HUGOBELLENHUGO BELLEN29.71010310000000-95.397396100000003292BELLEN, HUGOInvestigator, Howard Hughes Medical Institute, Baylor College of Medicine
Professor, Departments of Molecular and Human Genetics and Neuroscience
Director, Program in Developmental Biology
I am interested in (1) providing a better fundamental understanding of the biology that governs the proper function and maintenance of neurons in aging adults (2) developing tools that can be applied to most genes to control transcript and protein levels in adult neurons to assess which proteins are required for neuronal survival and proper function (3) creating genome wide libraries to manipulate most genes in vivo. My lab uses the fruit fly Drosophila melanogaster as a model system because most biological processes are evolutionarily conserved and studies in fruit flies provide many important clues about the aging process in animals and human diseases.Distinguished Service Professorprns:coAuthorOfcoauthor ofprns:endDateend dateFaculty Rankprns:fullNamefull nameprns:grantAwardedBygrant awarded byprns:hasAuthorListauthor listprns:hasFacultyRankhas faculty rankprns:hasNetworkhas networkprns:hasPublicationVenuepublished inprns:informationResourceReferenceinformation resource referenceprns:isPrimaryPositionis primary positionprns:latitudelatitudeprns:longitudelongitudeprns:mainImagephotoprns:maxWeightmaximum weightprns:medlineTAjournal title abbreviationprns:meshDescriptorUIMeSH DescriptorUIprns:meshSemanticGroupNameMeSH semantic group nameprns:minWeightminimum weightprns:numberOfAuthorsnumber of authorsprns:numberOfConnectionsnumber of connectionsprns:numberOfPublicationsnumber of publicationsprns:personIdPerson IDprns:personInPrimaryPositionperson in primary positionprns:physicalNeighborOfphysical neighborprns:positionInDepartmentposition in departmentprns:positionInDivisionposition in divisionprns:predicateNodepredicate nodeprns:principalInvestigatorNameprincipal investigator nameprns:publicationDatepublication dateprns:similarTosimilar toprns:sortOrdersort orderprns:startDatestart dateprns:uniquenessWeightuniqueness weightprns:yearyearAcademic ArticleArticleDocumentbibo:pmidPubMed IdentifierAddressvivo:address1address line 1vivo:addressCitycityvivo:addressPostalCodepostal codevivo:addressStatestate or provinceAgreementvivo:authorInAuthorshipselected publicationsvivo:authorRankauthor rank in publicationAuthorshipDepartmentDivisionGrantvivo:hasMemberRolemember ofvivo:hasResearchArearesearch areasvivo:hasResearcherRoleresearch activitiesvivo:hrJobTitleHR job titleInformation Resourcevivo:linkAnchorTextlink anchor textvivo:linkedAuthorlinked authorvivo:linkedInformationResourcelinked information resourcevivo:mailingAddressmailing addressMember Rolevivo:memberRoleOfmember role ofvivo:overviewoverviewvivo:personInPositionpositionsPositionvivo:positionInOrganizationposition in organizationvivo:preferredTitlepreferred titleResearcher Rolevivo:researcherRoleOfresearcher role ofRolevivo:roleContributesTocontributes tovivo:sponsorAwardIdsponsor award idURLLinkvivo:webpagewebpagerdf:predicatepredicaterdf:typetyperdfs:labellabelConceptAgentfoaf:firstNamefirst nameGroupfoaf:lastNamelast nameOrganizationPersonAuthorship 1564491225313867Sandoval H, Yao CK, Chen K, Jaiswal M, Donti T, Lin YQ, Bayat V, Xiong B, Zhang K, David G, Charng WL, Yamamoto S, Duraine L, Graham BH, Bellen HJeLifeMitochondrial fusion but not fission regulates larval growth and synaptic development through steroid hormone production. Elife. 2014 Oct 14; 3.Elife2014-10-14T00:00:002014Mitochondrial fusion but not fission regulates larval growth and synaptic development through steroid hormone production.25451193Wong CO, Chen K, Lin YQ, Chao Y, Duraine L, Lu Z, Yoon WH, Sullivan JM, Broadhead GT, Sumner CJ, Lloyd TE, Macleod GT, Bellen HJ, Venkatachalam KNeuronA TRPV channel in Drosophila motor neurons regulates presynaptic resting Ca2+ levels, synapse growth, and synaptic transmission. Neuron. 2014 Nov 19; 84(4):764-77.Neuron2014-10-30T00:00:002014A TRPV channel in Drosophila motor neurons regulates presynaptic resting Ca2+ levels, synapse growth, and synaptic transmission.Authorship 813878Authorship 885106Authorship 8734Authorship 874604Authorship 939403Authorship 1083392Authorship 1136502Authorship 1172029Authorship 1176254Authorship 1225112Authorship 171781Authorship 206845Authorship 225978Authorship 16125211Authorship 1613699Authorship 16204715Authorship 1625473Authorship 1627991725594180Liu L, Zhang K, Sandoval H, Yamamoto S, Jaiswal M, Sanz E, Li Z, Hui J, Graham BH, Quintana A, Bellen HJCellGlial lipid droplets and ROS induced by mitochondrial defects promote neurodegeneration. Cell. 2015 Jan 15; 160(1-2):177-90.Cell2015-01-15T00:00:002015Glial lipid droplets and ROS induced by mitochondrial defects promote neurodegeneration.25624315Wangler MF, Yamamoto S, Bellen HJGeneticsFruit flies in biomedical research. Genetics. 