2020
Regulation and function of calcium in the cilium
Sun Z. Regulation and function of calcium in the cilium. Current Opinion In Physiology 2020, 17: 278-283. PMID: 35937971, PMCID: PMC9351618, DOI: 10.1016/j.cophys.2020.08.019.Peer-Reviewed Original Research
2017
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. Palmitoylation of the ciliary GTPase ARL13b is necessary for its stability and its role in cilia formation. Journal Of Biological Chemistry 2017, 292: 17703-17717. PMID: 28848045, PMCID: PMC5663873, DOI: 10.1074/jbc.m117.792937.Peer-Reviewed Original ResearchConceptsPost-translational attachmentMost mammalian cellsCiliary GTPase Arl13bCilia localizationProtein palmitoylationCiliary proteinsCilia proteinsProtein localizationCilia formationMammalian cellsCilia functionPalmitoylationPrimary ciliaPlasma membraneCilia resorptionArl13bFunctional importanceMyristoylationCiliaCritical roleProteinMouse kidneyLocalizationDepalmitoylationCells
2015
Intraciliary Calcium Oscillations Initiate Vertebrate Left-Right Asymmetry
Yuan S, Zhao L, Brueckner M, Sun Z. Intraciliary Calcium Oscillations Initiate Vertebrate Left-Right Asymmetry. Current Biology 2015, 25: 556-567. PMID: 25660539, PMCID: PMC4469357, DOI: 10.1016/j.cub.2014.12.051.Peer-Reviewed Original ResearchConceptsLeft-right organizerLR developmentCiliary motilityVertebrate left–right asymmetryLeft-right signalingLive zebrafish embryosVertebrate developmentLeft-right asymmetryZebrafish embryosSensory ciliaPolycystin-2Signaling cascadesMolecular signalsMolecular mechanismsIntraciliary calciumCation channelsMotilityBilateral symmetryCalcium sinkCiliaCalcium oscillationsPKD2SignalingEmbryosExtracellular fluid
2014
Endothelial Cilia Are Essential for Developmental Vascular Integrity in Zebrafish
Kallakuri S, Yu JA, Li J, Li Y, Weinstein BM, Nicoli S, Sun Z. Endothelial Cilia Are Essential for Developmental Vascular Integrity in Zebrafish. Journal Of The American Society Of Nephrology 2014, 26: 864-875. PMID: 25214579, PMCID: PMC4378100, DOI: 10.1681/asn.2013121314.Peer-Reviewed Original ResearchConceptsIntraflagellar transport genesTransport genesModel organism zebrafishChemical genetic toolsKidney disease genesEndothelial ciliaInvolvement of ciliaVascular integrityInhibition of HedgehogHemorrhage phenotypeCilia biogenesisVertebrate cellsCiliary mutantsZebrafish vasculatureGenetic toolsRespective mutantsDisease genesEndothelial cellsNovel roleInactivating mutationGenesCiliaEssential roleZebrafishMutantsIFT27, encoding a small GTPase component of IFT particles, is mutated in a consanguineous family with Bardet–Biedl syndrome
Aldahmesh MA, Li Y, Alhashem A, Anazi S, Alkuraya H, Hashem M, Awaji AA, Sogaty S, Alkharashi A, Alzahrani S, Al Hazzaa S, Xiong Y, Kong S, Sun Z, Alkuraya FS. IFT27, encoding a small GTPase component of IFT particles, is mutated in a consanguineous family with Bardet–Biedl syndrome. Human Molecular Genetics 2014, 23: 3307-3315. PMID: 24488770, PMCID: PMC4047285, DOI: 10.1093/hmg/ddu044.Peer-Reviewed Original ResearchMeSH KeywordsAdolescentAmino Acid SequenceAnimalsBardet-Biedl SyndromeConsanguinityEvolution, MolecularExomeFemaleGenetic Predisposition to DiseaseHigh-Throughput Nucleotide SequencingHumansMaleModels, MolecularMonomeric GTP-Binding ProteinsPedigreePoint MutationSaudi ArabiaSequence AlignmentZebrafishConceptsBardet-Biedl syndromeBBS genesNovel BBS geneIntraflagellar transport genesAutosomal recessive ciliopathyIFT particlesProtein complexesTransport genesMembrane proteinsFunctional validationGenetic complexityRecessive ciliopathyHuman geneticsGenesIFT27Genetic heterogeneityConsanguineous familyBBS casesBBSomeZebrafishCiliopathiesGeneticsProteinCiliaFirst time
