2025
Multicilia dynamically transduce Sonic Hedgehog signaling to regulate choroid plexus functions
Mao S, Song R, Jin S, Pang S, Jovanovic A, Zimmerman A, Li P, Wu X, Wendland M, Lin K, Chen W, Choksi S, Chen G, Holtzman M, Reiter J, Wan Y, Xuan Z, Xiang Y, Xu C, Upadhyayula S, Hess H, He L. Multicilia dynamically transduce Sonic Hedgehog signaling to regulate choroid plexus functions. Cell Reports 2025, 44: 115383. PMID: 40057957, DOI: 10.1016/j.celrep.2025.115383.Peer-Reviewed Original ResearchConceptsCSF productionChoroid plexusCerebrospinal fluidSonic hedgehog signalingWater channel AQP1Increased CSF productionHedgehog signalingChoroid plexus functionMotile ciliaMulticiliaSensory ciliaShh signalingNeonatal hydrocephalusSonic hedgehogCiliary lengthRegulate CSF productionSignal intensityCiliary ultrastructureChoroidEpithelial monolayersAQP1Developmental dynamicsCiliaATP1A2Plexus
2024
Loss of Katnal2 leads to ependymal ciliary hyperfunction and autism-related phenotypes in mice
Kang R, Kim K, Jung Y, Choi S, Lee C, Im G, Shin M, Ryu K, Choi S, Yang E, Shin W, Lee S, Lee S, Papadopoulos Z, Ahn J, Koh G, Kipnis J, Kang H, Kim H, Cho W, Park S, Kim S, Kim E. Loss of Katnal2 leads to ependymal ciliary hyperfunction and autism-related phenotypes in mice. PLOS Biology 2024, 22: e3002596. PMID: 38718086, PMCID: PMC11104772, DOI: 10.1371/journal.pbio.3002596.Peer-Reviewed Original ResearchConceptsAutism spectrum disorderBehavioral phenotypesASD-relatedSocial communication deficitsAutism-related phenotypesEnlarged lateral ventriclesProgressive ventricular enlargementCommunication deficitsSpectrum disorderSynaptic deficitsEnlargement of brain ventriclesTranscriptomic changesMicrotubule-regulatory proteinsGenes down-regulatedBrain ventriclesVentricular enlargementLateral ventricleDeficitsHippocampal neuronsMotile ciliaKATNAL2Potential treatmentDown-regulationCiliary functionEpendymal cells
2022
Cryo-EM structure of an active central apparatus
Han L, Rao Q, Yang R, Wang Y, Chai P, Xiong Y, Zhang K. Cryo-EM structure of an active central apparatus. Nature Structural & Molecular Biology 2022, 29: 472-482. PMID: 35578022, PMCID: PMC9113940, DOI: 10.1038/s41594-022-00769-9.Peer-Reviewed Original ResearchConceptsCentral apparatusDiverse cellular activitiesKinesin-like proteinCryo-EM structureArmadillo repeat proteinsCryo-electron microscopyHigh-resolution structuresEukaryotic speciesProtein subunitsMotile ciliaBridge proteinsPair of microtubulesRegulatory roleCellular activitiesProteinDynamic conformational behaviorCiliary motilityCiliaCiliary beatingStructural frameworkConformational behaviorSubunitsMicrotubulesRegulatorSpecies
2021
Cryo‐EM Structures of Outer‐arm Dynein Array Bound to Microtubule Doublet Reveal a Mechanism for Motor Coordination
Rao Q, Wang Y, Chai P, Kuo Y, Han L, Yang R, Yang Y, Howard J, Zhang K. Cryo‐EM Structures of Outer‐arm Dynein Array Bound to Microtubule Doublet Reveal a Mechanism for Motor Coordination. The FASEB Journal 2021, 35 DOI: 10.1096/fasebj.2021.35.s1.03099.Peer-Reviewed Original ResearchOuter arm dyneinMicrotubule-bound stateInner arm dyneinsCentral pair complexMicrotubule-binding domainMicrotubule doubletsIntermediate chainATP hydrolysisFundamental cellular processesHeavy chainCryo-EM structureCryo-EM analysisCryo-electron tomographyKey motor proteinCryo-electron microscopyLight chainCellular processesEukaryotic ciliaT. thermophilaEmbryonic developmentAdjacent microtubule doubletsCellular motilityMotor proteinsAxonemal dyneinsMotile cilia
2019