2015 Mar; 199(3):639-53.Genetics2015-01-26T00:00:002015Fruit flies in biomedical research.25686248Rui YN, Xu Z, Patel B, Chen Z, Chen D, Tito A, David G, Sun Y, Stimming EF, Bellen HJ, Cuervo AM, Zhang SNature cell biologyHuntingtin functions as a scaffold for selective macroautophagy. Nat Cell Biol. 2015 Mar; 17(3):262-75.Nat Cell Biol2015-02-16T00:00:002015Huntingtin functions as a scaffold for selective macroautophagy.25811491Tian X, Gala U, Zhang Y, Shang W, Nagarkar Jaiswal S, di Ronza A, Jaiswal M, Yamamoto S, Sandoval H, Duraine L, Sardiello M, Sillitoe RV, Venkatachalam K, Fan H, Bellen HJ, Tong CPLoS biologyA voltage-gated calcium channel regulates lysosomal fusion with endosomes and autophagosomes and is required for neuronal homeostasis. PLoS Biol. 2015 Mar; 13(3):e1002103.PLoS Biol2015-03-26T00:00:002015A voltage-gated calcium channel regulates lysosomal fusion with endosomes and autophagosomes and is required for neuronal homeostasis.25824290Nagarkar-Jaiswal S, Lee PT, Campbell ME, Chen K, Anguiano-Zarate S, Gutierrez MC, Busby T, Lin WW, He Y, Schulze KL, Booth BW, Evans-Holm M, Venken KJ, Levis RW, Spradling AC, Hoskins RA, Bellen HJeLifeA library of MiMICs allows tagging of genes and reversible, spatial and temporal knockdown of proteins in Drosophila. Elife. 2015 Mar 31; 4.Elife2015-03-31T00:00:002015A library of MiMICs allows tagging of genes and reversible, spatial and temporal knockdown of proteins in Drosophila.Authorship 269813Authorship 526515Authorship 595222Authorship 71133Authorship 718459Authorship 722503Authorship 732296Authorship 760805Authorship 8123111Authorship 8434740Authorship 8652612Authorship 879606Authorship 8890711Authorship 924784OGUZKANCAOGUZ KANCA10276KANCA, OGUZInstructorAuthorship 2963132Authorship 29693810Authorship 29793910Authorship 2982781Authorship 2984542Authorship 2989275Authorship 30247513Authorship 3024767Authorship 3043899Authorship 3050641526020534Wang S, Tan KL, Agosto MA, Xiong B, Yamamoto S, Sandoval H, Jaiswal M, Bayat V, Zhang K, Charng WL, David G, Duraine L, Venkatachalam K, Wensel TG, Bellen HJPLoS biologyCorrection: the retromer complex is required for rhodopsin recycling and its loss leads to photoreceptor degeneration. PLoS Biol. 2015 May; 13(5):e1002170.PLoS Biol2015-05-28T00:00:002015Correction: the retromer complex is required for rhodopsin recycling and its loss leads to photoreceptor degeneration.26102525Nagarkar-Jaiswal S, DeLuca SZ, Lee PT, Lin WW, Pan H, Zuo Z, Lv J, Spradling AC, Bellen HJeLifeA genetic toolkit for tagging intronic MiMIC containing genes. Elife. 2015 Jun 23; 4.Elife2015-06-23T00:00:002015A genetic toolkit for tagging intronic MiMIC containing genes.26176594Jaiswal M, Haelterman NA, Sandoval H, Xiong B, Donti T, Kalsotra A, Yamamoto S, Cooper TA, Graham BH, Bellen HJPLoS biologyImpaired Mitochondrial Energy Production Causes Light-Induced Photoreceptor Degeneration Independent of Oxidative Stress. PLoS Biol. 2015 Jul; 13(7):e1002197.PLoS Biol2015-07-15T00:00:002015Impaired Mitochondrial Energy Production Causes Light-Induced Photoreceptor Degeneration Independent of Oxidative Stress.26199408Wang S, Bellen HJDevelopment (Cambridge, England)The retromer complex in development and disease. Development. 2015 Jul 15; 142(14):2392-6.Development2015-07-15T00:00:002015The retromer complex in development and disease.26229113Akbari OS, Bellen HJ, Bier E, Bullock SL, Burt A, Church GM, Cook KR, Duchek P, Edwards OR, Esvelt KM, Gantz VM, Golic KG, Gratz SJ, Harrison MM, Hayes KR, James AA, Kaufman TC, Knoblich J, Malik HS, Matthews KA, O'Connor-Giles KM, Parks AL, Perrimon N, Port F, Russell S, Ueda R, Wildonger JScience (New York, N.Y.)BIOSAFETY. Safeguarding gene drive experiments in the laboratory. Science. 2015 Aug 28; 349(6251):927-9.Science2015-07-30T00:00:002015BIOSAFETY. Safeguarding gene drive experiments in the laboratory.26289428Wang F, Jiang L, Chen Y, Haelterman NA, Bellen HJ, Chen RDatabase : the journal of biological databases and curationFlyVar: a database for genetic variation in Drosophila melanogaster. Database (Oxford). 2015; 2015.Database (Oxford)2015-08-19T00:00:002015FlyVar: a database for genetic variation in Drosophila melanogaster.26356605Wu SP, Kao CY, Wang L, Creighton CJ, Yang J, Donti TR, Harmancey R, Vasquez HG, Graham BH, Bellen HJ, Taegtmeyer H, Chang CP, Tsai MJ, Tsai SYNature communicationsIncreased COUP-TFII expression in adult hearts induces mitochondrial dysfunction resulting in heart failure. Nat Commun. 2015 Sep 10; 6:8245.Nat Commun2015-09-10T00:00:002015Increased COUP-TFII expression in adult hearts induces mitochondrial dysfunction resulting in heart failure.26406362Bellen HJ, Yamamoto SCellMorgan's legacy: fruit flies and the functional annotation of conserved genes. Cell. 2015 Sep 24; 163(1):12-4.Cell2015-09-24T00:00:002015Morgan's legacy: fruit flies and the functional annotation of conserved genes.26687360Tan L, Zhang KX, Pecot MY, Nagarkar-Jaiswal S, Lee PT, Takemura SY, McEwen JM, Nern A, Xu S, Tadros W, Chen Z, Zinn K, Bellen HJ, Morey M, Zipursky SLCellIg Superfamily Ligand and Receptor Pairs Expressed in Synaptic Partners in Drosophila. Cell. 2015 Dec 17; 163(7):1756-69.Cell2015-12-17T00:00:002015Ig Superfamily Ligand and Receptor Pairs Expressed in Synaptic Partners in Drosophila.26687361Carrillo RA, ?zkan E, Menon KP, Nagarkar-Jaiswal S, Lee PT, Jeon M, Birnbaum ME, Bellen HJ, Garcia KC, Zinn KCellControl of Synaptic Connectivity by a Network of Drosophila IgSF Cell Surface Proteins. Cell. 2015 Dec 17; 163(7):1770-1782.Cell2015-12-17T00:00:002015Control of Synaptic Connectivity by a Network of Drosophila IgSF Cell Surface Proteins.11790807Chen S, Zheng X, Schulze KL, Morris T, Bellen H, Stanley EFThe Journal of physiologyEnhancement of presynaptic calcium current by cysteine string protein. J Physiol. 