2023
TIF1γ Counteracts Ferroptosis to Drive Erythroid Progenitor Differentiation
Rossmann M, Yang S, Abraham B, Wang Y, Young R, Hekimi S, Zon L. TIF1γ Counteracts Ferroptosis to Drive Erythroid Progenitor Differentiation. Blood 2023, 142: 8. DOI: 10.1182/blood-2023-184853.Peer-Reviewed Original ResearchTranscription elongationErythroid differentiationErythroid lineageNucleotide metabolismHematopoietic stem cell differentiationCell fate decisionsGenome-wide expressionChromatin immunoprecipitation analysisOnset of hematopoiesisTranscription regulatory processesErythroid progenitor differentiationStem cell differentiationElectron transport chainInhibitor of ferroptosisTranscriptional intermediary factor 1 gammaEnzyme dihydroorotate dehydrogenaseChromatin factorsSuppressor screenMutant embryosFate decisionsTranscriptome profilingZebrafish embryosProgenitor differentiationLineage differentiationBlood differentiationShared retinoic acid responsive enhancers coordinately regulate nascent transcription of Hoxb coding and non-coding RNAs in the developing mouse neural tube
Afzal Z, Lange J, Nolte C, McKinney S, Wood C, Paulson A, De Kumar B, Unruh J, Slaughter B, Krumlauf R. Shared retinoic acid responsive enhancers coordinately regulate nascent transcription of Hoxb coding and non-coding RNAs in the developing mouse neural tube. Development 2023, 150: dev201259. PMID: 37102683, PMCID: PMC10233718, DOI: 10.1242/dev.201259.Peer-Reviewed Original ResearchConceptsNascent transcriptionDynamic regulatory interactionsHox gene expressionCis-regulatory elementsRetinoic acid response elementMouse neural tubeTranscription of genesNon-coding RNAAcid response elementSingle-molecule fluorescentRetinoic acid responseMutant embryosHOXB clusterHox expressionAxial identityHoxb genesRegulatory interactionsTranscriptional mechanismsGene expressionDependent enhancersTranscriptionResponse elementResponsive enhancerNeural tubeCompetitive interactions169 Exome Sequencing Implicates Endothelial Ras Signaling Network in Vein of Galen Aneurysmal Malformation
Mekbib K, Zhao S, Nelson-Williams C, Prendergast A, Zeng X, Rolle M, Shohfi J, Smith H, Ocken J, Moyer Q, Piwowarczyk P, Allington G, Dong W, van der Ent M, Chen D, Li B, Duran D, Mane S, Walcott B, Stapleton C, Aagaard-Kienitz B, Rodesch G, Jackson E, Smith E, Orbach D, Berenstein A, Bilguvar K, Zhao H, Erson-Omay Z, King P, Huttner A, Lifton R, Boggon T, Nicoli S, Jin S, Kahle K. 169 Exome Sequencing Implicates Endothelial Ras Signaling Network in Vein of Galen Aneurysmal Malformation. Neurosurgery 2023, 69: 22-22. DOI: 10.1227/neu.0000000000002375_169.Peer-Reviewed Original ResearchPathway analysisP120 Ras-GAPExome sequencingSevere vascular defectsGalen aneurysmal malformationReceptor tyrosine kinase activityTyrosine kinase activityDamaging de novoMutant embryosRas-GAPSignaling networksGenetic regulationRas activationAneurysmal malformationZebrafish modelDe novo mutationsKinase activityDisease genesAxon guidanceGenetic samplesWhole-exome sequencingHigh-output heart failureFunctional studiesCollected specimensSequencing
2021
HOXA5 Participates in Brown Adipose Tissue and Epaxial Skeletal Muscle Patterning and in Brown Adipocyte Differentiation
