2021
Structural insights into the cause of human RSPH4A primary ciliary dyskinesia
Zhao Y, Pinskey J, Lin J, Yin W, Sears P, Daniels L, Zariwala M, Knowles M, Ostrowski L, Nicastro D. Structural insights into the cause of human RSPH4A primary ciliary dyskinesia. Molecular Biology Of The Cell 2021, 32: 1202-1209. PMID: 33852348, PMCID: PMC8351563, DOI: 10.1091/mbc.e20-12-0806.Peer-Reviewed Original ResearchMeSH KeywordsAxonemeCiliaCiliary Motility DisordersCytoskeletal ProteinsElectron Microscope TomographyHumansMutationRespiratory SystemConceptsStructural basisCryo-electron tomographyRadial spokesCentral pair complexUnderlying structural basisAxonemal repeatEukaryotic organellesArch domainThree-dimensional structureSubtomogram averagingOrgan positioningCell motilityStructural insightsPrimary ciliary dyskinesiaCiliaHuman ciliaHuman respiratory ciliaRS1Primary defectStructure determinationCiliary dyskinesiaHuman healthOrganellesFlagellaRepeats
2019
FAP57/WDR65 targets assembly of a subset of inner arm dyneins and connects to regulatory hubs in cilia
Lin J, Le TV, Augspurger K, Tritschler D, Bower R, Fu G, Perrone C, O’Toole E, Mills KV, Dymek E, Smith E, Nicastro D, Porter ME. FAP57/WDR65 targets assembly of a subset of inner arm dyneins and connects to regulatory hubs in cilia. Molecular Biology Of The Cell 2019, 30: 2659-2680. PMID: 31483737, PMCID: PMC6761771, DOI: 10.1091/mbc.e19-07-0367.Peer-Reviewed Original ResearchConceptsInner dynein armsRegulatory complexCryo-electron tomographyInner arm dyneinsCiliary motilityMultiple dynein motorsPrecise spatial organizationAxonemal repeatDocking factorUnique binding siteWD repeatsDynein assemblyAssembly factorsDomain proteinsRegulatory hubDynein complexDynein isoformsDynein subunitsInsertional mutagenesisNew lociRegulatory proteinsDynein motorsDifferent dyneinsDoublet microtubulesTransport factors
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 ResearchMeSH KeywordsAnimalsChlamydomonasCiliaDyneinsElectron Microscope TomographyFlagellaMaleMicrotubulesSea UrchinsSperm MotilitySpermatozoaConceptsDynein activitySea urchin sperm cellsCryo-electron tomographyIndividual dyneinsDynein isoformsAsymmetric distributionMotile ciliaDyneinFlagellaHypothesis positsSperm cellsSmall populationCiliary motilityFlagellar bendingCoordinated activityActive stateMotilityActivity stateOrganellesIsoformsCiliaActivity
2015
DRC3 connects the N-DRC to dynein g to regulate flagellar waveform
Awata J, Song K, Lin J, King SM, Sanderson MJ, Nicastro D, Witman GB. DRC3 connects the N-DRC to dynein g to regulate flagellar waveform. Molecular Biology Of The Cell 2015, 26: 2788-2800. PMID: 26063732, PMCID: PMC4571338, DOI: 10.1091/mbc.e15-01-0018.Peer-Reviewed Original Research
2014
Cryo-electron tomography reveals ciliary defects underlying human RSPH1 primary ciliary dyskinesia
Lin J, Yin W, Smith MC, Song K, Leigh MW, Zariwala MA, Knowles MR, Ostrowski LE, Nicastro D. Cryo-electron tomography reveals ciliary defects underlying human RSPH1 primary ciliary dyskinesia. Nature Communications 2014, 5: 5727. PMID: 25473808, PMCID: PMC4267722, DOI: 10.1038/ncomms6727.Peer-Reviewed Original ResearchMeSH KeywordsCiliaCryoelectron MicroscopyDNA-Binding ProteinsElectron Microscope TomographyHumansKartagener SyndromeConceptsCryo-electron tomographyNative 3D structureNormal human developmentDistant speciesHuman ciliaCilia structurePCD phenotypeCilia dysfunctionHuman diseasesNative structurePrimary ciliary dyskinesiaRadial spokesCiliaEssential roleFunctional heterogeneityHuman respiratory ciliaUnprecedented detailPrimary defectCiliary dyskinesiaUnknown primary defect