PACRG and FAP20 form the inner junction of axonemal doublet microtubules and regulate ciliary motility
Dymek EE, Lin J, Fu G, Porter ME, Nicastro D, Smith EF. PACRG and FAP20 form the inner junction of axonemal doublet microtubules and regulate ciliary motility. Molecular Biology Of The Cell 2019, 30: 1805-1816. PMID: 31116684, PMCID: PMC6727744, DOI: 10.1091/mbc.e19-01-0063.Peer-Reviewed Original ResearchConceptsDoublet microtubulesAxonemal doublet microtubulesCryo-electron tomographyCiliary doublet microtubulesInner junctionCiliary assemblyCoordinated ciliary beatingFAP20Ciliary componentsMotility defectsCiliary beatingPACRGMotile ciliaFunctional studiesMutant axonemesMicrotubulesB-tubuleCiliary motilityEssential roleStructural studiesAssemblyMotilityChlamydomonasMutantsAxoneme
2018
Asymmetric distribution and spatial switching of dynein activity generates ciliary motility
Lin J, Nicastro D. Asymmetric distribution and spatial switching of dynein activity generates ciliary motility. Science 2018, 360 PMID: 29700238, PMCID: PMC6640125, DOI: 10.1126/science.aar1968.Peer-Reviewed Original ResearchConceptsDynein activitySea urchin sperm cellsCryo-electron tomographyIndividual dyneinsDynein isoformsAsymmetric distributionMotile ciliaDyneinFlagellaHypothesis positsSperm cellsSmall populationCiliary motilityFlagellar bendingCoordinated activityActive stateMotilityActivity stateOrganellesIsoformsCiliaActivityThe I1 dynein-associated tether and tether head complex is a conserved regulator of ciliary motility
Fu G, Wang Q, Phan N, Urbanska P, Joachimiak E, Lin J, Wloga D, Nicastro D. The I1 dynein-associated tether and tether head complex is a conserved regulator of ciliary motility. Molecular Biology Of The Cell 2018, 29: mbc.e18-02-0142. PMID: 29514928, PMCID: PMC5921572, DOI: 10.1091/mbc.e18-02-0142.Peer-Reviewed Original ResearchConceptsI1 dyneinCiliary motilityAxonemal dynein motorsDynein motor domainMotor domainCiliary doublet microtubulesCK1 kinasesProper phosphorylationNormal ciliary motilityBiochemical approachesCryoelectron tomographyDynein motorsRegulatory mechanismsMotility regulatorMotile ciliaDoublet microtubulesRegulatorDyneinStable anchoringMotilityCiliary beatingComplexesHead complexMechanical feedbackImportant role
2013
Expanding Horizons: Ciliary Proteins Reach Beyond Cilia
Yuan S, Sun Z. Expanding Horizons: Ciliary Proteins Reach Beyond Cilia. Annual Review Of Genetics 2013, 47: 353-376. PMID: 24016188, PMCID: PMC5703194, DOI: 10.1146/annurev-genet-111212-133243.Peer-Reviewed Original ResearchMeSH KeywordsAbnormalities, MultipleAnimalsBardet-Biedl SyndromeCell MovementCerebellar DiseasesCerebellumCiliaCiliary Motility DisordersDisease Models, AnimalDNA DamageDNA RepairEncephaloceleEye AbnormalitiesFlagellaHeterotaxy SyndromeHomeostasisHumansKidney Diseases, CysticMolecular Motor ProteinsNervous SystemPolycystic Kidney DiseasesPolycystic Kidney, Autosomal DominantPolycystic Kidney, Autosomal RecessiveRetinaRetinitis PigmentosaZMYND10 Is Mutated in Primary Ciliary Dyskinesia and Interacts with LRRC6