2002 Jan 15; 538(Pt 2):383-9.J Physiol2002-01-15T00:00:002002Enhancement of presynaptic calcium current by cysteine string protein.12127745Verstreken P, Bellen HJTrends in neurosciencesMeaningless minis? Mechanisms of neurotransmitter-receptor clustering. Trends Neurosci. 2002 Aug; 25(8):383-5.Trends Neurosci2002-08-01T00:00:002002Meaningless minis? Mechanisms of neurotransmitter-receptor clustering.12932322Norga KK, Gurganus MC, Dilda CL, Yamamoto A, Lyman RF, Patel PH, Rubin GM, Hoskins RA, Mackay TF, Bellen HJCurrent biology : CBQuantitative analysis of bristle number in Drosophila mutants identifies genes involved in neural development. Curr Biol. 2003 Aug 19; 13(16):1388-96.Curr Biol2003-08-19T00:00:002003Quantitative analysis of bristle number in Drosophila mutants identifies genes involved in neural development.14665671Jafar-Nejad H, Acar M, Nolo R, Lacin H, Pan H, Parkhurst SM, Bellen HJGenes & developmentSenseless acts as a binary switch during sensory organ precursor selection. Genes Dev. 2003 Dec 01; 17(23):2966-78.Genes Dev2003-12-01T00:00:002003Senseless acts as a binary switch during sensory organ precursor selection.11751581Peter A, Sch?ttler P, Werner M, Beinert N, Dowe G, Burkert P, Mourkioti F, Dentzer L, He Y, Deak P, Benos PV, Gatt MK, Murphy L, Harris D, Barrell B, Ferraz C, Vidal S, Brun C, Demaille J, Cadieu E, Dreano S, Gloux S, Lelaure V, Mottier S, Galibert F, Borkova D, Mi?ana B, Kafatos FC, Bolshakov S, Sid?n-Kiamos I, Papagiannakis G, Spanos L, Louis C, Madue?o E, de Pablos B, Modolell J, Bucheton A, Callister D, Campbell L, Henderson NS, McMillan PJ, Salles C, Tait E, Valenti P, Saunders RD, Billaud A, Pachter L, Klapper R, Janning W, Glover DM, Ashburner M, Bellen HJ, J?ckle H, Sch?fer UEMBO reportsMapping and identification of essential gene functions on the X chromosome of Drosophila. EMBO Rep. 2002 Jan; 3(1):34-8.EMBO Rep2001-12-19T00:00:002001Mapping and identification of essential gene functions on the X chromosome of Drosophila.12160747Pennetta G, Hiesinger PR, Fabian-Fine R, Meinertzhagen IA, Bellen HJNeuronDrosophila VAP-33A directs bouton formation at neuromuscular junctions in a dosage-dependent manner. Neuron. 2002 Jul 18; 35(2):291-306.Neuron2002-07-18T00:00:002002Drosophila VAP-33A directs bouton formation at neuromuscular junctions in a dosage-dependent manner.74Professor10Assistant Professor54Instructor98Visiting Assistant Professor14Associate Professor6Adjunct Professor47Distinguished Service Professor15703278Escudero LM, Caminero E, Schulze KL, Bellen HJ, Modolell JDevelopment (Cambridge, England)Charlatan, a Zn-finger transcription factor, establishes a novel level of regulation of the proneural achaete/scute genes of Drosophila. Development. 2005 Mar; 132(6):1211-22.Development2005-02-09T00:00:002005Charlatan, a Zn-finger transcription factor, establishes a novel level of regulation of the proneural achaete/scute genes of Drosophila.One Baylor PlazaHouston, 77030TX15907473Hiesinger PR, Fayyazuddin A, Mehta SQ, Rosenmund T, Schulze KL, Zhai RG, Verstreken P, Cao Y, Zhou Y, Kunz J, Bellen HJCellThe v-ATPase V0 subunit a1 is required for a late step in synaptic vesicle exocytosis in Drosophila. Cell. 2005 May 20; 121(4):607-620.Cell2005-05-20T00:00:002005The v-ATPase V0 subunit a1 is required for a late step in synaptic vesicle exocytosis in Drosophila.15998804Dermaut B, Norga KK, Kania A, Verstreken P, Pan H, Zhou Y, Callaerts P, Bellen HJThe Journal of cell biologyAberrant lysosomal carbohydrate storage accompanies endocytic defects and neurodegeneration in Drosophila benchwarmer. J Cell Biol. 2005 Jul 04; 170(1):127-39.J Cell Biol2005-07-04T00:00:002005Aberrant lysosomal carbohydrate storage accompanies endocytic defects and neurodegeneration in Drosophila benchwarmer.16155582Wu S, Mehta SQ, Pichaud F, Bellen HJ, Quiocho FANature structural & molecular biologySec15 interacts with Rab11 via a novel domain and affects Rab11 localization in vivo. Nat Struct Mol Biol. 2005 Oct; 12(10):879-85.Nat Struct Mol Biol2005-09-11T00:00:002005Sec15 interacts with Rab11 via a novel domain and affects Rab11 localization in vivo.16230531Shroyer NF, Wallis D, Venken KJ, Bellen HJ, Zoghbi HYGenes & developmentGfi1 functions downstream of Math1 to control intestinal secretory cell subtype allocation and differentiation. Genes Dev. 2005 Oct 15; 19(20):2412-7.Genes Dev2005-10-15T00:00:002005Gfi1 functions downstream of Math1 to control intestinal secretory cell subtype allocation and differentiation.Authorship 32210214Authorship 32220614Authorship 32229513Authorship 3226813Authorship 3236771126812014David-Morrison G, Xu Z, Rui YN, Charng WL, Jaiswal M, Yamamoto S, Xiong B, Zhang K, Sandoval H, Duraine L, Zuo Z, Zhang S, Bellen HJDevelopmental cellWAC Regulates mTOR Activity by Acting as an Adaptor for the TTT and Pontin/Reptin Complexes. Dev Cell. 2016 Jan 25; 36(2):139-51.Dev Cell2016-01-25T00:00:002016WAC Regulates mTOR Activity by Acting as an Adaptor for the TTT and Pontin/Reptin Complexes.26935102Ugur B, Chen K, Bellen HJDisease models & mechanismsDrosophila tools and assays for the study of human diseases. Dis Model Mech. 2016 Mar; 9(3):235-44.Dis Model Mech2016-03-01T00:00:002016Drosophila tools and assays for the study of human diseases.27195754Li T, Fan J, Blanco-S?nchez B, Giagtzoglou N, Lin G, Yamamoto S, Jaiswal M, Chen K, Zhang J, Wei W, Lewis MT, Groves AK, Westerfield M, Jia J, Bellen HJPLoS geneticsUbr3, a Novel Modulator of Hh Signaling Affects the Degradation of Costal-2 and Kif7 through Poly-ubiquitination. PLoS Genet. 