliary tip kinesins.
Schwarz N, Lane A, Jovanovic K, Parfitt DA, Aguila M, Thompson CL, da Cruz L, Coffey PJ, Chapple JP, Hardcastle AJ, Cheetham ME
Human molecular genetics 2017 Jul 1;26(13):2480-2492
Human molecular genetics 2017 Jul 1;26(13):2480-2492
Primary Cilia in the Murine Cerebellum and in Mutant Models of Medulloblastoma.
Di Pietro C, Marazziti D, La Sala G, Abbaszadeh Z, Golini E, Matteoni R, Tocchini-Valentini GP
Cellular and molecular neurobiology 2017 Jan;37(1):145-154
Cellular and molecular neurobiology 2017 Jan;37(1):145-154
KIF13B establishes a CAV1-enriched microdomain at the ciliary transition zone to promote Sonic hedgehog signalling.
Schou KB, Mogensen JB, Morthorst SK, Nielsen BS, Aleliunaite A, Serra-Marques A, Fürstenberg N, Saunier S, Bizet AA, Veland IR, Akhmanova A, Christensen ST, Pedersen LB
Nature communications 2017 Jan 30;8:14177
Nature communications 2017 Jan 30;8:14177
Dynamic Remodeling of Membrane Composition Drives Cell Cycle through Primary Cilia Excision.
Phua SC, Chiba S, Suzuki M, Su E, Roberson EC, Pusapati GV, Schurmans S, Setou M, Rohatgi R, Reiter JF, Ikegami K, Inoue T
Cell 2017 Jan 12;168(1-2):264-279.e15
Cell 2017 Jan 12;168(1-2):264-279.e15
HDAC2 promotes loss of primary cilia in pancreatic ductal adenocarcinoma.
Kobayashi T, Nakazono K, Tokuda M, Mashima Y, Dynlacht BD, Itoh H
EMBO reports 2017 Feb;18(2):334-343
EMBO reports 2017 Feb;18(2):334-343
Zfp462 deficiency causes anxiety-like behaviors with excessive self-grooming in mice.
Wang B, Zheng Y, Shi H, Du X, Zhang Y, Wei B, Luo M, Wang H, Wu X, Hua X, Sun M, Xu X
Genes, brain, and behavior 2017 Feb;16(2):296-307
Genes, brain, and behavior 2017 Feb;16(2):296-307
Regulation of ciliary retrograde protein trafficking by the Joubert syndrome proteins ARL13B and INPP5E.
Nozaki S, Katoh Y, Terada M, Michisaka S, Funabashi T, Takahashi S, Kontani K, Nakayama K
Journal of cell science 2017 Feb 1;130(3):563-576
Journal of cell science 2017 Feb 1;130(3):563-576
A novel homozygous ARL13B variant in patients with Joubert syndrome impairs its guanine nucleotide-exchange factor activity.
Rafiullah R, Long AB, Ivanova AA, Ali H, Berkel S, Mustafa G, Paramasivam N, Schlesner M, Wiemann S, Wade RC, Bolthauser E, Blum M, Kahn RA, Caspary T, Rappold GA
European journal of human genetics : EJHG 2017 Dec;25(12):1324-1334
European journal of human genetics : EJHG 2017 Dec;25(12):1324-1334
Primary Cilium Formation and Ciliary Protein Trafficking Is Regulated by the Atypical MAP Kinase MAPK15 in Caenorhabditis elegans and Human Cells.
Kazatskaya A, Kuhns S, Lambacher NJ, Kennedy JE, Brear AG, McManus GJ, Sengupta P, Blacque OE
Genetics 2017 Dec;207(4):1423-1440
Genetics 2017 Dec;207(4):1423-1440
IFT proteins spatially control the geometry of cleavage furrow ingression and lumen positioning.
Taulet N, Vitre B, Anguille C, Douanier A, Rocancourt M, Taschner M, Lorentzen E, Echard A, Delaval B
Nature communications 2017 Dec 4;8(1):1928
Nature communications 2017 Dec 4;8(1):1928
An apicosome initiates self-organizing morphogenesis of human pluripotent stem cells.
Taniguchi K, Shao Y, Townshend RF, Cortez CL, Harris CE, Meshinchi S, Kalantry S, Fu J, O'Shea KS, Gumucio DL
The Journal of cell biology 2017 Dec 4;216(12):3981-3990
The Journal of cell biology 2017 Dec 4;216(12):3981-3990
The guanine nucleotide exchange factor Arf-like protein 13b is essential for assembly of the mouse photoreceptor transition zone and outer segment.
Hanke-Gogokhia C, Wu Z, Sharif A, Yazigi H, Frederick JM, Baehr W
The Journal of biological chemistry 2017 Dec 29;292(52):21442-21456
The Journal of biological chemistry 2017 Dec 29;292(52):21442-21456
Evolutionary Proteomics Uncovers Ancient Associations of Cilia with Signaling Pathways.
Sigg MA, Menchen T, Lee C, Johnson J, Jungnickel MK, Choksi SP, Garcia G 3rd, Busengdal H, Dougherty GW, Pennekamp P, Werner C, Rentzsch F, Florman HM, Krogan N, Wallingford JB, Omran H, Reiter JF
Developmental cell 2017 Dec 18;43(6):744-762.e11
Developmental cell 2017 Dec 18;43(6):744-762.e11
Eml1 loss impairs apical progenitor spindle length and soma shape in the developing cerebral cortex.
Bizzotto S, Uzquiano A, Dingli F, Ershov D, Houllier A, Arras G, Richards M, Loew D, Minc N, Croquelois A, Houdusse A, Francis F
Scientific reports 2017 Dec 11;7(1):17308
Scientific reports 2017 Dec 11;7(1):17308
A human patient-derived cellular model of Joubert syndrome reveals ciliary defects which can be rescued with targeted therapies.
Srivastava S, Ramsbottom SA, Molinari E, Alkanderi S, Filby A, White K, Henry C, Saunier S, Miles CG, Sayer JA
Human molecular genetics 2017 Dec 1;26(23):4657-4667
Human molecular genetics 2017 Dec 1;26(23):4657-4667
Folate-dependent methylation of septins governs ciliogenesis during neural tube closure.
Toriyama M, Toriyama M, Wallingford JB, Finnell RH
FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2017 Aug;31(8):3622-3635
FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2017 Aug;31(8):3622-3635
A novel role for primary cilia in airway remodeling.
Trempus CS, Song W, Lazrak A, Yu Z, Creighton JR, Young BM, Heise RL, Yu YR, Ingram JL, Tighe RM, Matalon S, Garantziotis S
American journal of physiology. Lung cellular and molecular physiology 2017 Aug 1;313(2):L328-L338
American journal of physiology. Lung cellular and molecular physiology 2017 Aug 1;313(2):L328-L338
Primary cilia on porcine testicular somatic cells and their role in hedgehog signaling and tubular morphogenesis in vitro.