Zariwala MA, Gee HY, Kurkowiak M, Al-Mutairi DA, Leigh MW, Hurd TW, Hjeij R, Dell SD, Chaki M, Dougherty GW, Adan M, Spear PC, Esteve-Rudd J, Loges NT, Rosenfeld M, Diaz KA, Olbrich H, Wolf WE, Sheridan E, Batten TF, Halbritter J, Porath JD, Kohl S, Lovric S, Hwang DY, Pittman JE, Burns KA, Ferkol TW, Sagel SD, Olivier KN, Morgan LC, Werner C, Raidt J, Pennekamp P, Sun Z, Zhou W, Airik R, Natarajan S, Allen SJ, Amirav I, Wieczorek D, Landwehr K, Nielsen K, Schwerk N, Sertic J, Köhler G, Washburn J, Levy S, Fan S, Koerner-Rettberg C, Amselem S, Williams DS, Mitchell BJ, Drummond IA, Otto EA, Omran H, Knowles MR, Hildebrandt F. ZMYND10 Is Mutated in Primary Ciliary Dyskinesia and Interacts with LRRC6. American Journal Of Human Genetics 2013, 93: 336-345. PMID: 23891469, PMCID: PMC3738827, DOI: 10.1016/j.ajhg.2013.06.007.Peer-Reviewed Original ResearchMeSH KeywordsAnimalsAutoantigensAxonemal DyneinsBiomarkersCell Cycle ProteinsCiliaCytoskeletal ProteinsExomeGene Expression RegulationHigh-Throughput Nucleotide SequencingHumansKartagener SyndromeMaleMicrotubule-Associated ProteinsMutationPedigreeProtein BindingProtein Structure, TertiaryProteinsRatsRespiratory SystemTumor Suppressor ProteinsXenopus laevisZebrafishConceptsCytoplasmic protein complexesMotile ciliary functionC-terminal domainWhole-exome resequencingProtein complexesHuman primary ciliary dyskinesiaZMYND10LRRC6Motile ciliaHigh-throughput mutation analysisOtolith defectsPrimary ciliary dyskinesiaCiliary functionMutationsCS domainBiallelic mutationsKnockdownCystic kidneysMutation analysisCiliaCiliary dyskinesiaSAS6ResequencingZebrafishCiliogenesis
2012
The CSC connects three major axonemal complexes involved in dynein regulation
Heuser T, Dymek EE, Lin J, Smith EF, Nicastro D. The CSC connects three major axonemal complexes involved in dynein regulation. Molecular Biology Of The Cell 2012, 23: 3143-3155. PMID: 22740634, PMCID: PMC3418309, DOI: 10.1091/mbc.e12-05-0357.Peer-Reviewed Original ResearchConceptsNexin-dynein regulatory complexDynein regulationRadial spokesAxonemal complexCryo-electron tomographyWild-type motilityRegulatory complexFlagellar motilityDynein motorsChlamydomonas flagellaMotile ciliaStable assemblyFlagellaDifferent functionsRegulationRS1ComplexesMotilityStructural heterogeneityRS2Human healthImportant roleOrganellesHomologuesCSCThe structural heterogeneity of radial spokes in cilia and flagella is conserved
Lin J, Heuser T, Carbajal‐González B, Song K, Nicastro D. The structural heterogeneity of radial spokes in cilia and flagella is conserved. Cytoskeleton 2012, 69: 88-100. PMID: 22170736, PMCID: PMC3307805, DOI: 10.1002/cm.21000.Peer-Reviewed Original ResearchConceptsFlagellar motilityRadial spokesNexin-dynein regulatory complexRegulatory complexFlagellar structureMost organismsThree-dimensional structureSubtomogram averagingCryoelectron tomographyDynein motorsDynein activityChlamydomonas flagellaMotile ciliaFlagellaSperm flagellaUbiquitous componentOrganismsFunctional differencesEssential roleCiliaRepeat unitsUnprecedented detailBasal partRS1Motility
2011
Building Blocks of the Nexin-Dynein Regulatory Complex in Chlamydomonas Flagella*
Lin J, Tritschler D, Song K, Barber CF, Cobb JS, Porter ME, Nicastro D. Building Blocks of the Nexin-Dynein Regulatory Complex in Chlamydomonas Flagella*. Journal Of Biological Chemistry 2011, 286: 29175-29191. PMID: 21700706, PMCID: PMC3190725, DOI: 10.1074/jbc.m111.241760.Peer-Reviewed Original ResearchConceptsNexin-dynein regulatory complexRegulatory complexImportant regulatory nodeTwo-dimensional electrophoresisRegulatory nodesProtein phosphorylationSignal transductionPhosphorylation statusMALDI-TOF mass spectrometryDynein motorsDynein activityChlamydomonas flagellaMotile ciliaRegulatory functionsPhosphorylated isoformsCoordination of thousandsFlagellar axonemeBiochemical comparisonRadial spokesPCR analysisFlagellar bendingIsoform patternFlagellaMolecular compositionProtein
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