2016 05; 12(5):e1006054.PLoS Genet2016-05-19T00:00:002016Ubr3, a Novel Modulator of Hh Signaling Affects the Degradation of Costal-2 and Kif7 through Poly-ubiquitination.27254664Ali YO, Allen HM, Yu L, Li-Kroeger D, Bakhshizadehmahmoudi D, Hatcher A, McCabe C, Xu J, Bjorklund N, Taglialatela G, Bennett DA, De Jager PL, Shulman JM, Bellen HJ, Lu HCPLoS biologyNMNAT2:HSP90 Complex Mediates Proteostasis in Proteinopathies. PLoS Biol. 2016 06; 14(6):e1002472.PLoS Biol2016-06-02T00:00:002016NMNAT2:HSP90 Complex Mediates Proteostasis in Proteinopathies.27338814Chouhan AK, Guo C, Hsieh YC, Ye H, Senturk M, Zuo Z, Li Y, Chatterjee S, Botas J, Jackson GR, Bellen HJ, Shulman JMActa neuropathologica communicationsUncoupling neuronal death and dysfunction in Drosophila models of neurodegenerative disease. Acta Neuropathol Commun. 2016 06 23; 4(1):62.Acta Neuropathol Commun2016-06-23T00:00:002016Uncoupling neuronal death and dysfunction in Drosophila models of neurodegenerative disease.Authorship 3247258Authorship 32527112Authorship 325676827331610Li T, Giagtzoglou N, Eberl DF, Jaiswal SN, Cai T, Godt D, Groves AK, Bellen HJeLifeThe E3 ligase Ubr3 regulates Usher syndrome and MYH9 disorder proteins in the auditory organs of Drosophila and mammals. Elife. 2016 06 22; 5.Elife2016-06-22T00:00:002016The E3 ligase Ubr3 regulates Usher syndrome and MYH9 disorder proteins in the auditory organs of Drosophila and mammals.27343351Chen K, Lin G, Haelterman NA, Ho TS, Li T, Li Z, Duraine L, Graham BH, Jaiswal M, Yamamoto S, Rasband MN, Bellen HJeLifeLoss of Frataxin induces iron toxicity, sphingolipid synthesis, and Pdk1/Mef2 activation, leading to neurodegeneration. Elife. 2016 06 25; 5.Elife2016-06-25T00:00:002016Loss of Frataxin induces iron toxicity, sphingolipid synthesis, and Pdk1/Mef2 activation, leading to neurodegeneration.26931468Chao YH, Robak LA, Xia F, Koenig MK, Adesina A, Bacino CA, Scaglia F, Bellen HJ, Wangler MFHuman molecular geneticsMissense variants in the middle domain of DNM1L in cases of infantile encephalopathy alter peroxisomes and mitochondria when assayed in Drosophila. Hum Mol Genet. 2016 05 01; 25(9):1846-56.Hum Mol Genet2016-02-29T00:00:002016Missense variants in the middle domain of DNM1L in cases of infantile encephalopathy alter peroxisomes and mitochondria when assayed in Drosophila.17620409Koh TW, Korolchuk VI, Wairkar YP, Jiao W, Evergren E, Pan H, Zhou Y, Venken KJ, Shupliakov O, Robinson IM, O'Kane CJ, Bellen HJThe Journal of cell biologyEps15 and Dap160 control synaptic vesicle membrane retrieval and synapse development. J Cell Biol. 2007 Jul 16; 178(2):309-22.J Cell Biol2007-07-09T00:00:002007Eps15 and Dap160 control synaptic vesicle membrane retrieval and synapse development.12960394Zhai RG, Hiesinger PR, Koh TW, Verstreken P, Schulze KL, Cao Y, Jafar-Nejad H, Norga KK, Pan H, Bayat V, Greenbaum MP, Bellen HJProceedings of the National Academy of Sciences of the United States of AmericaMapping Drosophila mutations with molecularly defined P element insertions. Proc Natl Acad Sci U S A. 2003 Sep 16; 100(19):10860-5.Proc Natl Acad Sci U S A2003-09-05T00:00:002003Mapping Drosophila mutations with molecularly defined P element insertions.18391075Ly CV, Yao CK, Verstreken P, Ohyama T, Bellen HJThe Journal of cell biologystraightjacket is required for the synaptic stabilization of cacophony, a voltage-gated calcium channel alpha1 subunit. J Cell Biol. 2008 Apr 07; 181(1):157-70.J Cell Biol2008-04-07T00:00:002008straightjacket is required for the synaptic stabilization of cacophony, a voltage-gated calcium channel alpha1 subunit.15466176Kazanjian A, Wallis D, Au N, Nigam R, Venken KJ, Cagle PT, Dickey BF, Bellen HJ, Gilks CB, Grimes HLCancer researchGrowth factor independence-1 is expressed in primary human neuroendocrine lung carcinomas and mediates the differentiation of murine pulmonary neuroendocrine cells. Cancer Res. 2004 Oct 01; 64(19):6874-82.Cancer Res2004-10-01T00:00:002004Growth factor independence-1 is expressed in primary human neuroendocrine lung carcinomas and mediates the differentiation of murine pulmonary neuroendocrine cells.18809725Tien AC, Rajan A, Schulze KL, Ryoo HD, Acar M, Steller H, Bellen HJThe Journal of cell biologyEro1L, a thiol oxidase, is required for Notch signaling through cysteine bridge formation of the Lin12-Notch repeats in Drosophila melanogaster. J Cell Biol. 2008 Sep 22; 182(6):1113-25.J Cell Biol2008-09-22T00:00:002008Ero1L, a thiol oxidase, is required for Notch signaling through cysteine bridge formation of the Lin12-Notch repeats in Drosophila melanogaster.19465919Venken KJ, Carlson JW, Schulze KL, Pan H, He Y, Spokony R, Wan KH, Koriabine M, de Jong PJ, White KP, Bellen HJ, Hoskins RANature methodsVersatile P[acman] BAC libraries for transgenesis studies in Drosophila melanogaster. Nat Methods. 