Dores C, Alpaugh W, Su L, Biernaskie J, Dobrinski I
Cell and tissue research 2017 Apr;368(1):215-223
Cell and tissue research 2017 Apr;368(1):215-223
Estrogen receptor α is required for oviductal transport of embryos.
Li S, O'Neill SR, Zhang Y, Holtzman MJ, Takemaru KI, Korach KS, Winuthayanon W
FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2017 Apr;31(4):1595-1607
FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2017 Apr;31(4):1595-1607
Mutations in KIAA0753 cause Joubert syndrome associated with growth hormone deficiency.
Stephen J, Vilboux T, Mian L, Kuptanon C, Sinclair CM, Yildirimli D, Maynard DM, Bryant J, Fischer R, Vemulapalli M, Mullikin JC, NISC Comparative Sequencing Program, Huizing M, Gahl WA, Malicdan MCV, Gunay-Aygun M
Human genetics 2017 Apr;136(4):399-408
Human genetics 2017 Apr;136(4):399-408
KDM3A coordinates actin dynamics with intraflagellar transport to regulate cilia stability.
Yeyati PL, Schiller R, Mali G, Kasioulis I, Kawamura A, Adams IR, Playfoot C, Gilbert N, van Heyningen V, Wills J, von Kriegsheim A, Finch A, Sakai J, Schofield CJ, Jackson IJ, Mill P
The Journal of cell biology 2017 Apr 3;216(4):999-1013
The Journal of cell biology 2017 Apr 3;216(4):999-1013
Cilia-dependent GLI processing in neural crest cells is required for tongue development.
Millington G, Elliott KH, Chang YT, Chang CF, Dlugosz A, Brugmann SA
Developmental biology 2017 Apr 15;424(2):124-137
Developmental biology 2017 Apr 15;424(2):124-137
Generation of iPSC-derived Human Brain Organoids to Model Early Neurodevelopmental Disorders.
Gabriel E, Gopalakrishnan J
Journal of visualized experiments : JoVE 2017 Apr 14;(122)
Journal of visualized experiments : JoVE 2017 Apr 14;(122)
Practical method for targeted disruption of cilia-related genes by using CRISPR/Cas9-mediated, homology-independent knock-in system.
Katoh Y, Michisaka S, Nozaki S, Funabashi T, Hirano T, Takei R, Nakayama K
Molecular biology of the cell 2017 Apr 1;28(7):898-906
Molecular biology of the cell 2017 Apr 1;28(7):898-906
A novel ICK mutation causes ciliary disruption and lethal endocrine-cerebro-osteodysplasia syndrome.
Oud MM, Bonnard C, Mans DA, Altunoglu U, Tohari S, Ng AYJ, Eskin A, Lee H, Rupar CA, de Wagenaar NP, Wu KM, Lahiry P, Pazour GJ, Nelson SF, Hegele RA, Roepman R, Kayserili H, Venkatesh B, Siu VM, Reversade B, Arts HH
Cilia 2016;5:8
Cilia 2016;5:8
Measuring the primary cilium length: improved method for unbiased high-throughput analysis.
Dummer A, Poelma C, DeRuiter MC, Goumans MJ, Hierck BP
Cilia 2016;5:7
Cilia 2016;5:7
Three-dimensional retinal organoids from mouse pluripotent stem cells mimic in vivo development with enhanced stratification and rod photoreceptor differentiation.
Chen HY, Kaya KD, Dong L, Swaroop A
Molecular vision 2016;22:1077-1094
Molecular vision 2016;22:1077-1094
Morphological and Functional Characterization of the Ciliary Pocket by Electron and Fluorescence Microscopy.
Ghossoub R, Lindbæk L, Molla-Herman A, Schmitt A, Christensen ST, Benmerah A
Methods in molecular biology (Clifton, N.J.) 2016;1454:35-51
Methods in molecular biology (Clifton, N.J.) 2016;1454:35-51
Methods for Visualization of Neuronal Cilia.
Caspary T, Marazziti D, Berbari NF
Methods in molecular biology (Clifton, N.J.) 2016;1454:203-14
Methods in molecular biology (Clifton, N.J.) 2016;1454:203-14
Targeting of ASH Domain-Containing Proteins to the Centrosome.
Verdier P, Morthorst SK, Pedersen LB
Methods in molecular biology (Clifton, N.J.) 2016;1454:15-33
Methods in molecular biology (Clifton, N.J.) 2016;1454:15-33
Immunofluorescent staining of septins in primary cilia.
Kim MS, Froese CD, Xie H, Trimble WS
Methods in cell biology 2016;136:269-83
Methods in cell biology 2016;136:269-83
Primary Cilia Negatively Regulate Melanogenesis in Melanocytes and Pigmentation in a Human Skin Model.
Choi H, Shin JH, Kim ES, Park SJ, Bae IH, Jo YK, Jeong IY, Kim HJ, Lee Y, Park HC, Jeon HB, Kim KW, Lee TR, Cho DH
PloS one 2016;11(12):e0168025
PloS one 2016;11(12):e0168025
KIF3A binds to β-arrestin for suppressing Wnt/β-catenin signalling independently of primary cilia in lung cancer.
Kim M, Suh YA, Oh JH, Lee BR, Kim J, Jang SJ
Scientific reports 2016 Sep 6;6:32770
Scientific reports 2016 Sep 6;6:32770
Destabilization of the IFT-B cilia core complex due to mutations in IFT81 causes a Spectrum of Short-Rib Polydactyly Syndrome.
Duran I, Taylor SP, Zhang W, Martin J, Forlenza KN, Spiro RP, Nickerson DA, Bamshad M, Cohn DH, Krakow D
Scientific reports 2016 Sep 26;6:34232
Scientific reports 2016 Sep 26;6:34232
Nestin Mediates Hedgehog Pathway Tumorigenesis.
Li P, Lee EH, Du F, Gordon RE, Yuelling LW, Liu Y, Ng JM, Zhang H, Wu J, Korshunov A, Pfister SM, Curran T, Yang ZJ
Cancer research 2016 Sep 15;76(18):5573-83
Cancer research 2016 Sep 15;76(18):5573-83
RPGR, a prenylated retinal ciliopathy protein, is targeted to cilia in a prenylation- and PDE6D-dependent manner.
Dutta N, Seo S
Biology open 2016 Sep 15;5(9):1283-9
Biology open 2016 Sep 15;5(9):1283-9
IFT52 mutations destabilize anterograde complex assembly, disrupt ciliogenesis and result in short rib polydactyly syndrome.
