2022
Two neuronal peptides encoded from a single transcript regulate mitochondrial complex III in Drosophila
Bosch J, Ugur B, Pichardo-Casas I, Rabasco J, Escobedo F, Zuo Z, Brown B, Celniker S, Sinclair D, Bellen H, Perrimon N. Two neuronal peptides encoded from a single transcript regulate mitochondrial complex III in Drosophila. ELife 2022, 11: e82709. PMID: 36346220, PMCID: PMC9681215, DOI: 10.7554/elife.82709.Peer-Reviewed Original ResearchConceptsSmall open reading framesClasses of genesShares sequence similarityOpen reading frameSequence similarityBicistronic transcriptBiological functionsPhenotypic analysisMitochondrial functionImportant regulatorThousands of peptidesNeuronal functionGenesWealth of informationTranscriptsAnimal lethalityPeptidesRecent studiesParalogsDrosophilaSmORFsMitochondriaRegulatorRegulatesNeuronal peptides
2019
Ubiquilins regulate autophagic flux through mTOR signalling and lysosomal acidification
Şentürk M, Lin G, Zuo Z, Mao D, Watson E, Mikos A, Bellen H. Ubiquilins regulate autophagic flux through mTOR signalling and lysosomal acidification. Nature Cell Biology 2019, 21: 384-396. PMID: 30804504, PMCID: PMC6534127, DOI: 10.1038/s41556-019-0281-x.Peer-Reviewed Original ResearchMeSH KeywordsAmyotrophic Lateral SclerosisAnimalsAnimals, Genetically ModifiedAutophagyCarrier ProteinsCell Cycle ProteinsDrosophila melanogasterDrosophila ProteinsGene Expression Regulation, DevelopmentalHEK293 CellsHumansHydrogen-Ion ConcentrationLysosomesMutationNervous SystemSignal TransductionTOR Serine-Threonine KinasesConceptsAutophagic fluxDefective autophagic fluxEndoplasmic reticulum stressReticulum stressRegulator of autophagyConserved roleAmyotrophic lateral sclerosisMammalian cellsProteasomal degradationImpaired proteostasisDemise of neuronsUbiquilinLysosome acidificationFamilial amyotrophic lateral sclerosisLysosomal acidificationATPase activityMTORMutantsAutophagyDrosophilaProteostasisAcidificationCommon featureGenesLateral sclerosis
2018
An expanded toolkit for gene tagging based on MiMIC and scarless CRISPR tagging in Drosophila
Li-Kroeger D, Kanca O, Lee P, Cowan S, Lee M, Jaiswal M, Salazar J, He Y, Zuo Z, Bellen H. An expanded toolkit for gene tagging based on MiMIC and scarless CRISPR tagging in Drosophila. ELife 2018, 7: e38709. PMID: 30091705, PMCID: PMC6095692, DOI: 10.7554/elife.38709.Peer-Reviewed Original ResearchConceptsNew genetic toolsDouble headerTagged alleleGene taggingPrecise gene manipulationEndogenous genesGenetic toolsProtein functionProtein localizationMultiple downstream applicationsArtificial exonCassette exchangeProtein trappingEmbryo injectionNull allelesExpression patternsGene manipulationGene expressionDNA fragmentsDominant markersGenesCRISPRIntegration efficiencyDownstream applicationsAllelesIRF2BPL Is Associated with Neurological Phenotypes
Marcogliese P, Shashi V, Spillmann R, Stong N, Rosenfeld J, Koenig M, Martínez-Agosto J, Herzog M, Chen A, Dickson P, Lin H, Vera M, Salamon N, Graham J, Ortiz D, Infante E, Steyaert W, Dermaut B, Poppe B, Chung H, Zuo Z, Lee P, Kanca O, Xia F, Yang Y, Smith E, Jasien J, Kansagra S, Spiridigliozzi G, El-Dairi M, Lark R, Riley K, Koeberl D, Golden-Grant K, Diseases P, Callens S, Coucke P, Dermaut B, Hemelsoet D, Poppe B, Steyaert W, Terryn W, Van Coster R, Network U, Adams D, Alejandro M, Allard P, Azamian M, Bacino C, Balasubramanyam A, Barseghyan H, Batzli G, Beggs A, Behnam B, Bican A, Bick D, Birch C, Bonner D, Boone B, Bostwick B, Briere L, Brown D, Brush M, Burke E, Burrage L, Chen S, Clark G, Coakley T, Cogan J, Cooper C, Cope H, Craigen