2009 Jun; 6(6):431-4.Nat Methods2009-06-01T00:00:002009Versatile P[acman] BAC libraries for transgenesis studies in Drosophila melanogaster.14988715Hiesinger PR, Bellen HJNature geneticsFlying in the face of total disruption. Nat Genet. 2004 Mar; 36(3):211-2.Nat Genet2004-03-01T00:00:002004Flying in the face of total disruption.15043825Bellen HJCurrent biology : CBHugo J. Bellen. Curr Biol. 2004 Mar 23; 14(6):R218.Curr Biol2004-03-23T00:00:002004Hugo J. Bellen.20066100Giagtzoglou N, Ly CV, Bellen HJCold Spring Harbor perspectives in biologyCell adhesion, the backbone of the synapse: "vertebrate" and "invertebrate" perspectives. Cold Spring Harb Perspect Biol. 2009 Oct; 1(4):a003079.Cold Spring Harb Perspect Biol2009-10-01T00:00:002009Cell adhesion, the backbone of the synapse: "vertebrate" and "invertebrate" perspectives.20383202Bellen HJ, Tong C, Tsuda HNature reviews. Neuroscience100 years of Drosophila research and its impact on vertebrate neuroscience: a history lesson for the future. Nat Rev Neurosci. 2010 Jul; 11(7):514-22.Nat Rev Neurosci2010-07-01T00:00:002010100 years of Drosophila research and its impact on vertebrate neuroscience: a history lesson for the future.16055061Verstreken P, Ly CV, Venken KJ, Koh TW, Zhou Y, Bellen HJNeuronSynaptic mitochondria are critical for mobilization of reserve pool vesicles at Drosophila neuromuscular junctions. Neuron. 2005 Aug 04; 47(3):365-78.Neuron2005-08-04T00:00:002005Synaptic mitochondria are critical for mobilization of reserve pool vesicles at Drosophila neuromuscular junctions.20832291Bayat V, Jaiswal M, Bellen HJCurrent opinion in neurobiologyThe BMP signaling pathway at the Drosophila neuromuscular junction and its links to neurodegenerative diseases. Curr Opin Neurobiol. 2011 Feb; 21(1):182-8.Curr Opin Neurobiol2010-09-09T00:00:002010The BMP signaling pathway at the Drosophila neuromuscular junction and its links to neurodegenerative diseases.Authorship 3281454927640307Harel T, Yoon WH, Garone C, Gu S, Coban-Akdemir Z, Eldomery MK, Posey JE, Jhangiani SN, Rosenfeld JA, Cho MT, Fox S, Withers M, Brooks SM, Chiang T, Duraine L, Erdin S, Yuan B, Shao Y, Moussallem E, Lamperti C, Donati MA, Smith JD, McLaughlin HM, Eng CM, Walkiewicz M, Xia F, Pippucci T, Magini P, Seri M, Zeviani M, Hirano M, Hunter JV, Srour M, Zanigni S, Lewis RA, Muzny DM, Lotze TE, Boerwinkle E, Baylor-Hopkins Center for Mendelian Genomics, University of Washington Center for Mendelian Genomics, Gibbs RA, Hickey SE, Graham BH, Yang Y, Buhas D, Martin DM, Potocki L, Graziano C, Bellen HJ, Lupski JRAmerican journal of human geneticsRecurrent De Novo and Biallelic Variation of ATAD3A, Encoding a Mitochondrial Membrane Protein, Results in Distinct Neurological Syndromes. Am J Hum Genet. 2016 Oct 06; 99(4):831-845.Am J Hum Genet2016-09-15T00:00:002016Recurrent De Novo and Biallelic Variation of ATAD3A, Encoding a Mitochondrial Membrane Protein, Results in Distinct Neurological Syndromes.15738961Venken KJ, Bellen HJNature reviews. GeneticsEmerging technologies for gene manipulation in Drosophila melanogaster. Nat Rev Genet. 2005 Mar; 6(3):167-78.Nat Rev Genet2005-03-01T00:00:002005Emerging technologies for gene manipulation in Drosophila melanogaster.16606693Seto ES, Bellen HJThe Journal of cell biologyInternalization is required for proper Wingless signaling in Drosophila melanogaster. J Cell Biol. 2006 Apr 10; 173(1):95-106.J Cell Biol2006-04-10T00:00:002006Internalization is required for proper Wingless signaling in Drosophila melanogaster.15848801Mehta SQ, Hiesinger PR, Beronja S, Zhai RG, Schulze KL, Verstreken P, Cao Y, Zhou Y, Tepass U, Crair MC, Bellen HJNeuronMutations in Drosophila sec15 reveal a function in neuronal targeting for a subset of exocyst components. Neuron. 2005 Apr 21; 46(2):219-32.Neuron2005-04-21T00:00:002005Mutations in Drosophila sec15 reveal a function in neuronal targeting for a subset of exocyst components.22017985Venken KJ, Simpson JH, Bellen HJNeuronGenetic manipulation of genes and cells in the nervous system of the fruit fly. Neuron. 2011 Oct 20; 72(2):202-30.Neuron2011-10-20T00:00:002011Genetic manipulation of genes and cells in the nervous system of the fruit fly.16122429Tsuda H, Jafar-Nejad H, Patel AJ, Sun Y, Chen HK, Rose MF, Venken KJ, Botas J, Orr HT, Bellen HJ, Zoghbi HYCellThe AXH domain of Ataxin-1 mediates neurodegeneration through its interaction with Gfi-1/Senseless proteins. Cell. 2005 Aug 26; 122(4):633-44.Cell2005-08-26T00:00:002005The AXH domain of Ataxin-1 mediates neurodegeneration through its interaction with Gfi-1/Senseless proteins.16137928Jafar-Nejad H, Andrews HK, Acar M, Bayat V, Wirtz-Peitz F, Mehta SQ, Knoblich JA, Bellen HJDevelopmental cellSec15, a component of the exocyst, promotes notch signaling during the asymmetric division of Drosophila sensory organ precursors. Dev Cell. 2005 Sep; 9(3):351-63.Dev Cell2005-09-01T00:00:002005Sec15, a component of the exocyst, promotes notch signaling during the asymmetric division of Drosophila sensory organ precursors.22764224Alves-Silva J, S?nchez-Soriano N, Beaven R, Klein M, Parkin J, Millard TH, Bellen HJ, Venken KJ, Ballestrem C, Kammerer RA, Prokop AThe Journal of neuroscience : the official journal of the Society for NeuroscienceSpectraplakins promote microtubule-mediated axonal growth by functioning as structural microtubule-associated proteins and EB1-dependent +TIPs (tip interacting proteins). J Neurosci. 