Holzman MA, Ryckman A, Finkelstein TM, Landry-Truchon K, Schindler KA, Bergmann JM, Jeannotte L, Mansfield JH. HOXA5 Participates in Brown Adipose Tissue and Epaxial Skeletal Muscle Patterning and in Brown Adipocyte Differentiation. Frontiers In Cell And Developmental Biology 2021, 9: 632303. PMID: 33732701, PMCID: PMC7959767, DOI: 10.3389/fcell.2021.632303.Peer-Reviewed Original ResearchSkeletal muscle fateNull mutant embryosBAT developmentBrown adipocyte differentiationEmbryonic day 12.5Muscle fateBrown adipose tissueSkeletal muscleMutant embryosHOXA5 proteinLipid droplet morphologyForelimb levelEmbryonic developmentMolecular roleMuscle developmentLineage tracingMuscle patterningCommon progenitorDependent regulationMuscle phenotypeAdipocyte differentiationMultiple tissuesConditional deletionDay 12.5Progenitors
2020
BMP signalling is required for extra-embryonic ectoderm development during pre-to-post-implantation transition of the mouse embryo
Sozen B, Demir N, Zernicka-Goetz M. BMP signalling is required for extra-embryonic ectoderm development during pre-to-post-implantation transition of the mouse embryo. Developmental Biology 2020, 470: 84-94. PMID: 33217407, PMCID: PMC8219371, DOI: 10.1016/j.ydbio.2020.11.005.Peer-Reviewed Original ResearchConceptsBMP signalingEctoderm developmentNon-canonical mechanismMouse embryosMechanism of BMPMutant embryosGenetic studiesEarly embryogenesisSignaling pathwayPost-implantation developmentBMP activityTissue organisationStem cell populationStage in vitroSpatiotemporal controlUterine tissueEmbryosCell populationsBMPStem cellsSignalImplantationPost-implantationMiceMorphogenesis
2019
A Zebrafish Model for Selenoprotein Synthesis and Function (OR11-01-19)
Copeland P, Vetick M. A Zebrafish Model for Selenoprotein Synthesis and Function (OR11-01-19). Current Developments In Nutrition 2019, 3: 3131070. PMCID: PMC6578458, DOI: 10.1093/cdn/nzz044.or11-01-19.Peer-Reviewed Original ResearchZebrafish model systemSelenoprotein expressionSelenoprotein synthesisCo-translational insertionHomozygous mutant animalsModel systemMutant embryosSelenoprotein functionOxidative stressUGA codonMutant animalsZebrafish modelCRISPR/Selenoprotein mRNAsNull animalsOvert phenotypePeroxide exposureProtein 2Radioactive seleniumEmbryosProtein expressionExpressionProteinΜM H2O2Later time points
2017
MicroRNAs Establish Uniform Traits during the Architecture of Vertebrate Embryos
Kasper DM, Moro A, Ristori E, Narayanan A, Hill-Teran G, Fleming E, Moreno-Mateos M, Vejnar CE, Zhang J, Lee D, Gu M, Gerstein M, Giraldez A, Nicoli S. MicroRNAs Establish Uniform Traits during the Architecture of Vertebrate Embryos. Developmental Cell 2017, 40: 552-565.e5. PMID: 28350988, PMCID: PMC5404386, DOI: 10.1016/j.devcel.2017.02.021.Peer-Reviewed Original ResearchConceptsDevelopment of vertebratesEmbryonic blood vesselsVertebrate miRNAsMutant embryosVertebrate embryosHigher organismsMiRNAs functionVascular traitsTrait varianceUniform traitTissue developmentEnvironmental perturbationsSignaling pathwaysPhenotypic variabilityEmbryosPhenotypic heterogeneityOrganismsTraitsProper functioningVascular systemVertebratesMutagenesisMiRNAsMicroRNAsPhenotype
2013
Murine craniofacial development requires Hdac3-mediated repression of Msx gene expression