W, D’Souza P, Davids M, Dayal J, Dell’Angelica E, Dhar S, Dillon A, Dipple K, Donnell-Fink L, Dorrani N, Dorset D, Douine E, Draper D, Eckstein D, Emrick L, Eng C, Eskin A, Esteves C, Estwick T, Ferreira C, Fogel B, Friedman N, Gahl W, Glanton E, Godfrey R, Goldstein D, Gould S, Gourdine J, Groden C, Gropman A, Haendel M, Hamid R, Hanchard N, Handley L, Herzog M, Holm I, Hom J, Howerton E, Huang Y, Jacob H, Jain M, Jiang Y, Johnston J, Jones A, Kohane I, Krasnewich D, Krieg E, Krier J, Lalani S, Lau C, Lazar J, Lee B, Lee H, Levy S, Lewis R, Lincoln S, Lipson A, Loo S, Loscalzo J, Maas R, Macnamara E, MacRae C, Maduro V, Majcherska M, Malicdan M, Mamounas L, Manolio T, Markello T, Marom R, Martínez-Agosto J, Marwaha S, May T, McConkie-Rosell A, McCormack C, McCray A, Might M, Moretti P, Morimoto M, Mulvihill J, Murphy J, Muzny D, Nehrebecky M, Nelson S, Newberry J, Newman J, Nicholas S, Novacic D, Orange J, Pallais J, Palmer C, Papp J, Parker N, Pena L, Phillips J, Posey J, Postlethwait J, Potocki L, Pusey B, Reuter C, Robertson A, Rodan L, Rosenfeld J, Sampson J, Samson S, Schoch K, Schroeder M, Scott D, Sharma P, Shashi V, Signer R, Silverman E, Sinsheimer J, Smith K, Spillmann R, Splinter K, Stoler J, Stong N, Sullivan J, Sweetser D, Tifft C, Toro C, Tran A, Urv T, Valivullah Z, Vilain E, Vogel T, Wahl C, Walley N, Walsh C, Ward P, Waters K, Westerfield M, Wise A, Wolfe L, Worthey E, Yamamoto S, Yang Y, Yu G, Zastrow D, Zheng A, Yamamoto S, Wangler M, Mirzaa G, Hemelsoet D, Lee B, Nelson S, Goldstein D, Bellen H, Pena L. IRF2BPL Is Associated with Neurological Phenotypes. American Journal Of Human Genetics 2018, 103: 245-260. PMID: 30057031, PMCID: PMC6081494, DOI: 10.1016/j.ajhg.2018.07.006.Peer-Reviewed Original ResearchMissense variantsRange of phenotypesNeurological phenotypeProper neuronal functionNonsense variantPopulation genomicsModel organismsTranscriptional regulatorsFunction allelesPartial knockdownEctopic expressionRNA interferenceNonsense allelesBiological functionsMendelian diseasesDamaging heterozygous variantsGenesIRF2BPLNeuronal functionPhenotypeAdditional individualsComplete lossNervous systemMild phenotypeAllelesA gene-specific T2A-GAL4 library for Drosophila
Lee P, Zirin J, Kanca O, Lin W, Schulze K, Li-Kroeger D, Tao R, Devereaux C, Hu Y, Chung V, Fang Y, He Y, Pan H, Ge M, Zuo Z, Housden B, Mohr S, Yamamoto S, Levis R, Spradling A, Perrimon N, Bellen H. A gene-specific T2A-GAL4 library for Drosophila. ELife 2018, 7: e35574. PMID: 29565247, PMCID: PMC5898912, DOI: 10.7554/elife.35574.Peer-Reviewed Original ResearchConceptsFunction phenotypesIntrons of genesExpression of hundredsCell-type specificityLethal insertionsEssential genesGene functionEndogenous promoterLethal mutationsCDNA constructsGene expressionInserted cassettesGenesPowerful resourceExpressionPhenotypeSignal 3DrosophilaGAL4Severe lossIntronsChromosomesTranscriptionCRISPRPromoter
2015
A genetic toolkit for tagging intronic MiMIC containing genes
Nagarkar-Jaiswal S, DeLuca S, Lee P, Lin W, Pan H, Zuo Z, Lv J, Spradling A, Bellen H. A genetic toolkit for tagging intronic MiMIC containing genes. ELife 2015, 4: e08469. PMID: 26102525, PMCID: PMC4499919, DOI: 10.7554/elife.08469.Peer-Reviewed Original ResearchConceptsProtein interaction partnersGenes/proteinsProtein expression patternsUncharacterized genesGenetic toolkitIntegration cassetteMiMIC insertionsPhiC31 recombinaseProtein tagsFRT sequencesNew exonsGenetic strategiesInteraction partnersExpression patternsGenesExonsPlasmid DNAProteinMass specTagsIntronsLarge collectionGFPImmunoprecipitationMimics