2012 Jul 04; 32(27):9143-58.J Neurosci2012-07-04T00:00:002012Spectraplakins promote microtubule-mediated axonal growth by functioning as structural microtubule-associated proteins and EB1-dependent +TIPs (tip interacting proteins).17132048Zhai RG, Cao Y, Hiesinger PR, Zhou Y, Mehta SQ, Schulze KL, Verstreken P, Bellen HJPLoS biologyDrosophila NMNAT maintains neural integrity independent of its NAD synthesis activity. PLoS Biol. 2006 Nov; 4(12):e416.PLoS Biol2006-11-01T00:00:002006Drosophila NMNAT maintains neural integrity independent of its NAD synthesis activity.17138868Venken KJ, He Y, Hoskins RA, Bellen HJScience (New York, N.Y.)P[acman]: a BAC transgenic platform for targeted insertion of large DNA fragments in D. melanogaster. 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Am J Hum Genet. 2018 08 02; 103(2):245-260.Am J Hum Genet2018-07-26T00:00:002018IRF2BPL Is Associated with Neurological Phenotypes.Authorship 1080662D035583Disorders2323500.770924Rare DiseasesD029721Chemicals & Drugs2288520.604861Drosophila ProteinsAuthorship 91566313Authorship 9208582Authorship 9227762Authorship 9254239Authorship 93008910Authorship 93076510Authorship 93201612Authorship 93298320Authorship 93355212Authorship 9350214Authorship 93551129Authorship 93572731Authorship 93640943Authorship 9376494Authorship 9396054328978772Kanca O, Bellen HJ, Schnorrer FGeneticsGene Tagging Strategies To Assess Protein Expression, Localization, and Function in Drosophila. Genetics. 2017 10; 207(2):389-412.Genetics2017-10-01T00:00:002017Gene Tagging Strategies To Assess Protein Expression, Localization, and Function in Drosophila.29853544Li T, Bellen HJ, Groves AKDisease models & mechanismsUsing Drosophila to study mechanisms of hereditary hearing loss. 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N Engl J Med. 2018 11 29; 379(22):2131-2139.N Engl J Med2018-10-10T00:00:002018Effect of Genetic Diagnosis on Patients with Previously Undiagnosed Disease.2013-09-29NIHBELLEN, HUGO J.2010-09-30Mapping and rescuing EMS-induced lethal mutations on the Drosophila X ChromosomeRC4GM0963552021-05-31NIHBELLEN, HUGO J2003-05-01A Comprehensive Resource for Manipulating the Drosophila GenomeR01GM0678582001-06-30NIHBELLEN, HUGO J1996-07-01NEUROTRANSMITTER RELEASE IN DROSOPHILAR01GM0535712007-06-30NIHBELLEN, HUGO J2003-07-01Hrs and Endocytic Regulation of Notch and WinglessR01GM0689492013-04-30NIHBELLEN, HUGO J.2007-06-05Developmental Biology Training ProgramT32HD0552002010-11-30NIHBELLEN, HUGO J.2007-08-01Molecularly defined duplications for the X-chromosome and large autosomal genesR01GM0804152024-05-31NIHBELLEN, HUGO J2016-06-01A Human cDNA Library for Functional Gene Replacement in DrosophilaR24OD0220052007-06-30NIHBELLEN, HUGO J2000-07-01DEVELOPMENT BIOLOGY TRAINING PROGRAMT32ES0073322021-11-30NIHGROVES, ANDREW K2016-12-01A multi-species approach to find regulators of deafness genesR01DC0149322022-06-30NIHPERRIMON, NORBERT2018-07-01Using CRISPR technology to study the function of paralogous genesR24OD0264352023-06-30NIHBELLEN, HUGO J2015-09-15Center for functional analysis of human UDN gene homologs in Drosophila and zebrafishU54NS093793Principal InvestigatorPrincipal InvestigatorPrincipal InvestigatorCo-Principal InvestigatorPrincipal InvestigatorPrincipal InvestigatorPrincipal InvestigatorPrincipal InvestigatorPrincipal InvestigatorPrincipal InvestigatorCo-Principal InvestigatorD004330Living Beings2288180.663794DrosophilaDepartment of NeuroscienceDepartment of PediatricsDepartment of Molecular & Cellular BiologyDepartment of Molecular & Human GeneticsDepartment of Pathology & ImmunologyMolecular & Cellular BiologyMolecular & Human GeneticsNeurosciencePathologyPediatrics-NeurologyBaylor College of MedicineHUDAZOGHBIHUDA ZOGHBI29.71010310000000-95.397396100000002830ZOGHBI, HUDADistinguished Service ProfessorJAMESLUPSKIJAMES LUPSKI29.71073630000000-95.396604500000003140LUPSKI, JAMESProfessorRONALDDAVISRONALD DAVIS0.000000000000000.000000000000003208DAVIS, RONALDAdjunct ProfessorGRAEMEMARDONGRAEME MARDON0.000000000000000.000000000000003439MARDON, GRAEMEProfessor111groups17.40650.0106324851research areas12.04480.0161895124coauthor of391.5148.31660similar to11264selected publicationsMICHAELWANGLERMICHAEL WANGLER29.71009470000000-95.397401400000003839WANGLER, MICHAELAssociate ProfessorAuthorship 941645630804504Sent?rk M, Lin G, Zuo Z, Mao D, Watson E, Mikos AG, Bellen HJNature cell biologyUbiquilins regulate autophagic flux through mTOR signalling and lysosomal acidification. Nat Cell Biol. 2019 03; 21(3):384-396.Nat Cell Biol2019-02-25T00:00:002019Ubiquilins regulate autophagic flux through mTOR signalling and lysosomal acidification.HAMEDJAFAR-NEJADHAMED JAFAR-NEJAD29.71073630000000-95.396604500000003802JAFAR-NEJAD, HAMEDProfessorAuthorship 946203231032688Sent?rk M, Mao D, Bellen HJAutophagyLoss of proteins associated with amyotrophic lateral sclerosis affects lysosomal acidification via different routes. Autophagy. 