Zhang W, Taylor SP, Nevarez L, Lachman RS, Nickerson DA, Bamshad M, University of Washington Center for Mendelian Genomics Consortium, Krakow D, Cohn DH
Human molecular genetics 2016 Sep 15;25(18):4012-4020
Human molecular genetics 2016 Sep 15;25(18):4012-4020
An inactivating mutation in intestinal cell kinase, ICK, impairs hedgehog signalling and causes short rib-polydactyly syndrome.
Paige Taylor S, Kunova Bosakova M, Varecha M, Balek L, Barta T, Trantirek L, Jelinkova I, Duran I, Vesela I, Forlenza KN, Martin JH, Hampl A, University of Washington Center for Mendelian Genomics, Bamshad M, Nickerson D, Jaworski ML, Song J, Ko HW, Cohn DH, Krakow D, Krejci P
Human molecular genetics 2016 Sep 15;25(18):3998-4011
Human molecular genetics 2016 Sep 15;25(18):3998-4011
Zfp423 Regulates Sonic Hedgehog Signaling via Primary Cilium Function.
Hong CJ, Hamilton BA
PLoS genetics 2016 Oct;12(10):e1006357
PLoS genetics 2016 Oct;12(10):e1006357
Loss of MACF1 Abolishes Ciliogenesis and Disrupts Apicobasal Polarity Establishment in the Retina.
May-Simera HL, Gumerson JD, Gao C, Campos M, Cologna SM, Beyer T, Boldt K, Kaya KD, Patel N, Kretschmer F, Kelley MW, Petralia RS, Davey MG, Li T
Cell reports 2016 Oct 25;17(5):1399-1413
Cell reports 2016 Oct 25;17(5):1399-1413
Prenylated retinal ciliopathy protein RPGR interacts with PDE6δ and regulates ciliary localization of Joubert syndrome-associated protein INPP5E.
Rao KN, Zhang W, Li L, Anand M, Khanna H
Human molecular genetics 2016 Oct 15;25(20):4533-4545
Human molecular genetics 2016 Oct 15;25(20):4533-4545
Craniofacial Ciliopathies Reveal Specific Requirements for GLI Proteins during Development of the Facial Midline.
Chang CF, Chang YT, Millington G, Brugmann SA
PLoS genetics 2016 Nov;12(11):e1006351
PLoS genetics 2016 Nov;12(11):e1006351
Defective ciliogenesis in thyroid hürthle cell tumors is associated with increased autophagy.
Lee J, Yi S, Kang YE, Chang JY, Kim JT, Sul HJ, Kim JO, Kim JM, Kim J, Porcelli AM, Kim KS, Shong M
Oncotarget 2016 Nov 29;7(48):79117-79130
Oncotarget 2016 Nov 29;7(48):79117-79130
Neuroanatomical characterization of the cellular and axonal architecture of subcortical band heterotopia in the BXD29-Tlr4(lps-2J)/J mouse cortex.
Ramos RL, Toia AR, Pasternack DM, Dotzler TP, Cuoco JA, Esposito AW, Le MM, Parker AK, Goodman JH, Sarkisian MR
Neuroscience 2016 Nov 19;337:48-65
Neuroscience 2016 Nov 19;337:48-65
Role for the IFT-A Complex in Selective Transport to the Primary Cilium.
Fu W, Wang L, Kim S, Li J, Dynlacht BD
Cell reports 2016 Nov 1;17(6):1505-1517
Cell reports 2016 Nov 1;17(6):1505-1517
Cyclic expression of the voltage-gated potassium channel KV10.1 promotes disassembly of the primary cilium.
Sánchez A, Urrego D, Pardo LA
EMBO reports 2016 May;17(5):708-23
EMBO reports 2016 May;17(5):708-23
An organelle-specific protein landscape identifies novel diseases and molecular mechanisms.
Boldt K, van Reeuwijk J, Lu Q, Koutroumpas K, Nguyen TM, Texier Y, van Beersum SE, Horn N, Willer JR, Mans DA, Dougherty G, Lamers IJ, Coene KL, Arts HH, Betts MJ, Beyer T, Bolat E, Gloeckner CJ, Haidari K, Hetterschijt L, Iaconis D, Jenkins D, Klose F, Knapp B, Latour B, Letteboer SJ, Marcelis CL, Mitic D, Morleo M, Oud MM, Riemersma M, Rix S, Terhal PA, Toedt G, van Dam TJ, de Vrieze E, Wissinger Y, Wu KM, Apic G, Beales PL, Blacque OE, Gibson TJ, Huynen MA, Katsanis N, Kremer H, Omran H, van Wijk E, Wolfrum U, Kepes F, Davis EE, Franco B, Giles RH, Ueffing M, Russell RB, Roepman R, UK10K Rare Diseases Group
Nature communications 2016 May 13;7:11491
Nature communications 2016 May 13;7:11491
Recessive NEK9 mutation causes a lethal skeletal dysplasia with evidence of cell cycle and ciliary defects.
Casey JP, Brennan K, Scheidel N, McGettigan P, Lavin PT, Carter S, Ennis S, Dorkins H, Ghali N, Blacque OE, Mc Gee MM, Murphy H, Lynch SA
Human molecular genetics 2016 May 1;25(9):1824-35
Human molecular genetics 2016 May 1;25(9):1824-35
Centrosomal protein CP110 controls maturation of the mother centriole during cilia biogenesis.
Yadav SP, Sharma NK, Liu C, Dong L, Li T, Swaroop A
Development (Cambridge, England) 2016 May 1;143(9):1491-501
Development (Cambridge, England) 2016 May 1;143(9):1491-501
Arf-like Protein 3 (ARL3) Regulates Protein Trafficking and Ciliogenesis in Mouse Photoreceptors.
Hanke-Gogokhia C, Wu Z, Gerstner CD, Frederick JM, Zhang H, Baehr W
The Journal of biological chemistry 2016 Mar 25;291(13):7142-55
The Journal of biological chemistry 2016 Mar 25;291(13):7142-55
MCRS1 associates with cytoplasmic dynein and mediates pericentrosomal material recruitment.
Lee SH, Lee MS, Choi TI, Hong H, Seo JY, Kim CH, Kim J
Scientific reports 2016 Jun 6;6:27284
Scientific reports 2016 Jun 6;6:27284
Identification and Correction of Mechanisms Underlying Inherited Blindness in Human iPSC-Derived Optic Cups.
Parfitt DA, Lane A, Ramsden CM, Carr AF, Munro PM, Jovanovic K, Schwarz N, Kanuga N, Muthiah MN, Hull S, Gallo JM, da Cruz L, Moore AT, Hardcastle AJ, Coffey PJ, Cheetham ME
Cell stem cell 2016 Jun 2;18(6):769-781
Cell stem cell 2016 Jun 2;18(6):769-781
Hedgehog signaling promotes basal progenitor expansion and the growth and folding of the neocortex.
