2023
In Utero Electroporated Neurons for Medium-Throughput Screening of Compounds Regulating Neuron Morphology
Sokolov A, Aurich M, Bordey A. In Utero Electroporated Neurons for Medium-Throughput Screening of Compounds Regulating Neuron Morphology. ENeuro 2023, 10: eneuro.0160-23.2023. PMID: 37620147, PMCID: PMC10464655, DOI: 10.1523/eneuro.0160-23.2023.Peer-Reviewed Original ResearchConceptsSomatosensory cortexCortical pyramidal neuronsTreatment of epilepsyNeurite overgrowthNeurologic disabilityPyramidal neuronsSoma sizeNovel agentsCircuit alterationsSide effectsMorphologic assessmentMouse neuronsRelated disordersDiseased neuronsMTOR activatorDrug efficacyCandidate therapeuticsNeuronsNeuron morphologyMTOR activityMedium-throughput screeningNeurodevelopmental disordersNeurite lengthMorphologic measurementsDisorders
2020
ATP Synthase c-Subunit Leak Causes Aberrant Cellular Metabolism in Fragile X Syndrome
Licznerski P, Park HA, Rolyan H, Chen R, Mnatsakanyan N, Miranda P, Graham M, Wu J, Cruz-Reyes N, Mehta N, Sohail S, Salcedo J, Song E, Effman C, Effman S, Brandao L, Xu GN, Braker A, Gribkoff VK, Levy RJ, Jonas EA. ATP Synthase c-Subunit Leak Causes Aberrant Cellular Metabolism in Fragile X Syndrome. Cell 2020, 182: 1170-1185.e9. PMID: 32795412, PMCID: PMC7484101, DOI: 10.1016/j.cell.2020.07.008.Peer-Reviewed Original ResearchConceptsFragile X syndromeC subunitAberrant synaptic developmentHuman fragile X syndromeATP synthase enzymeMental retardation proteinX syndromeATP production efficiencyMRNA translation rateAberrant cellular metabolismATP synthaseMRNA translationTranslation rateCellular metabolismSynaptic growthSynthase enzymeMouse neuronsSynapse maturationSynaptic developmentPharmacological inhibitionLeak channelsSynaptic maturationMembrane leakMaturationMetabolismThe stress-responsive gene GDPGP1/mcp-1 regulates neuronal glycogen metabolism and survival
Schulz A, Sekine Y, Oyeyemi MJ, Abrams AJ, Basavaraju M, Han SM, Groth M, Morrison H, Strittmatter SM, Hammarlund M. The stress-responsive gene GDPGP1/mcp-1 regulates neuronal glycogen metabolism and survival. Journal Of Cell Biology 2020, 219: e201807127. PMID: 31968056, PMCID: PMC7041677, DOI: 10.1083/jcb.201807127.Peer-Reviewed Original ResearchConceptsNeuronal stress resistanceStress resistanceNovel cellular responsesMouse neuronsVariety of stressesCaenorhabditis elegansC. elegansTranscriptional analysisSingle homologueEnvironmental stressFunctional characterizationCellular responsesCell deathNeuronal cell deathNeuronal glycogenGlycogen metabolismWidespread neuronal cell deathElegansSurvival of animalsTauopathy modelMaladaptive responsesKey roleHomologuesGlycogen levelsKnockdown
2019
Potassium channel dysfunction in human neuronal models of Angelman syndrome
Sun A, Yuan Q, Fukuda M, Yu W, Yan H, Lim G, Nai M, D'Agostino G, Tran H, Itahana Y, Wang D, Lokman H, Itahana K, Lim S, Tang J, Chang Y, Zhang M, Cook S, Rackham O, Lim C, Tan E, Ng H, Lim K, Jiang Y, Je H. Potassium channel dysfunction in human neuronal models of Angelman syndrome. Science 2019, 366: 1486-1492. PMID: 31857479, PMCID: PMC7735558, DOI: 10.1126/science.aav5386.Peer-Reviewed Original ResearchConceptsAngelman syndromePotassium channel dysfunctionAS mouse modelUbiquitin protein ligase E3A (UBE3A) geneHuman neuronal modelNeuronal hyperexcitabilityNetwork hyperactivityAS patientsSeizure susceptibilitySynaptic dysfunctionModel miceIntrinsic excitabilityNeuronal excitabilityMouse modelBig potassium channelsHuman neuronsChannel dysfunctionEpilepsy susceptibilityBK channelopathyMouse neuronsPotassium channelsIndividual neuronsBrain organoidsNeuronsDysfunction
2017
The methyltransferase SETDB1 regulates a large neuron-specific topological chromatin domain
Jiang Y, Loh YE, Rajarajan P, Hirayama T, Liao W, Kassim BS, Javidfar B, Hartley BJ, Kleofas L, Park RB, Labonte B, Ho SM, Chandrasekaran S, Do C, Ramirez BR, Peter CJ, C W JT, Safaie BM, Morishita H, Roussos P, Nestler EJ, Schaefer A, Tycko B, Brennand KJ, Yagi T, Shen L, Akbarian S. The methyltransferase SETDB1 regulates a large neuron-specific topological chromatin domain. Nature Genetics 2017, 49: 1239-1250. PMID: 28671686, PMCID: PMC5560095, DOI: 10.1038/ng.3906.Peer-Reviewed Original Research
2015
Exchange Protein Directly Activated by cAMP (EPAC) Regulates Neuronal Polarization through Rap1B
Muñoz-Llancao P, Henríquez D, Wilson C, Bodaleo F, Boddeke E, Lezoualc'h F, Schmidt M, González-Billault C. Exchange Protein Directly Activated by cAMP (EPAC) Regulates Neuronal Polarization through Rap1B. Journal Of Neuroscience 2015, 35: 11315-11329. PMID: 26269639, PMCID: PMC6605123, DOI: 10.1523/jneurosci.3645-14.2015.Peer-Reviewed Original ResearchConceptsNeuronal polarityAcquisition of neuronal polarityAxon specificationGenetic manipulationRap1b activationExchange proteinGuanine exchange factorKnock-outEpac signalingRegulate neuronal polarityProtein kinase A.Axon determinationRap GTPasesWild-type neuronsExchange factorActivation of protein kinase A.Polarity defectsMouse neuronsRat hippocampal neuronsMessenger cAMPMolecular eventsEpacMature axonsCAMP-dependent mechanismPharmacological inhibition
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