2019 08; 15(8):1467-1469.Autophagy2019-04-28T00:00:002019Loss of proteins associated with amyotrophic lateral sclerosis affects lysosomal acidification via different routes.Authorship 1274264Authorship 1277153Authorship 128109723788431Giagtzoglou N, Li T, Yamamoto S, Bellen HJJournal of cell scienceDrosophila EHBP1 regulates Scabrous secretion during Notch-mediated lateral inhibition. J Cell Sci. 2013 Aug 15; 126(Pt 16):3686-96.J Cell Sci2013-06-20T00:00:002013Drosophila EHBP1 regulates Scabrous secretion during Notch-mediated lateral inhibition.23936219Yamamoto S, Bayat V, Bellen HJ, Tan CPloS oneProtein phosphatase 1? limits ring canal constriction during Drosophila germline cyst formation. PLoS One. 2013; 8(7):e70502.PLoS One2013-07-25T00:00:002013Protein phosphatase 1? limits ring canal constriction during Drosophila germline cyst formation.24039594Han SM, El Oussini H, Scekic-Zahirovic J, Vibbert J, Cottee P, Prasain JK, Bellen HJ, Dupuis L, Miller MAPLoS geneticsVAPB/ALS8 MSP ligands regulate striated muscle energy metabolism critical for adult survival in caenorhabditis elegans. 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Hum Mol Genet. 2019 11 21; 28(R2):R207-R214.Hum Mol Genet2019-11-21T00:00:002019The fruit fly at the interface of diagnosis and pathogenic mechanisms of rare and common human diseases.Authorship 9523882731327508Kanca O, Andrews JC, Lee PT, Patel C, Braddock SR, Slavotinek AM, Cohen JS, Gubbels CS, Aldinger KA, Williams J, Indaram M, Fatemi A, Yu TW, Agrawal PB, Vezina G, Simons C, Crawford J, Lau CC, Undiagnosed Diseases Network, Chung WK, Markello TC, Dobyns WB, Adams DR, Gahl WA, Wangler MF, Yamamoto S, Bellen HJ, Malicdan MCVAmerican journal of human geneticsDe Novo Variants in WDR37 Are Associated with Epilepsy, Colobomas, Dysmorphism, Developmental Delay, Intellectual Disability, and Cerebellar Hypoplasia. Am J Hum Genet. 2019 08 01; 105(2):413-424.Am J Hum Genet2019-07-18T00:00:002019De Novo Variants in WDR37 Are Associated with Epilepsy, Colobomas, Dysmorphism, Developmental Delay, Intellectual Disability, and Cerebellar Hypoplasia.Authorship 9538411331412248Ojelade SA, Lee TV, Giagtzoglou N, Yu L, Ugur B, Li Y, Duraine L, Zuo Z, Petyuk V, De Jager PL, Bennett DA, Arenkiel BR, Bellen HJ, Shulman JMCell reportscindr, the Drosophila Homolog of the CD2AP Alzheimer's Disease Risk Gene, Is Required for Synaptic Transmission and Proteostasis. Cell Rep. 2019 08 13; 28(7):1799-1813.e5.Cell Rep2019-08-13T00:00:002019cindr, the Drosophila Homolog of the CD2AP Alzheimer's Disease Risk Gene, Is Required for Synaptic Transmission and Proteostasis.Authorship 1076393Authorship 111064Authorship 9548123Authorship 9550432731491411Kanca O, Andrews JC, Lee PT, Patel C, Braddock SR, Slavotinek AM, Cohen JS, Gubbels CS, Aldinger KA, Williams J, Indaram M, Fatemi A, Yu TW, Agrawal PB, Vezina G, Simons C, Crawford J, Lau CC, Undiagnosed Diseases Network, Chung WK, Markello TC, Dobyns WB, Adams DR, Gahl WA, Wangler MF, Yamamoto S, Bellen HJ, Malicdan MCVAmerican journal of human geneticsDe Novo Variants in WDR37 Are Associated with Epilepsy, Colobomas, Dysmorphism, Developmental Delay, Intellectual Disability, and Cerebellar Hypoplasia. Am J Hum Genet. 2019 Sep 05; 105(3):672-674.Am J Hum Genet2019-09-05T00:00:002019De Novo Variants in WDR37 Are Associated with Epilepsy, Colobomas, Dysmorphism, Developmental Delay, Intellectual Disability, and Cerebellar Hypoplasia.31475990Wang J, Liu Z, Bellen HJ, Yamamoto SJournal of visualized experiments : JoVENavigating MARRVEL, a Web-Based Tool that Integrates Human Genomics and Model Organism Genetics Information. J Vis Exp. 2019 08 15; (150).J Vis Exp2019-08-15T00:00:002019Navigating MARRVEL, a Web-Based Tool that Integrates Human Genomics and Model Organism Genetics Information.Dr. Shaulsky's GroupMemberAuthorship 955597631524990Wang J, Mao D, Fazal F, Kim SY, Yamamoto S, Bellen H, Liu ZCurrent protocols in bioinformaticsUsing MARRVEL v1.2 for Bioinformatics Analysis of Human Genes and Variant Pathogenicity. Curr Protoc Bioinformatics. 2019 09; 67(1):e85.Curr Protoc Bioinformatics2019-09-01T00:00:002019Using MARRVEL v1.2 for Bioinformatics Analysis of Human Genes and Variant Pathogenicity.Authorship 95715121Authorship 9573026131616000Guo H, Bettella E, Marcogliese PC, Zhao R, Andrews JC, Nowakowski TJ, Gillentine MA, Hoekzema K, Wang T, Wu H, Jangam S, Liu C, Ni H, Willemsen MH, van Bon BW, Rinne T, Stevens SJC, Kleefstra T, Brunner HG, Yntema HG, Long M, Zhao W, Hu Z, Colson C, Richard N, Schwartz CE, Romano C, Castiglia L, Bottitta M, Dhar SU, Erwin DJ, Emrick L, Keren B, Afenjar A, Zhu B, Bai B, Stankiewicz P, Herman K, University of Washington Center for Mendelian Genomics, Mercimek-Andrews S, Juusola J, Wilfert AB, Abou Jamra R, B?ttner B, Mefford HC, Muir AM, Scheffer IE, Regan BM, Malone S, Gecz J, Cobben J, Weiss MM, Waisfisz Q, Bijlsma EK, Hoffer MJV, Ruivenkamp CAL, Sartori S, Xia F, Rosenfeld JA, Bernier RA, Wangler MF, Yamamoto S, Xia K, Stegmann APA, Bellen HJ, Murgia A, Eichler EENature communicationsDisruptive mutations in TANC2 define a neurodevelopmental syndrome associated with psychiatric disorders. Nat Commun. 