Wang L, Hou S, Han YG
Nature neuroscience 2016 Jul;19(7):888-96
Nature neuroscience 2016 Jul;19(7):888-96
MKS1 regulates ciliary INPP5E levels in Joubert syndrome.
Slaats GG, Isabella CR, Kroes HY, Dempsey JC, Gremmels H, Monroe GR, Phelps IG, Duran KJ, Adkins J, Kumar SA, Knutzen DM, Knoers NV, Mendelsohn NJ, Neubauer D, Mastroyianni SD, Vogt J, Worgan L, Karp N, Bowdin S, Glass IA, Parisi MA, Otto EA, Johnson CA, Hildebrandt F, van Haaften G, Giles RH, Doherty D
Journal of medical genetics 2016 Jan;53(1):62-72
Journal of medical genetics 2016 Jan;53(1):62-72
TMEM107 recruits ciliopathy proteins to subdomains of the ciliary transition zone and causes Joubert syndrome.
Lambacher NJ, Bruel AL, van Dam TJ, Szymańska K, Slaats GG, Kuhns S, McManus GJ, Kennedy JE, Gaff K, Wu KM, van der Lee R, Burglen L, Doummar D, Rivière JB, Faivre L, Attié-Bitach T, Saunier S, Curd A, Peckham M, Giles RH, Johnson CA, Huynen MA, Thauvin-Robinet C, Blacque OE
Nature cell biology 2016 Jan;18(1):122-31
Nature cell biology 2016 Jan;18(1):122-31
Arl13b and the exocyst interact synergistically in ciliogenesis.
Seixas C, Choi SY, Polgar N, Umberger NL, East MP, Zuo X, Moreiras H, Ghossoub R, Benmerah A, Kahn RA, Fogelgren B, Caspary T, Lipschutz JH, Barral DC
Molecular biology of the cell 2016 Jan 15;27(2):308-20
Molecular biology of the cell 2016 Jan 15;27(2):308-20
Lithium chloride modulates chondrocyte primary cilia and inhibits Hedgehog signaling.
Thompson CL, Wiles A, Poole CA, Knight MM
FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2016 Feb;30(2):716-26
FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2016 Feb;30(2):716-26
NudC regulates actin dynamics and ciliogenesis by stabilizing cofilin 1.
Zhang C, Zhang W, Lu Y, Yan X, Yan X, Zhu X, Liu W, Yang Y, Zhou T
Cell research 2016 Feb;26(2):239-53
Cell research 2016 Feb;26(2):239-53
Endothelial primary cilia inhibit atherosclerosis.
Dinsmore C, Reiter JF
EMBO reports 2016 Feb;17(2):156-66
EMBO reports 2016 Feb;17(2):156-66
Disruption of KIF3A in patient-derived glioblastoma cells: effects on ciliogenesis, hedgehog sensitivity, and tumorigenesis.
Hoang-Minh LB, Deleyrolle LP, Siebzehnrubl D, Ugartemendia G, Futch H, Griffith B, Breunig JJ, De Leon G, Mitchell DA, Semple-Rowland S, Reynolds BA, Sarkisian MR
Oncotarget 2016 Feb 9;7(6):7029-43
Oncotarget 2016 Feb 9;7(6):7029-43
WDR8 is a centriolar satellite and centriole-associated protein that promotes ciliary vesicle docking during ciliogenesis.
Kurtulmus B, Wang W, Ruppert T, Neuner A, Cerikan B, Viol L, Dueñas-Sánchez R, Gruss OJ, Pereira G
Journal of cell science 2016 Feb 1;129(3):621-36
Journal of cell science 2016 Feb 1;129(3):621-36
Deletion of ADP Ribosylation Factor-Like GTPase 13B Leads to Kidney Cysts.
Li Y, Tian X, Ma M, Jerman S, Kong S, Somlo S, Sun Z
Journal of the American Society of Nephrology : JASN 2016 Dec;27(12):3628-3638
Journal of the American Society of Nephrology : JASN 2016 Dec;27(12):3628-3638
Airway Epithelial KIF3A Regulates Th2 Responses to Aeroallergens.
Giridhar PV, Bell SM, Sridharan A, Rajavelu P, Kitzmiller JA, Na CL, Kofron M, Brandt EB, Ericksen M, Naren AP, Moon C, Khurana Hershey GK, Whitsett JA
Journal of immunology (Baltimore, Md. : 1950) 2016 Dec 1;197(11):4228-4239
Journal of immunology (Baltimore, Md. : 1950) 2016 Dec 1;197(11):4228-4239
Neonatal seizures induced by pentylenetetrazol or kainic acid disrupt primary cilia growth on developing mouse cortical neurons.
Parker AK, Le MM, Smith TS, Hoang-Minh LB, Atkinson EW, Ugartemendia G, Semple-Rowland S, Coleman JE, Sarkisian MR
Experimental neurology 2016 Aug;282:119-27
Experimental neurology 2016 Aug;282:119-27
Severe NDE1-mediated microcephaly results from neural progenitor cell cycle arrests at multiple specific stages.
Doobin DJ, Kemal S, Dantas TJ, Vallee RB
Nature communications 2016 Aug 24;7:12551
Nature communications 2016 Aug 24;7:12551
Arl13b Interacts With Vangl2 to Regulate Cilia and Photoreceptor Outer Segment Length in Zebrafish.
Song P, Dudinsky L, Fogerty J, Gaivin R, Perkins BD
Investigative ophthalmology & visual science 2016 Aug 1;57(10):4517-26
Investigative ophthalmology & visual science 2016 Aug 1;57(10):4517-26
Sonic Hedgehog promotes proliferation of Notch-dependent monociliated choroid plexus tumour cells.
Li L, Grausam KB, Wang J, Lun MP, Ohli J, Lidov HG, Calicchio ML, Zeng E, Salisbury JL, Wechsler-Reya RJ, Lehtinen MK, Schüller U, Zhao H
Nature cell biology 2016 Apr;18(4):418-30
Nature cell biology 2016 Apr;18(4):418-30
CPAP promotes timely cilium disassembly to maintain neural progenitor pool.
Gabriel E, Wason A, Ramani A, Gooi LM, Keller P, Pozniakovsky A, Poser I, Noack F, Telugu NS, Calegari F, Šarić T, Hescheler J, Hyman AA, Gottardo M, Callaini G, Alkuraya FS, Gopalakrishnan J
The EMBO journal 2016 Apr 15;35(8):803-19
The EMBO journal 2016 Apr 15;35(8):803-19
Culture and detection of primary cilia in endothelial cell models.
