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 dynamicsCiliaATP1A2PlexusPTEN mutations impair CSF dynamics and cortical networks by dysregulating periventricular neural progenitors
DeSpenza T, Kiziltug E, Allington G, Barson D, McGee S, O’Connor D, Robert S, Mekbib K, Nanda P, Greenberg A, Singh A, Duy P, Mandino F, Zhao S, Lynn A, Reeves B, Marlier A, Getz S, Nelson-Williams C, Shimelis H, Walsh L, Zhang J, Wang W, Prina M, OuYang A, Abdulkareem A, Smith H, Shohfi J, Mehta N, Dennis E, Reduron L, Hong J, Butler W, Carter B, Deniz E, Lake E, Constable R, Sahin M, Srivastava S, Winden K, Hoffman E, Carlson M, Gunel M, Lifton R, Alper S, Jin S, Crair M, Moreno-De-Luca A, Luikart B, Kahle K. PTEN mutations impair CSF dynamics and cortical networks by dysregulating periventricular neural progenitors. Nature Neuroscience 2025, 28: 536-557. PMID: 39994410, PMCID: PMC12038823, DOI: 10.1038/s41593-024-01865-3.Peer-Reviewed Original ResearchConceptsNeural progenitor cellsCongenital hydrocephalusCSF dynamicsIncreased CSF productionDe novo mutationsFrequent monogenic causeEverolimus treatmentCSF shuntingNonsurgical treatmentPTEN mutationsAqueductal stenosisInhibitory interneuronsVentriculomegalyProgenitor cellsChoroid plexusMonogenic causeCortical networksIncreased survivalBrain ventriclesCortical deficitsNeural progenitorsGene PTENCSF productionNkx2.1PTEN
2023
Linking enlarged choroid plexus with plasma analyte and structural phenotypes in clinical high risk for psychosis: A multisite neuroimaging study
Bannai D, Reuter M, Hegde R, Hoang D, Adhan I, Gandu S, Pong S, Raymond N, Zeng V, Chung Y, He G, Sun D, van Erp T, Addington J, Bearden C, Cadenhead K, Cornblatt B, Mathalon D, McGlashan T, Jeffries C, Stone W, Tsuang M, Walker E, Woods S, Cannon T, Perkins D, Keshavan M, Lizano P. Linking enlarged choroid plexus with plasma analyte and structural phenotypes in clinical high risk for psychosis: A multisite neuroimaging study. Brain Behavior And Immunity 2023, 117: 70-79. PMID: 38169244, PMCID: PMC10932816, DOI: 10.1016/j.bbi.2023.12.021.Peer-Reviewed Original ResearchNorth American Prodrome Longitudinal Study 2Plasma analytesTotal white matter volumeChoroid plexus enlargementLarger lateral ventricle volumesLateral ventricle volumeWhite matter volumeClinical high riskSubcortical gray matterChronic psychosisNeuroanatomical alterationsBaseline scanPsychosis onsetHigh riskChoroid plexusMatter volumeVentricle volumeConversion statusGray matterNeuroimaging studiesPsychosisStructural phenotypesImportant biomarkerEnlargementLongitudinal Study 2The choroid plexus links innate immunity to CSF dysregulation in hydrocephalus
Robert S, Reeves B, Kiziltug E, Duy P, Karimy J, Mansuri M, Marlier A, Allington G, Greenberg A, DeSpenza T, Singh A, Zeng X, Mekbib K, Kundishora A, Nelson-Williams C, Hao L, Zhang J, Lam T, Wilson R, Butler W, Diluna M, Feinberg P, Schafer D, Movahedi K, Tannenbaum A, Koundal S, Chen X, Benveniste H, Limbrick D, Schiff S, Carter B, Gunel M, Simard J, Lifton R, Alper S, Delpire E, Kahle K. The choroid plexus links innate immunity to CSF dysregulation in hydrocephalus. Cell 2023, 186: 764-785.e21. PMID: 36803604, PMCID: PMC10069664, DOI: 10.1016/j.cell.2023.01.017.Peer-Reviewed Original ResearchConceptsPost-infectious hydrocephalusTLR4-dependent immune responseBlood-cerebrospinal fluid barrierSmall molecule pharmacotherapyCell cross talkPharmacological immunomodulationCytokine stormNeuroimmune disordersBrain infectionDrug treatmentImmune responseAcquired hydrocephalusHydrocephalus modelChoroid plexusFluid barrierHydrocephalusEpithelial cellsCSFMulti-omics investigationsCross talkHypersecretionHemorrhagePharmacotherapyImmunomodulationPlexus