Singh N, Gupta M, Trivedi CM, Singh MK, Li L, Epstein JA. Murine craniofacial development requires Hdac3-mediated repression of Msx gene expression. Developmental Biology 2013, 377: 333-344. PMID: 23506836, PMCID: PMC3652235, DOI: 10.1016/j.ydbio.2013.03.008.Peer-Reviewed Original ResearchConceptsNeural crest cellsCrest cellsCraniofacial developmentNeural crestCranial neural crest cellsCore apoptotic pathwayMurine craniofacial developmentMsx gene expressionCleft secondary palateCell cycle genesCell cycle progressionEpigenomic levelsMutant embryosCraniofacial morphogenesisCycle genesCranial mesenchymeGenetic pathwaysGene expressionCycle progressionApoptotic pathwayMurine geneticsCritical regulatorHDAC3Mesodermal originSecondary palateA truncation allele in vascular endothelial growth factor c reveals distinct modes of signaling during lymphatic and vascular development
Villefranc JA, Nicoli S, Bentley K, Jeltsch M, Zarkada G, Moore JC, Gerhardt H, Alitalo K, Lawson ND. A truncation allele in vascular endothelial growth factor c reveals distinct modes of signaling during lymphatic and vascular development. Development 2013, 140: 1497-1506. PMID: 23462469, PMCID: PMC3596992, DOI: 10.1242/dev.084152.Peer-Reviewed Original ResearchMeSH KeywordsAllelesAnimalsAnimals, Genetically ModifiedAutocrine CommunicationBlood VesselsCell MovementCodon, NonsenseEmbryo, NonmammalianFemaleLymphatic SystemMiceMice, KnockoutNeovascularization, PhysiologicParacrine CommunicationProtein IsoformsSignal TransductionVascular Endothelial Growth Factor CZebrafishZebrafish ProteinsConceptsMigratory persistenceLymphatic developmentSensitized genetic backgroundTip cell positionVascular endothelial growth factor CEndothelial cell dynamicsDeficient endothelial cellsFactor CTime-lapse analysisMutant embryosVertebrate embryosTruncation alleleEctopic blood vesselsFilopodia stabilityAngiogenesis defectsDistinct modesEndothelial cellsDevelopmental angiogenesisLymphatic vasculatureVascular developmentLymphatic defectsGenetic backgroundReceptor FLT4VEGFCCell position
2012
NogoB receptor is essential for extraembryonic vascular development and protein glycosylation
Park E, Sessa W. NogoB receptor is essential for extraembryonic vascular development and protein glycosylation. The FASEB Journal 2012, 26: 607.5-607.5. DOI: 10.1096/fasebj.26.1_supplement.607.5.Peer-Reviewed Original ResearchExtraembryonic vascular developmentDehydrodolichyl diphosphate synthaseProtein glycosylationPeri-implantation embryonic lethalityVascular developmentMutant yolk sacsProtein N-glycosylationYolk sacCKO embryosMutant embryosUseful model systemNon-functional formEmbryonic lethalityDiphosphate synthaseExtraembryonic tissuesN-glycosylationProtein stabilityEndothelial cellsConditional knockout miceDevelopment defectsTube formationMutantsCKO mouse modelModel systemEmbyronic development
2009
Separating genetic and hemodynamic defects in neuropilin 1
Jones E, Yuan L, Breant C, Watts R, Eichmann A. Separating genetic and hemodynamic defects in neuropilin 1. The FASEB Journal 2009, 23: 311.1-311.1. DOI: 10.1096/fasebj.23.1_supplement.311.1.Peer-Reviewed Original ResearchVascular developmentProper vascular developmentYolk sac blood vesselsGenetic defectsMutant embryosBiochemical signalingYolk sac arteriesCardiovascular developmentMouse embryo cultureEmbryo growthBlood flowPhysiological functionsHemodynamic defectsVascular defectsVascular formationEmbryo cultureBiological senseNeuropilin-1Proper blood flowPhysical forcesElevated P75NTR expression causes death of engrailed-deficient midbrain dopaminergic neurons by Erk1/2 suppression