Lim YC, McGlashan SR, Cooling MT, Long DS
Cilia 2015;4:11
Cilia 2015;4:11
Analysis of primary cilia in the developing mouse brain.
Paridaen JT, Huttner WB, Wilsch-Bräuninger M
Methods in cell biology 2015;127:93-129
Methods in cell biology 2015;127:93-129
Imaging centrosomes and cilia in the mouse kidney.
Hoshi M, Wang J, Jain S, Mahjoub MR
Methods in cell biology 2015;127:1-17
Methods in cell biology 2015;127:1-17
A Group of ent-Kaurane Diterpenoids Inhibit Hedgehog Signaling and Induce Cilia Elongation.
Jiang S, Du J, Kong Q, Li C, Li Y, Sun H, Pu J, Mao B
PloS one 2015;10(10):e0139830
PloS one 2015;10(10):e0139830
Cell Cycle-Dependent Localization of Dynactin Subunit p150 glued at Centrosome.
Chen TY, Syu JS, Han TY, Cheng HL, Lu FI, Wang CY
Journal of cellular biochemistry 2015 Sep;116(9):2049-60
Journal of cellular biochemistry 2015 Sep;116(9):2049-60
Primary Cilia on Horizontal Basal Cells Regulate Regeneration of the Olfactory Epithelium.
Joiner AM, Green WW, McIntyre JC, Allen BL, Schwob JE, Martens JR
The Journal of neuroscience : the official journal of the Society for Neuroscience 2015 Oct 7;35(40):13761-72
The Journal of neuroscience : the official journal of the Society for Neuroscience 2015 Oct 7;35(40):13761-72
Mutations in TRAF3IP1/IFT54 reveal a new role for IFT proteins in microtubule stabilization.
Bizet AA, Becker-Heck A, Ryan R, Weber K, Filhol E, Krug P, Halbritter J, Delous M, Lasbennes MC, Linghu B, Oakeley EJ, Zarhrate M, Nitschké P, Garfa-Traore M, Serluca F, Yang F, Bouwmeester T, Pinson L, Cassuto E, Dubot P, Elshakhs NAS, Sahel JA, Salomon R, Drummond IA, Gubler MC, Antignac C, Chibout S, Szustakowski JD, Hildebrandt F, Lorentzen E, Sailer AW, Benmerah A, Saint-Mezard P, Saunier S
Nature communications 2015 Oct 21;6:8666
Nature communications 2015 Oct 21;6:8666
PDGFRβ and oncogenic mutant PDGFRα D842V promote disassembly of primary cilia through a PLCγ- and AURKA-dependent mechanism.
Nielsen BS, Malinda RR, Schmid FM, Pedersen SF, Christensen ST, Pedersen LB
Journal of cell science 2015 Oct 1;128(19):3543-9
Journal of cell science 2015 Oct 1;128(19):3543-9
Conserved Genetic Interactions between Ciliopathy Complexes Cooperatively Support Ciliogenesis and Ciliary Signaling.
Yee LE, Garcia-Gonzalo FR, Bowie RV, Li C, Kennedy JK, Ashrafi K, Blacque OE, Leroux MR, Reiter JF
PLoS genetics 2015 Nov;11(11):e1005627
PLoS genetics 2015 Nov;11(11):e1005627
The Interaction of CCDC104/BARTL1 with Arl3 and Implications for Ciliary Function.
Lokaj M, Kösling SK, Koerner C, Lange SM, van Beersum SE, van Reeuwijk J, Roepman R, Horn N, Ueffing M, Boldt K, Wittinghofer A
Structure (London, England : 1993) 2015 Nov 3;23(11):2122-32
Structure (London, England : 1993) 2015 Nov 3;23(11):2122-32
Loss of primary cilia in melanoma cells is likely independent of proliferation and cell cycle progression.
Snedecor ER, Sung CC, Moncayo A, Rothstein BE, Mockler DC, Tonnesen MG, Jones EC, Fujita M, Clark RA, Shroyer KR, Chen J
The Journal of investigative dermatology 2015 May;135(5):1456-1458
The Journal of investigative dermatology 2015 May;135(5):1456-1458
Myosin heavy chain 10 (MYH10) is required for centriole migration during the biogenesis of primary cilia.
Hong H, Kim J, Kim J
Biochemical and biophysical research communications 2015 May 22;461(1):180-5
Biochemical and biophysical research communications 2015 May 22;461(1):180-5
Cell context-specific expression of primary cilia in the human testis and ciliary coordination of Hedgehog signalling in mouse Leydig cells.
Nygaard MB, Almstrup K, Lindbæk L, Christensen ST, Svingen T
Scientific reports 2015 May 20;5:10364
Scientific reports 2015 May 20;5:10364
Centrin2 regulates CP110 removal in primary cilium formation.
Prosser SL, Morrison CG
The Journal of cell biology 2015 Mar 16;208(6):693-701
The Journal of cell biology 2015 Mar 16;208(6):693-701
The heterotrimeric kinesin-2 complex interacts with and regulates GLI protein function.
Carpenter BS, Barry RL, Verhey KJ, Allen BL
Journal of cell science 2015 Mar 1;128(5):1034-50
Journal of cell science 2015 Mar 1;128(5):1034-50
Mutations in DYNC2LI1 disrupt cilia function and cause short rib polydactyly syndrome.
Taylor SP, Dantas TJ, Duran I, Wu S, Lachman RS, University of Washington Center for Mendelian Genomics Consortium, Nelson SF, Cohn DH, Vallee RB, Krakow D
Nature communications 2015 Jun 16;6:7092
Nature communications 2015 Jun 16;6:7092
Ablation of retinal ciliopathy protein RPGR results in altered photoreceptor ciliary composition.
Rao KN, Li L, Anand M, Khanna H
Scientific reports 2015 Jun 11;5:11137
Scientific reports 2015 Jun 11;5:11137
Non-Overlapping Distributions and Functions of the VDAC Family in Ciliogenesis.
Majumder S, Cash A, Fisk HA
Cells 2015 Jul 31;4(3):331-53
Cells 2015 Jul 31;4(3):331-53
A function for the Joubert syndrome protein Arl13b in ciliary membrane extension and ciliary length regulation.
Lu H, Toh MT, Narasimhan V, Thamilselvam SK, Choksi SP, Roy S
Developmental biology 2015 Jan 15;397(2):225-36
Developmental biology 2015 Jan 15;397(2):225-36
Polycystin-1 maturation requires polycystin-2 in a dose-dependent manner.
Gainullin VG, Hopp K, Ward CJ, Hommerding CJ, Harris PC
The Journal of clinical investigation 2015 Feb;125(2):607-20
The Journal of clinical investigation 2015 Feb;125(2):607-20
The Microtubule-Depolymerizing Activity of a Mitotic Kinesin Protein KIF2A Drives Primary Cilia Disassembly Coupled with Cell Proliferation.