2022
In Vivo Head‐To‐Head Comparison of [18F]GTP1 and [18F]PI2620 in Alzheimer’s Disease
Bohorquez S, Constantinescu C, Manser P, Gunn R, Russell D, Tonietto M, Bullich S, Stephens A, Mueller A, Klein G, Teng E, Pickthorn K. In Vivo Head‐To‐Head Comparison of [18F]GTP1 and [18F]PI2620 in Alzheimer’s Disease. Alzheimer's & Dementia 2022, 18 DOI: 10.1002/alz.063517.Peer-Reviewed Original ResearchChoroid plexusAlzheimer's diseaseTau pathology distributionModerate AD subjectsTarget cortical regionsTherapeutic trialsTau pathologyHead studiesPathology distributionCentrum semiovaleCortical greyBraak regionsAD subjectsHuman studiesInferior cerebellumSubcortical regionsHead comparisonQuantification scaleCortical regionsSubcortical structuresMinute imagesCSF spaceDiseaseUnimpaired subjectsSUVRIn Vivo Head‐To‐Head Comparison of [18F]GTP1 and [18F]PI2620 in Alzheimer’s Disease
Bohorquez S, Constantinescu C, Manser P, Gunn R, Russell D, Tonietto M, Bullich S, Stephens A, Mueller A, Klein G, Teng E, Pickthorn K. In Vivo Head‐To‐Head Comparison of [18F]GTP1 and [18F]PI2620 in Alzheimer’s Disease. Alzheimer's & Dementia 2022, 18 DOI: 10.1002/alz.063513.Peer-Reviewed Original ResearchChoroid plexusAlzheimer's diseaseTau pathology distributionModerate AD subjectsTarget cortical regionsTherapeutic trialsTau pathologyHead studiesPathology distributionCentrum semiovaleCortical greyBraak regionsAD subjectsHuman studiesInferior cerebellumSubcortical regionsHead comparisonQuantification scaleCortical regionsSubcortical structuresMinute imagesCSF spaceDiseaseUnimpaired subjectsSUVRChoroid plexus tissue perfusion and blood to CSF barrier function in rats measured with continuous arterial spin labeling
Lee H, Ozturk B, Stringer MS, Koundal S, MacIntosh BJ, Rothman D, Benveniste H. Choroid plexus tissue perfusion and blood to CSF barrier function in rats measured with continuous arterial spin labeling. NeuroImage 2022, 261: 119512. PMID: 35882269, PMCID: PMC9969358, DOI: 10.1016/j.neuroimage.2022.119512.Peer-Reviewed Original ResearchConceptsWater flowBlood perfusionLow-dose isofluraneCerebral blood flowClinical translational studiesAnti-diuretic hormoneContinuous arterial spinLevels of bloodMeasurement accuracyMagnetic resonance imagingArterial spin labelingCerebrospinal fluid productionAnesthetic regimensBalanced anesthesiaCerebral ventricleContinuous arterial spin labelingIsoflurane anesthesiaKey parametersSystemic administrationVentricular CSFImmune surveillanceArterial bloodBlood flowTissue perfusionChoroid plexus
2021
N-methyl-D-aspartate receptor antibody and the choroid plexus in schizophrenia patients with tardive dyskinesia
Li N, Huang J, Zhang P, Tong J, Chen S, Cui Y, Tan S, Wang Z, Tian B, Li CR, Hong LE, Tian L, Tan Y. N-methyl-D-aspartate receptor antibody and the choroid plexus in schizophrenia patients with tardive dyskinesia. Journal Of Psychiatric Research 2021, 142: 290-298. PMID: 34411812, DOI: 10.1016/j.jpsychires.2021.08.010.Peer-Reviewed Original ResearchConceptsNMDAR antibody levelsNeuronal N-methyl-D-aspartate receptorAbnormal Involuntary Movement ScaleTardive dyskinesiaAntibody levelsCentral nervous systemChoroid plexusAIMS scoresN-methyl-D-aspartate receptor antibodiesSchizophrenia patientsN-methyl-D-aspartate receptorsOrofacial tardive dyskinesiaCP volumeNegative Syndrome ScaleEnzyme-linked immunosorbentNMDAR antibodiesAutoimmune abnormalitiesImmune disturbancesReceptor antibodiesHealthy controlsImmune barrierMovement ScaleNervous systemNTD groupSyndrome Scale