Alavian KN, Sgadò P, Alberi L, Subramaniam S, Simon HH. Elevated P75NTR expression causes death of engrailed-deficient midbrain dopaminergic neurons by Erk1/2 suppression. Neural Development 2009, 4: 11. PMID: 19291307, PMCID: PMC2667502, DOI: 10.1186/1749-8104-4-11.Peer-Reviewed Original ResearchMeSH KeywordsAnimalsBlotting, WesternCell DeathCells, CulturedDisease Models, AnimalDopamineHeterozygoteHomeodomain ProteinsImmunohistochemistryMesencephalonMiceMice, Inbred C57BLMice, KnockoutMitogen-Activated Protein Kinase 1Mitogen-Activated Protein Kinase 3MutationNerve Tissue ProteinsNeuronsParkinson DiseaseReceptors, Nerve Growth FactorReverse Transcriptase Polymerase Chain ReactionUp-RegulationConceptsMesDA neuronsBcl-2 familyAnti-apoptotic membersCell death signalsExpression levelsMutant embryosHaplotype variationDeath signalsMitochondrial stabilityEngrailedERK1/2 activityEngrailed-1Mitochondrial insultDopaminergic neuronsGenesPostnatal maintenanceReceptor geneElevated expressionMidbrain dopaminergic neuronsHeterozygote animalsDependent mannerMutant miceNigrostriatal dopaminergic systemERK1/2 suppressionMesencephalic dopaminergic neurons
2008
Separating Genetic and Hemodynamics Effects In Nrp1 Knockout Embryos
Jones E, Yuan L, Breant C, Watts R, Eichmann A. Separating Genetic and Hemodynamics Effects In Nrp1 Knockout Embryos. The FASEB Journal 2008, 22: 1143.2-1143.2. DOI: 10.1096/fasebj.22.1_supplement.1143.2.Peer-Reviewed Original Research
2006
GCN5 Functions in Telomere Maintenance and Neural Development
Dent S, Evrard Y, Lin W, Bu P, Phan H, Chang S, Multani A. GCN5 Functions in Telomere Maintenance and Neural Development. The FASEB Journal 2006, 20: a1472-a1472. DOI: 10.1096/fasebj.20.5.a1472-e.Peer-Reviewed Original ResearchMutant embryosNeural developmentTelomere maintenanceDouble mutant embryosCatalytic site mutationsNeural tube closureGcn5 functionsTelomere defectsTelomere fusionEmbryonic lethalityHypomorphic alleleProper expressionGCN5Tube closureSite mutationSimilar defectsEnd associationEmbryosFirst evidenceApoptosisBrain developmentP53CellsTelomeresGenes
2005
Otx2 Regulates Subtype Specification and Neurogenesis in the Midbrain
Vernay B, Koch M, Vaccarino F, Briscoe J, Simeone A, Kageyama R, Ang SL. Otx2 Regulates Subtype Specification and Neurogenesis in the Midbrain. Journal Of Neuroscience 2005, 25: 4856-4867. PMID: 15888661, PMCID: PMC6724764, DOI: 10.1523/jneurosci.5158-04.2005.Peer-Reviewed Original ResearchMeSH KeywordsAge FactorsAnimalsAnimals, NewbornBasic Helix-Loop-Helix Transcription FactorsBody PatterningBromodeoxyuridineCell CountCell DifferentiationDopamineEmbryo, MammalianEmbryonic InductionFibroblast Growth Factor 8Gene Expression Regulation, DevelopmentalHomeobox Protein Nkx-2.2Homeodomain ProteinsImmunohistochemistryIn Situ HybridizationIn Situ Nick-End LabelingIntermediate Filament ProteinsIntracellular Signaling Peptides and ProteinsKruppel-Like Transcription FactorsMembrane ProteinsMesencephalonMiceMice, TransgenicNerve Tissue ProteinsNestinNeuronsOrganizers, EmbryonicOtx Transcription FactorsPatched ReceptorsReceptors, Cell SurfaceSerotoninTranscription FactorsWnt1 ProteinZebrafish ProteinsZinc Finger Protein GLI1ConceptsMid-hindbrain organizerFunction of Otx2Neuronal subtype identityTranscription factor Otx2Rostral brain developmentEmbryonic day 10.5Cre/loxP systemRole of Otx2Neuronal progenitor cellsMutant embryosProgenitor identityConditional mutantsE10.5 onwardOtx2 activityAnterior hindbrainEctopic expressionCerebellar-like structuresSubtype specificationSubtype identityTransgenic animalsNovel roleLater roleNovel mouse modelOtx2Otx2 expressionMicroRNAs Regulate Brain Morphogenesis in Zebrafish