Miyamoto T, Hosoba K, Ochiai H, Royba E, Izumi H, Sakuma T, Yamamoto T, Dynlacht BD, Matsuura S
Cell reports 2015 Feb 10;10(5):664-673
Cell reports 2015 Feb 10;10(5):664-673
Mutations in human homologue of chicken talpid3 gene (KIAA0586) cause a hybrid ciliopathy with overlapping features of Jeune and Joubert syndromes.
Malicdan MC, Vilboux T, Stephen J, Maglic D, Mian L, Konzman D, Guo J, Yildirimli D, Bryant J, Fischer R, Zein WM, Snow J, Vemulapalli M, Mullikin JC, Toro C, Solomon BD, Niederhuber JE, NISC Comparative Sequencing Program, Gahl WA, Gunay-Aygun M
Journal of medical genetics 2015 Dec;52(12):830-9
Journal of medical genetics 2015 Dec;52(12):830-9
The Orphan G Protein-coupled Receptor Gpr175 (Tpra40) Enhances Hedgehog Signaling by Modulating cAMP Levels.
Singh J, Wen X, Scales SJ
The Journal of biological chemistry 2015 Dec 4;290(49):29663-75
The Journal of biological chemistry 2015 Dec 4;290(49):29663-75
An siRNA-based functional genomics screen for the identification of regulators of ciliogenesis and ciliopathy genes.
Wheway G, Schmidts M, Mans DA, Szymanska K, Nguyen TT, Racher H, Phelps IG, Toedt G, Kennedy J, Wunderlich KA, Sorusch N, Abdelhamed ZA, Natarajan S, Herridge W, van Reeuwijk J, Horn N, Boldt K, Parry DA, Letteboer SJF, Roosing S, Adams M, Bell SM, Bond J, Higgins J, Morrison EE, Tomlinson DC, Slaats GG, van Dam TJP, Huang L, Kessler K, Giessl A, Logan CV, Boyle EA, Shendure J, Anazi S, Aldahmesh M, Al Hazzaa S, Hegele RA, Ober C, Frosk P, Mhanni AA, Chodirker BN, Chudley AE, Lamont R, Bernier FP, Beaulieu CL, Gordon P, Pon RT, Donahue C, Barkovich AJ, Wolf L, Toomes C, Thiel CT, Boycott KM, McKibbin M, Inglehearn CF, UK10K Consortium, University of Washington Center for Mendelian Genomics, Stewart F, Omran H, Huynen MA, Sergouniotis PI, Alkuraya FS, Parboosingh JS, Innes AM, Willoughby CE, Giles RH, Webster AR, Ueffing M, Blacque O, Gleeson JG, Wolfrum U, Beales PL, Gibson T, Doherty D, Mitchison HM, Roepman R, Johnson CA
Nature cell biology 2015 Aug;17(8):1074-1087
Nature cell biology 2015 Aug;17(8):1074-1087
A role for Rab23 in the trafficking of Kif17 to the primary cilium.
Lim YS, Tang BL
Journal of cell science 2015 Aug 15;128(16):2996-3008
Journal of cell science 2015 Aug 15;128(16):2996-3008
Accumulation of non-outer segment proteins in the outer segment underlies photoreceptor degeneration in Bardet-Biedl syndrome.
Datta P, Allamargot C, Hudson JS, Andersen EK, Bhattarai S, Drack AV, Sheffield VC, Seo S
Proceedings of the National Academy of Sciences of the United States of America 2015 Aug 11;112(32):E4400-9
Proceedings of the National Academy of Sciences of the United States of America 2015 Aug 11;112(32):E4400-9
BBS4 is necessary for ciliary localization of TrkB receptor and activation by BDNF.
Leitch CC, Zaghloul NA
PloS one 2014;9(5):e98687
PloS one 2014;9(5):e98687
Centrosome-kinase fusions promote oncogenic signaling and disrupt centrosome function in myeloproliferative neoplasms.
Lee JY, Hong WJ, Majeti R, Stearns T
PloS one 2014;9(3):e92641
PloS one 2014;9(3):e92641
Prostaglandin signalling regulates ciliogenesis by modulating intraflagellar transport.
Jin D, Ni TT, Sun J, Wan H, Amack JD, Yu G, Fleming J, Chiang C, Li W, Papierniak A, Cheepala S, Conseil G, Cole SP, Zhou B, Drummond IA, Schuetz JD, Malicki J, Zhong TP
Nature cell biology 2014 Sep;16(9):841-51
Nature cell biology 2014 Sep;16(9):841-51
Centriole maturation requires regulated Plk1 activity during two consecutive cell cycles.
Kong D, Farmer V, Shukla A, James J, Gruskin R, Kiriyama S, Loncarek J
The Journal of cell biology 2014 Sep 29;206(7):855-65
The Journal of cell biology 2014 Sep 29;206(7):855-65
A mutation in the mouse ttc26 gene leads to impaired hedgehog signaling.
Swiderski RE, Nakano Y, Mullins RF, Seo S, Bánfi B
PLoS genetics 2014 Oct;10(10):e1004689
PLoS genetics 2014 Oct;10(10):e1004689
Primary cilia in the developing pig testis.
Ou Y, Dores C, Rodriguez-Sosa JR, van der Hoorn FA, Dobrinski I
Cell and tissue research 2014 Nov;358(2):597-605
Cell and tissue research 2014 Nov;358(2):597-605
A homozygous missense mutation in the ciliary gene TTC21B causes familial FSGS.
Huynh Cong E, Bizet AA, Boyer O, Woerner S, Gribouval O, Filhol E, Arrondel C, Thomas S, Silbermann F, Canaud G, Hachicha J, Ben Dhia N, Peraldi MN, Harzallah K, Iftene D, Daniel L, Willems M, Noel LH, Bole-Feysot C, Nitschké P, Gubler MC, Mollet G, Saunier S, Antignac C
Journal of the American Society of Nephrology : JASN 2014 Nov;25(11):2435-43
Journal of the American Society of Nephrology : JASN 2014 Nov;25(11):2435-43
Ciliary membrane proteins traffic through the Golgi via a Rabep1/GGA1/Arl3-dependent mechanism.
Kim H, Xu H, Yao Q, Li W, Huang Q, Outeda P, Cebotaru V, Chiaravalli M, Boletta A, Piontek K, Germino GG, Weinman EJ, Watnick T, Qian F
Nature communications 2014 Nov 18;5:5482
Nature communications 2014 Nov 18;5:5482
RFX7 is required for the formation of cilia in the neural tube.