2020
In Xenopus ependymal cilia drive embryonic CSF circulation and brain development independently of cardiac pulsatile forces
Dur AH, Tang T, Viviano S, Sekuri A, Willsey HR, Tagare HD, Kahle KT, Deniz E. In Xenopus ependymal cilia drive embryonic CSF circulation and brain development independently of cardiac pulsatile forces. Fluids And Barriers Of The CNS 2020, 17: 72. PMID: 33308296, PMCID: PMC7731788, DOI: 10.1186/s12987-020-00234-z.Peer-Reviewed Original ResearchConceptsCSF circulationOptical coherence tomographyCSF flowVentricular systemEpendymal ciliaCoherence tomographyBrain developmentCross-sectional imaging modalitiesBrain ventricular systemEarly time pointsVentricular morphologyCerebral ventricleRespiratory forceConclusionsOur dataCerebrospinal fluidChoroid plexusVentricular spaceCardiac forceEmbryonic brainPulsatile forcesDeadly diseaseTime pointsImaging modalitiesOCT imagingPathological expansionAdult H3K27M-mutant diffuse midline glioma with gliomatosis cerebri growth pattern: Case report and review of the literature
Yekula A, Gupta M, Coley N, U HS. Adult H3K27M-mutant diffuse midline glioma with gliomatosis cerebri growth pattern: Case report and review of the literature. International Journal Of Surgery Case Reports 2020, 68: 124-128. PMID: 32145563, PMCID: PMC7058855, DOI: 10.1016/j.ijscr.2020.02.046.Peer-Reviewed Original ResearchDiffuse midline gliomaH3K27M-mutant diffuse midline gliomaGliomatosis cerebri growth patternMidline gliomaGrade IV lesionsH3K27M mutationEarly biopsyClinicopathologic characteristicsDisease courseInfiltrative neoplasmPediatric patientsLeptomeningeal involvementPoor prognosisVentricular shuntingPatient counselingSuboccipital craniotomyVisual deteriorationCase reportCapsular lesionsPosterior fossaHigh indexChoroid plexusInfectious pathologyAccurate diagnosisGliomas
2019
Choroid Plexus Enlargement and Allostatic Load in Schizophrenia
Zhou Y, Huang J, Zhang P, Fan F, Chen S, Fan H, Cui Y, Luo X, Tan S, Wang Z, Feng W, Yuan Y, Yang F, Savransky A, Ryan M, Goldwaser E, Chiappelli J, Rowland L, Kochunov P, Tan Y, Hong L. Choroid Plexus Enlargement and Allostatic Load in Schizophrenia. Schizophrenia Bulletin 2019, 46: 722-731. PMID: 31603232, PMCID: PMC7147577, DOI: 10.1093/schbul/sbz100.Peer-Reviewed Original ResearchConceptsChoroid plexusHigher allostatic loadAllostatic loadSex-matched healthy controlsCentral nervous system abnormalitiesFirst-episode schizophrenia patientsChoroid plexus enlargementNervous system abnormalitiesTotal intracranial volumeHealthy controlsThalamus volumeLateral ventricleNeuroendocrine biomarkersSystem abnormalitiesAmygdala volumeSchizophrenia patientsBrain disordersInitial stabilizationIntracranial volumeStructural biomarkersPlexusBrain structuresPatientsSchizophreniaGroup differencesRecessive Inheritance of Congenital Hydrocephalus With Other Structural Brain Abnormalities Caused by Compound Heterozygous Mutations in ATP1A3