Giraldez AJ, Cinalli RM, Glasner ME, Enright AJ, Thomson JM, Baskerville S, Hammond SM, Bartel DP, Schier AF. MicroRNAs Regulate Brain Morphogenesis in Zebrafish. Science 2005, 308: 833-838. PMID: 15774722, DOI: 10.1126/science.1109020.Peer-Reviewed Original ResearchConceptsRNA-binding domainMultiple cell typesDicer mutantsMutant embryosSmall RNAsPrecursor miRNAsAxis formationMature miRNAsRibonuclease IIIHeart developmentGene expressionBrain morphogenesisAbnormal morphogenesisLater stepsMiRNA formationMorphogenesisMutantsMiRNAsCell typesBrain formationEssential roleZebrafishBrain defectsSomitogenesisGastrulation
2002
Progenitor cell maintenance requires numb and numblike during mouse neurogenesis
Petersen PH, Zou K, Hwang JK, Jan YN, Zhong W. Progenitor cell maintenance requires numb and numblike during mouse neurogenesis. Nature 2002, 419: 929-934. PMID: 12410312, DOI: 10.1038/nature01124.Peer-Reviewed Original ResearchConceptsMouse NumbNeural progenitor cellsMolecular mechanismsProgenitor cellsProgenitor cell maintenanceMammalian nervous systemOnset of neurogenesisConserved homologuesMutant embryosCourse of neurogenesisCell maintenanceMouse neurogenesisNeuronal fateNumbDividing cellsNeural cellsEarly neuronsNervous systemCritical roleProgenitorsCellsNeurogenesisDrosophilaTemporal patternsEmbryogenesis
1997
Differential expression of mammalian Numb, Numblike and Notch1 suggests distinct roles during mouse cortical neurogenesis
Zhong W, Jiang M, Weinmaster G, Jan L, Jan Y. Differential expression of mammalian Numb, Numblike and Notch1 suggests distinct roles during mouse cortical neurogenesis. Development 1997, 124: 1887-1897. PMID: 9169836, DOI: 10.1242/dev.124.10.1887.Peer-Reviewed Original ResearchMeSH KeywordsAmino Acid SequenceAnimalsBase SequenceCerebral CortexCloning, MolecularCytoplasmDrosophilaDrosophila ProteinsGene Expression Regulation, DevelopmentalGenesIntracellular Signaling Peptides and ProteinsJuvenile HormonesMembrane ProteinsMiceModels, NeurologicalMolecular Sequence DataMorphogenesisMutationNerve Tissue ProteinsNervous SystemNeuronsOrgan SpecificityReceptor, Notch1Receptors, Cell SurfaceRecombinant Fusion ProteinsRNA, MessengerSequence Homology, Amino AcidStem CellsTranscription FactorsConceptsDaughter cellsNeural precursorsMammalian cortical neurogenesisAsymmetric cell divisionSignificant sequence similarityMouse cortical neurogenesisCortical neurogenesisEmbryonic nervous systemFunction mutant embryosCell-cell interactionsCell-intrinsic mechanismsMammalian NumbMouse NumbNumb lossProgenitor cell proliferationDrosophila neurogenesisMammalian genesMutant embryosCell-extrinsic mechanismsSequence similarityCell divisionDifferential segregationNumblikePostmitotic neuronsNumb
1996
Decreased apoptosis in the brain and premature lethality in CPP32-deficient mice
Kuida K, Zheng T, Na S, Kuan C, Yang D, Karasuyama H, Rakic P, Flavell R. Decreased apoptosis in the brain and premature lethality in CPP32-deficient mice. Nature 1996, 384: 368-372. PMID: 8934524, DOI: 10.1038/384368a0.Peer-Reviewed Original ResearchConceptsCED-3Protease familyMajor morphogenetic changesProgrammed Cell DeathICE protease familyMutant embryosCaenorhabditis elegansDeath genesMorphogenetic cellApoptotic stimuliHomologous recombinationMorphogenetic changesMendelian geneticsSequence homologyHigh similarityCell deathPremature lethalitySupernumerary cellsEmbryonic day 12Mammalian brainCPP32Critical rolePostnatal stagesApoptosisBrain development
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