Manojlovic Z, Earwood R, Kato A, Stefanovic B, Kato Y
Mechanisms of development 2014 May;132:28-37
Mechanisms of development 2014 May;132:28-37
Hypomorphism for RPGRIP1L, a ciliary gene vicinal to the FTO locus, causes increased adiposity in mice.
Stratigopoulos G, Martin Carli JF, O'Day DR, Wang L, Leduc CA, Lanzano P, Chung WK, Rosenbaum M, Egli D, Doherty DA, Leibel RL
Cell metabolism 2014 May 6;19(5):767-79
Cell metabolism 2014 May 6;19(5):767-79
Primary cilia disassembly down-regulates mechanosensitive hedgehog signalling: a feedback mechanism controlling ADAMTS-5 expression in chondrocytes.
Thompson CL, Chapple JP, Knight MM
Osteoarthritis and cartilage 2014 Mar;22(3):490-8
Osteoarthritis and cartilage 2014 Mar;22(3):490-8
SIRT2 regulates ciliogenesis and contributes to abnormal centrosome amplification caused by loss of polycystin-1.
Zhou X, Fan LX, Li K, Ramchandran R, Calvet JP, Li X
Human molecular genetics 2014 Mar 15;23(6):1644-55
Human molecular genetics 2014 Mar 15;23(6):1644-55
Ciliopathy-associated gene Cc2d2a promotes assembly of subdistal appendages on the mother centriole during cilia biogenesis.
Veleri S, Manjunath SH, Fariss RN, May-Simera H, Brooks M, Foskett TA, Gao C, Longo TA, Liu P, Nagashima K, Rachel RA, Li T, Dong L, Swaroop A
Nature communications 2014 Jun 20;5:4207
Nature communications 2014 Jun 20;5:4207
ICK is essential for cell type-specific ciliogenesis and the regulation of ciliary transport.
Chaya T, Omori Y, Kuwahara R, Furukawa T
The EMBO journal 2014 Jun 2;33(11):1227-42
The EMBO journal 2014 Jun 2;33(11):1227-42
Α-tubulin K40 acetylation is required for contact inhibition of proliferation and cell-substrate adhesion.
Aguilar A, Becker L, Tedeschi T, Heller S, Iomini C, Nachury MV
Molecular biology of the cell 2014 Jun 15;25(12):1854-66
Molecular biology of the cell 2014 Jun 15;25(12):1854-66
Basal body proteins regulate Notch signaling through endosomal trafficking.
Leitch CC, Lodh S, Prieto-Echagüe V, Badano JL, Zaghloul NA
Journal of cell science 2014 Jun 1;127(Pt 11):2407-19
Journal of cell science 2014 Jun 1;127(Pt 11):2407-19
Specific polar subpopulations of astral microtubules control spindle orientation and symmetric neural stem cell division.
Mora-Bermúdez F, Matsuzaki F, Huttner WB
eLife 2014 Jul 4;3
eLife 2014 Jul 4;3
Arhgap36-dependent activation of Gli transcription factors.
Rack PG, Ni J, Payumo AY, Nguyen V, Crapster JA, Hovestadt V, Kool M, Jones DT, Mich JK, Firestone AJ, Pfister SM, Cho YJ, Chen JK
Proceedings of the National Academy of Sciences of the United States of America 2014 Jul 29;111(30):11061-6
Proceedings of the National Academy of Sciences of the United States of America 2014 Jul 29;111(30):11061-6
Cep164 triggers ciliogenesis by recruiting Tau tubulin kinase 2 to the mother centriole.
Čajánek L, Nigg EA
Proceedings of the National Academy of Sciences of the United States of America 2014 Jul 15;111(28):E2841-50
Proceedings of the National Academy of Sciences of the United States of America 2014 Jul 15;111(28):E2841-50
Disruption of the basal body protein POC1B results in autosomal-recessive cone-rod dystrophy.
Roosing S, Lamers IJ, de Vrieze E, van den Born LI, Lambertus S, Arts HH, POC1B Study Group, Peters TA, Hoyng CB, Kremer H, Hetterschijt L, Letteboer SJ, van Wijk E, Roepman R, den Hollander AI, Cremers FP
American journal of human genetics 2014 Aug 7;95(2):131-42
American journal of human genetics 2014 Aug 7;95(2):131-42
The master cell cycle regulator APC-Cdc20 regulates ciliary length and disassembly of the primary cilium.
Wang W, Wu T, Kirschner MW
eLife 2014 Aug 19;3:e03083
eLife 2014 Aug 19;3:e03083
The ciliary protein Ftm is required for ventricular wall and septal development.
Gerhardt C, Lier JM, Kuschel S, Rüther U
PloS one 2013;8(2):e57545
PloS one 2013;8(2):e57545
Disruption of CEP290 microtubule/membrane-binding domains causes retinal degeneration.
Drivas TG, Holzbaur EL, Bennett J
The Journal of clinical investigation 2013 Oct;123(10):4525-39
The Journal of clinical investigation 2013 Oct;123(10):4525-39
Asymmetric inheritance of centrosome-associated primary cilium membrane directs ciliogenesis after cell division.
Paridaen JT, Wilsch-Bräuninger M, Huttner WB
Cell 2013 Oct 10;155(2):333-44
Cell 2013 Oct 10;155(2):333-44
Genetically encoded calcium indicator illuminates calcium dynamics in primary cilia.
Su S, Phua SC, DeRose R, Chiba S, Narita K, Kalugin PN, Katada T, Kontani K, Takeda S, Inoue T
Nature methods 2013 Nov;10(11):1105-7
Nature methods 2013 Nov;10(11):1105-7
Defects in the IFT-B component IFT172 cause Jeune and Mainzer-Saldino syndromes in humans.
Halbritter J, Bizet AA, Schmidts M, Porath JD, Braun DA, Gee HY, McInerney-Leo AM, Krug P, Filhol E, Davis EE, Airik R, Czarnecki PG, Lehman AM, Trnka P, Nitschké P, Bole-Feysot C, Schueler M, Knebelmann B, Burtey S, Szabó AJ, Tory K, Leo PJ, Gardiner B, McKenzie FA, Zankl A, Brown MA, Hartley JL, Maher ER, Li C, Leroux MR, Scambler PJ, Zhan SH, Jones SJ, Kayserili H, Tuysuz B, Moorani KN, Constantinescu A, Krantz ID, Kaplan BS, Shah JV, UK10K Consortium, Hurd TW, Doherty D, Katsanis N, Duncan EL, Otto EA, Beales PL, Mitchison HM, Saunier S, Hildebrandt F
American journal of human genetics 2013 Nov 7;93(5):915-25
American journal of human genetics 2013 Nov 7;93(5):915-25
Reduction of oxidative stress during recovery accelerates normalization of primary cilia length that is altered after ischemic injury in murine kidneys.