Allocco AA, Jin SC, Duy PQ, Furey CG, Zeng X, Dong W, Nelson-Williams C, Karimy JK, DeSpenza T, Hao LT, Reeves B, Haider S, Gunel M, Lifton RP, Kahle KT. Recessive Inheritance of Congenital Hydrocephalus With Other Structural Brain Abnormalities Caused by Compound Heterozygous Mutations in ATP1A3. Frontiers In Cellular Neuroscience 2019, 13: 425. PMID: 31616254, PMCID: PMC6775207, DOI: 10.3389/fncel.2019.00425.Peer-Reviewed Original ResearchCongenital hydrocephalusWhole-exome sequencingNeural stem cellsImmunohistochemical studyType 1 Chiari malformationUnaffected parentsStructural brain abnormalitiesAutosomal dominant neurological diseaseHuman congenital hydrocephalusCompound heterozygous mutationsPatient's unaffected parentsEmbryonic brain tissueImpaired NaAqueductal stenosisChiari malformationBrain abnormalitiesCorpus callosumMouse embryonic brainSingle patientChoroid plexusNeurological diseasesΑ3 subunitBrain tissueDifferentiated neuronsBrain development
2018
9p24 triplication in syndromic hydrocephalus with diffuse villous hyperplasia of the choroid plexus
Furey C, Antwi P, Duran D, Timberlake AT, Nelson-Williams C, Matouk CC, DiLuna ML, Günel M, Kahle KT. 9p24 triplication in syndromic hydrocephalus with diffuse villous hyperplasia of the choroid plexus. Molecular Case Studies 2018, 4: a003145. PMID: 29895553, PMCID: PMC6169828, DOI: 10.1101/mcs.a003145.Peer-Reviewed Original ResearchConceptsDiffuse villous hyperplasiaVillous hyperplasiaChoroid plexusSyndromic hydrocephalusCerebrospinal fluid homeostasisSurgical managementPathological featuresHigh prevalenceHydrocephalus treatmentHydrocephalusDVHCPFluid homeostasisCSF productionHyperplasiaPlexusChromosome 9pCritical genesHypersecretionPatientsPathogenesisPrevalenceDisease
2017
Lessons learned about [F-18]-AV-1451 off-target binding from an autopsy-confirmed Parkinson’s case
Marquié M, Verwer E, Meltzer A, Kim S, Agüero C, Gonzalez J, Makaretz S, Siao Tick Chong M, Ramanan P, Amaral A, Normandin M, Vanderburg C, Gomperts S, Johnson K, Frosch M, Gómez-Isla T. Lessons learned about [F-18]-AV-1451 off-target binding from an autopsy-confirmed Parkinson’s case. Acta Neuropathologica Communications 2017, 5: 75. PMID: 29047416, PMCID: PMC5648451, DOI: 10.1186/s40478-017-0482-0.Peer-Reviewed Original ResearchConceptsPositron emission tomographyBasal gangliaBinding to neuromelaninPositron emission tomography studiesDementia of AD typeInferior temporal cortexParkinson's diseaseTau pathologyPositron emission tomography scanAlzheimer's diseaseMild cognitive impairmentTemporal cortexAutoradiographic bindingBrain regionsNeurofibrillary tau pathologyOccipital cortexPositron emission tomography signalCognitive impairmentPostmortem materialEntorhinal cortexSubstantia nigraParenchymal hemorrhagePD diagnosisAutoradiography experimentsChoroid plexusInflammation-dependent cerebrospinal fluid hypersecretion by the choroid plexus epithelium in posthemorrhagic hydrocephalus
Karimy JK, Zhang J, Kurland DB, Theriault BC, Duran D, Stokum JA, Furey CG, Zhou X, Mansuri MS, Montejo J, Vera A, DiLuna ML, Delpire E, Alper SL, Gunel M, Gerzanich V, Medzhitov R, Simard JM, Kahle KT. Inflammation-dependent cerebrospinal fluid hypersecretion by the choroid plexus epithelium in posthemorrhagic hydrocephalus. Nature Medicine 2017, 23: 997-1003. PMID: 28692063, DOI: 10.1038/nm.4361.Peer-Reviewed Original ResearchMeSH KeywordsAcetazolamideAnimalsAntioxidantsBlotting, WesternBumetanideCerebral HemorrhageCerebral VentriclesCerebrospinal FluidChoroid PlexusDiureticsGene Knockdown TechniquesGene Knockout TechniquesHydrocephalusImmunoblottingImmunohistochemistryImmunoprecipitationInflammationNF-kappa BProlineProtein Serine-Threonine KinasesRatsRats, WistarSalicylanilidesSolute Carrier Family 12, Member 2SulfonamidesThiocarbamatesToll-Like Receptor 4CROI 2017: Neurologic Complications of HIV Infection.