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
2011
Chapter 3 Analysis of Cilia Structure and Function in Zebrafish
Malicki J, Avanesov A, Li J, Yuan S, Sun Z. Chapter 3 Analysis of Cilia Structure and Function in Zebrafish. Methods In Cell Biology 2011, 101: 39-74. PMID: 21550439, DOI: 10.1016/b978-0-12-387036-0.00003-7.Peer-Reviewed Original ResearchConceptsExcellent vertebrate model systemVertebrate model systemNormal embryonic developmentCell surface protrusionsCilia biologyGenetic accessibilityVertebrate cellsLimb morphogenesisCilia formationImportant organellesEmbryonic developmentLarval organsLeft-right asymmetryCilia structureDistribution of ciliaNephric ductZebrafishCiliary malfunctionVariety of processesPhotoreceptor cellsSensory cellsKidney cystsCiliaModel systemChapter 3 Analysis
2010
Cilia in cell signaling and human disorders
Duldulao NA, Li J, Sun Z. Cilia in cell signaling and human disorders. Protein & Cell 2010, 1: 726-736. PMID: 21203914, PMCID: PMC4875200, DOI: 10.1007/s13238-010-0098-7.Peer-Reviewed Original ResearchConceptsRole of ciliaUnrelated human diseasesMulticellular organismsVestigial organelleCell signalingCellular organellesNormal organogenesisHuman disordersHuman diseasesCiliaKidney cystsOrganellesSensory roleNeural tube defectsCiliogenesisOrganogenesisDevelopmental disordersMotileOrganismsSignalingTube defectsRoleDefects
2009
Cilia localization is essential for in vivo functions of the Joubert syndrome protein Arl13b/Scorpion
Duldulao NA, Lee S, Sun Z. Cilia localization is essential for in vivo functions of the Joubert syndrome protein Arl13b/Scorpion. Development 2009, 136: 4033-4042. PMID: 19906870, PMCID: PMC2778746, DOI: 10.1242/dev.036350.Peer-Reviewed Original ResearchConceptsCilia formationVivo functionJoubert syndromeSeries of deletionsSonic hedgehog (Shh) signalingCilia localizationCiliary localizationNull mutantsPoint mutantsGene productsHedgehog signalingArl13bCiliary defectsMutantsKidney ductsZebrafishCiliaAutosomal recessive disorderAbnormal ultrastructureScorpionsRecessive disorderLocalizationRecent studiesKnockdownSignaling
2008
Cystic Kidney Gene seahorse Regulates Cilia-Mediated Processes and Wnt Pathways
Kishimoto N, Cao Y, Park A, Sun Z. Cystic Kidney Gene seahorse Regulates Cilia-Mediated Processes and Wnt Pathways. Developmental Cell 2008, 14: 954-961. PMID: 18539122, DOI: 10.1016/j.devcel.2008.03.010.Peer-Reviewed Original ResearchConceptsWnt pathwayCiliary signalsImportant sensory organelleKidney cyst formationNoncanonical Wnt pathwayCanonical Wnt pathwayLeft-right asymmetryCilia assemblySensory organellesCiliated tissuesCellular eventsCell typesSeahorsesCiliary functionPathwayCiliaCyst formationDishevelledInversinGastrulationVertebratesOrganellesTranscriptsProteinCascade