Kim JI, Kim J, Jang HS, Noh MR, Lipschutz JH, Park KM
American journal of physiology. Renal physiology 2013 May 15;304(10):F1283-94
American journal of physiology. Renal physiology 2013 May 15;304(10):F1283-94
Visualizing renal primary cilia.
Deane JA, Verghese E, Martelotto LG, Cain JE, Galtseva A, Rosenblum ND, Watkins DN, Ricardo SD
Nephrology (Carlton, Vic.) 2013 Mar;18(3):161-8
Nephrology (Carlton, Vic.) 2013 Mar;18(3):161-8
VDAC3 and Mps1 negatively regulate ciliogenesis.
Majumder S, Fisk HA
Cell cycle (Georgetown, Tex.) 2013 Mar 1;12(5):849-58
Cell cycle (Georgetown, Tex.) 2013 Mar 1;12(5):849-58
Prickle1 is expressed in distinct cell populations of the central nervous system and contributes to neuronal morphogenesis.
Liu C, Lin C, Whitaker DT, Bakeri H, Bulgakov OV, Liu P, Lei J, Dong L, Li T, Swaroop A
Human molecular genetics 2013 Jun 1;22(11):2234-46
Human molecular genetics 2013 Jun 1;22(11):2234-46
The ciliary G-protein-coupled receptor Gpr161 negatively regulates the Sonic hedgehog pathway via cAMP signaling.
Mukhopadhyay S, Wen X, Ratti N, Loktev A, Rangell L, Scales SJ, Jackson PK
Cell 2013 Jan 17;152(1-2):210-23
Cell 2013 Jan 17;152(1-2):210-23
The microtubule affinity regulating kinase MARK4 promotes axoneme extension during early ciliogenesis.
Kuhns S, Schmidt KN, Reymann J, Gilbert DF, Neuner A, Hub B, Carvalho R, Wiedemann P, Zentgraf H, Erfle H, Klingmüller U, Boutros M, Pereira G
The Journal of cell biology 2013 Feb 18;200(4):505-22
The Journal of cell biology 2013 Feb 18;200(4):505-22
Mutations in ARL2BP, encoding ADP-ribosylation-factor-like 2 binding protein, cause autosomal-recessive retinitis pigmentosa.
Davidson AE, Schwarz N, Zelinger L, Stern-Schneider G, Shoemark A, Spitzbarth B, Gross M, Laxer U, Sosna J, Sergouniotis PI, Waseem NH, Wilson R, Kahn RA, Plagnol V, Wolfrum U, Banin E, Hardcastle AJ, Cheetham ME, Sharon D, Webster AR
American journal of human genetics 2013 Aug 8;93(2):321-9
American journal of human genetics 2013 Aug 8;93(2):321-9
Molecular basis of tubulin transport within the cilium by IFT74 and IFT81.
Bhogaraju S, Cajanek L, Fort C, Blisnick T, Weber K, Taschner M, Mizuno N, Lamla S, Bastin P, Nigg EA, Lorentzen E
Science (New York, N.Y.) 2013 Aug 30;341(6149):1009-12
Science (New York, N.Y.) 2013 Aug 30;341(6149):1009-12
CCDC41 is required for ciliary vesicle docking to the mother centriole.
Joo K, Kim CG, Lee MS, Moon HY, Lee SH, Kim MJ, Kweon HS, Park WY, Kim CH, Gleeson JG, Kim J
Proceedings of the National Academy of Sciences of the United States of America 2013 Apr 9;110(15):5987-92
Proceedings of the National Academy of Sciences of the United States of America 2013 Apr 9;110(15):5987-92
ARL13B, PDE6D, and CEP164 form a functional network for INPP5E ciliary targeting.
Humbert MC, Weihbrecht K, Searby CC, Li Y, Pope RM, Sheffield VC, Seo S
Proceedings of the National Academy of Sciences of the United States of America 2012 Nov 27;109(48):19691-6
Proceedings of the National Academy of Sciences of the United States of America 2012 Nov 27;109(48):19691-6
SUMOylation of the small GTPase ARL-13 promotes ciliary targeting of sensory receptors.
Li Y, Zhang Q, Wei Q, Zhang Y, Ling K, Hu J
The Journal of cell biology 2012 Nov 12;199(4):589-98
The Journal of cell biology 2012 Nov 12;199(4):589-98
Basolateral rather than apical primary cilia on neuroepithelial cells committed to delamination.
Wilsch-Bräuninger M, Peters J, Paridaen JT, Huttner WB
Development (Cambridge, England) 2012 Jan;139(1):95-105
Development (Cambridge, England) 2012 Jan;139(1):95-105
A novel protein LZTFL1 regulates ciliary trafficking of the BBSome and Smoothened.
Seo S, Zhang Q, Bugge K, Breslow DK, Searby CC, Nachury MV, Sheffield VC
PLoS genetics 2011 Nov;7(11):e1002358
PLoS genetics 2011 Nov;7(11):e1002358
A ciliopathy complex at the transition zone protects the cilia as a privileged membrane domain.
Chih B, Liu P, Chinn Y, Chalouni C, Komuves LG, Hass PE, Sandoval W, Peterson AS
Nature cell biology 2011 Dec 18;14(1):61-72
Nature cell biology 2011 Dec 18;14(1):61-72
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Supportive validation
- Submitted by
- Proteintech Group (provider)
- Main image
- Experimental details
- Transfected HEK-293 cells were subjected to SDS PAGE followed by western blot with 17711-1-AP( ARL13B Antibody) at dilution of 1:1000
- Sample type
- cell line
- Submitted by
- Proteintech Group (provider)
- Main image
- Experimental details
- mouse liver tissue were subjected to SDS PAGE followed by western blot with 17711-1-AP(ARL13B antibody) at dilution of 1:1000
- Sample type
- tissue
Supportive validation
- Submitted by
- Proteintech Group (provider)
- Main image
- Experimental details
- IP Result of anti-ARL13B (IP:17711-1-AP, 3ug; Detection:17711-1-AP 1:1000) with L02 cells lysate 2500ug.
- Sample type
- cell line
Supportive validation
- Submitted by
- Proteintech Group (provider)
- Main image
- Experimental details
- Immunohistochemical of paraffin-embedded human liver using 17711-1-AP(ARL13B antibody) at dilution of 1:50 (under 10x lens)
- Sample type
- tissue
- Submitted by
- Proteintech Group (provider)
- Main image
- Experimental details
- Immunohistochemical of paraffin-embedded human liver using 17711-1-AP(ARL13B antibody) at dilution of 1:50 (under 40x lens)
- Sample type
- tissue