Spudich SS, Ances BM. CROI 2017: Neurologic Complications of HIV Infection. Topics In Antiviral Medicine 2017, 25: 69-76. PMID: 28598791, PMCID: PMC5677044.Peer-Reviewed Original ResearchConceptsEarly HIV infectionHIV infectionCentral nervous systemAntiretroviral therapyCNS HIV infectionPotential viral reservoirAntiretroviral regimensSuppressive therapyVirologic controlNeurologic complicationsBrain macrophagesOpportunistic infectionsViral reservoirCognitive screening testsMicroglial cellsChronic infectionRisk factorsNeurocognitive disordersT cellsChoroid plexusNervous systemCognitive impairmentNumerous cohortsScreening testLongitudinal evaluation
2016
Expression of aquaporin-7 and aquaporin-9 in tanycyte cells and choroid plexus during mouse estrus cycle
Yaba A, Sozen B, Suzen B, Demir N. Expression of aquaporin-7 and aquaporin-9 in tanycyte cells and choroid plexus during mouse estrus cycle. Morphologie 2016, 101: 39-46. PMID: 27746040, DOI: 10.1016/j.morpho.2016.09.001.Peer-Reviewed Original ResearchConceptsMouse estrus cycleTanycyte cellsAQP-7Family of transmembrane proteinsEpithelial cells of choroid plexusPotential energy substratesExpression of aquaporin 7Oestrous cycleTransmembrane proteinsGlycerol transportRegulate reproductive functionMetestrus stageAquaporin 7Differential staining patternsProestrus stageAQP-9Diestrus stageProteinChoroid plexusEstrus stageHormonal controlMedian eminenceEpithelial cellsAquaporin-9Weak immunoreactivity
2012
A molecular characterization of the choroid plexus and stress-induced gene regulation
Sathyanesan M, Girgenti MJ, Banasr M, Stone K, Bruce C, Guilchicek E, Wilczak-Havill K, Nairn A, Williams K, Sass S, Duman JG, Newton SS. A molecular characterization of the choroid plexus and stress-induced gene regulation. Translational Psychiatry 2012, 2: e139-e139. PMID: 22781172, PMCID: PMC3410626, DOI: 10.1038/tp.2012.64.Peer-Reviewed Original ResearchConceptsStress-induced gene regulationGene expression changesGene expression analysisCP gene expressionGlial fibrillary acidic proteinChoroid plexusMolecular functionsGene regulationSitu hybridization analysisTranscriptomic characterizationHigh-resolution tandem mass spectrometryTarget genesExpression analysisGene expressionExpression changesTarget proteinsCP proteinsMolecular characterizationAdult choroid plexusHybridization analysisCP functionGene profilesProteinBlood-cerebrospinal fluid barrierResolution tandem mass spectrometry
2010
Prostaglandin E2 induces glutamate release from subventricular zone astrocytes.
Dave KA, Platel JC, Huang F, Tian D, Stamboulian-Platel S, Bordey A. Prostaglandin E2 induces glutamate release from subventricular zone astrocytes. Neuron Glia Biology 2010, 6: 201-7. PMID: 21211110, DOI: 10.1017/s1740925x10000244.Peer-Reviewed Original ResearchConceptsAmbient glutamate levelsProstaglandin E2Subventricular zoneGlutamate releaseAstrocyte-like cellsGlutamate levelsGramicidin-perforated patch-clamp techniquesIntracellular Ca2Application of PGE2Aspartate receptor channelsPatch-clamp techniqueLateral ventricleSVZ cellsPGE2 releaseChoroid plexusMature astrocytesNeuroblast survivalEnzyme immunoassayReceptor channelsAstrocytesE2Ca2CellsReleaseLesser extent
2009
Erythropoietin Induction by Electroconvulsive Seizure, Gene Regulation, and Antidepressant-Like Behavioral Effects
Girgenti MJ, Hunsberger J, Duman CH, Sathyanesan M, Terwilliger R, Newton SS. Erythropoietin Induction by Electroconvulsive Seizure, Gene Regulation, and Antidepressant-Like Behavioral Effects. Biological Psychiatry 2009, 66: 267-274. PMID: 19185286, DOI: 10.1016/j.biopsych.2008.12.005.Peer-Reviewed Original ResearchMeSH KeywordsAnimalsAntidepressive AgentsBehavior, AnimalBrain-Derived Neurotrophic FactorDepressionDisease Models, AnimalElectroshockErythropoietinExploratory BehaviorGene Expression ProfilingGene Expression RegulationHumansHypoxia-Inducible Factor 1, alpha SubunitLocomotionMaleOligonucleotide Array Sequence AnalysisRatsRats, Sprague-DawleyReceptors, ErythropoietinSeizuresSwimmingConceptsElectroconvulsive seizuresBehavioral effectsNIH testEPO receptorTrophic effectsRobust antidepressant-like effectsAntidepressant-like behavioral effectsBrain-derived neurotrophic factorAntidepressant-like efficacyTranscription factor hypoxiaAntidepressant-like effectsMultiple brain regionsQuantitative polymerase chain reactionExpression of erythropoietinAntidepressant actionAntidepressant effectsPeripheral administrationNeurotrophic factorIntracerebroventricular infusionPolymerase chain reactionTrophic actionNeurotrophic genesAnimal modelsChoroid plexusRegulation of erythropoietin
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