2019 10 15; 10(1):4679.Nat Commun2019-10-15T00:00:002019Disruptive mutations in TANC2 define a neurodevelopmental syndrome associated with psychiatric disorders.31607425Ansar M, Chung HL, Al-Otaibi A, Elagabani MN, Ravenscroft TA, Paracha SA, Scholz R, Abdel Magid T, Sarwar MT, Shah SF, Qaisar AA, Makrythanasis P, Marcogliese PC, Kamsteeg EJ, Falconnet E, Ranza E, Santoni FA, Aldhalaan H, Al-Asmari A, Faqeih EA, Ahmed J, Kornau HC, Bellen HJ, Antonarakis SEAmerican journal of human geneticsBi-allelic Variants in IQSEC1 Cause Intellectual Disability, Developmental Delay, and Short Stature. Am J Hum Genet. 2019 11 07; 105(5):907-920.Am J Hum Genet2019-10-10T00:00:002019Bi-allelic Variants in IQSEC1 Cause Intellectual Disability, Developmental Delay, and Short Stature.Authorship 957806231647888Kanca O, Bellen HJNeuronLa CaSSA da Drosophila: A Versatile Expansion of the Tool Box. Neuron. 2019 10 23; 104(2):177-179.Neuron2019-10-23T00:00:002019La CaSSA da Drosophila: A Versatile Expansion of the Tool Box.Authorship 9582392031674908Kanca O, Zirin J, Garcia-Marques J, Knight SM, Yang-Zhou D, Amador G, Chung H, Zuo Z, Ma L, He Y, Lin WW, Fang Y, Ge M, Yamamoto S, Schulze KL, Hu Y, Spradling AC, Mohr SE, Perrimon N, Bellen HJeLifeAn efficient CRISPR-based strategy to insert small and large fragments of DNA using short homology arms. Elife. 2019 11 01; 8.Elife2019-11-01T00:00:002019An efficient CRISPR-based strategy to insert small and large fragments of DNA using short homology arms.true1Adjunct ProfessorAdjunct Professortrue1ProfessorProfessor31735666Link N, Chung H, Jolly A, Withers M, Tepe B, Arenkiel BR, Shah PS, Krogan NJ, Aydin H, Geckinli BB, Tos T, Isikay S, Tuysuz B, Mochida GH, Thomas AX, Clark RD, Mirzaa GM, Lupski JR, Bellen HJDevelopmental cellMutations in ANKLE2, a ZIKA Virus Target, Disrupt an Asymmetric Cell Division Pathway in Drosophila Neuroblasts to Cause Microcephaly. Dev Cell. 2019 12 16; 51(6):713-729.e6.Dev Cell2019-11-14T00:00:002019Mutations in ANKLE2, a ZIKA Virus Target, Disrupt an Asymmetric Cell Division Pathway in Drosophila Neuroblasts to Cause Microcephaly.31785787Wang J, Rousseau J, Kim E, Ehresmann S, Cheng YT, Duraine L, Zuo Z, Park YJ, Li-Kroeger D, Bi W, Wong LJ, Rosenfeld J, Gleeson J, Faqeih E, Alkuraya FS, Wierenga KJ, Chen J, Afenjar A, Nava C, Doummar D, Keren B, Juusola J, Grompe M, Bellen HJ, Campeau PMAmerican journal of human geneticsLoss of Oxidation Resistance 1, OXR1, Is Associated with an Autosomal-Recessive Neurological Disease with Cerebellar Atrophy and Lysosomal Dysfunction. Am J Hum Genet. 2019 12 05; 105(6):1237-1253.Am J Hum Genet2019-11-27T00:00:002019Loss of Oxidation Resistance 1, OXR1, Is Associated with an Autosomal-Recessive Neurological Disease with Cerebellar Atrophy and Lysosomal Dysfunction.31869524Bosch JA, Knight S, Kanca O, Zirin J, Yang-Zhou D, Hu Y, Rodiger J, Amador G, Bellen HJ, Perrimon N, Mohr SECurrent protocols in molecular biologyUse of the CRISPR-Cas9 System in Drosophila Cultured Cells to Introduce Fluorescent Tags into Endogenous Genes. Curr Protoc Mol Biol. 2020 Mar; 130(1):e112.Curr Protoc Mol Biol2020-03-01T00:00:002020Use of the CRISPR-Cas9 System in Drosophila Cultured Cells to Introduce Fluorescent Tags into Endogenous Genes.71Postdoc Fellows and AssociatesPostDocSHINYAYAMAMOTOSHINYA YAMAMOTO0.000000000000000.000000000000005831YAMAMOTO, SHINYAAssistant ProfessorGUANGLINGUANG LIN0.000000000000000.000000000000006341LIN, GUANG32197074Mao D, Reuter CM, Ruzhnikov MRZ, Beck AE, Farrow EG, Emrick LT, Rosenfeld JA, Mackenzie KM, Robak L, Wheeler MT, Burrage LC, Jain M, Liu P, Calame D, K?ry S, Sillesen M, Schmitz-Abe K, Tonduti D, Spaccini L, Iascone M, Genetti CA, Koenig MK, Graf M, Tran A, Alejandro M, Undiagnosed Diseases Network, Lee BH, Thiffault I, Agrawal PB, Bernstein JA, Bellen HJ, Chao HTAmerican journal of human geneticsDe novo EIF2AK1 and EIF2AK2 Variants Are Associated with Developmental Delay, Leukoencephalopathy, and Neurologic Decompensation. Am J Hum Genet. 2020 04 02; 106(4):570-583.Am J Hum Genet2020-03-19T00:00:002020De novo EIF2AK1 and EIF2AK2 Variants Are Associated with Developmental Delay, Leukoencephalopathy, and Neurologic Decompensation.32169171Chung HL, Wangler MF, Marcogliese PC, Jo J, Ravenscroft TA, Zuo Z, Duraine L, Sadeghzadeh S, Li-Kroeger D, Schmidt RE, Pestronk A, Rosenfeld JA, Burrage L, Herndon MJ, Chen S, Members of Undiagnosed Diseases Network, Shillington A, Vawter-Lee M, Hopkin R, Rodriguez-Smith J, Henrickson M, Lee B, Moser AB, Jones RO, Watkins P, Yoo T, Mar S, Choi M, Bucelli RC, Yamamoto S, Lee HK, Prada CE, Chae JH, Vogel TP, Bellen HJNeuronLoss- or Gain-of-Function Mutations in ACOX1 Cause Axonal Loss via Different Mechanisms. Neuron. 2020 05 20; 106(4):589-606.e6.Neuron2020-03-12T00:00:002020Loss- or Gain-of-Function Mutations in ACOX1 Cause Axonal Loss via Different Mechanisms.Authorship 98440819Authorship 9851159Authorship 98521135Authorship 98524430Authorship 98583820Authorship 98732310Authorship 98740514Authorship 9879691732286230Ye H, Ojelade SA, Li-Kroeger D, Zuo Z, Wang L, Li Y, Gu JY, Tepass U, Rodal AA, Bellen HJ, Shulman JMeLifeRetromer subunit, VPS29, regulates synaptic transmission and is required for endolysosomal function in the aging brain. Elife. 2020 04 14; 9.Elife2020-04-14T00:00:002020Retromer subunit, VPS29, regulates synaptic transmission and is required for endolysosomal function in the aging brain.32294302Li X, Yang L, Mao Z, Pan X, Zhao Y, Gu X, Eckel-Mahan K, Zuo Z, Tong Q, Hartig SM, Cheng X, Du G, Moore DD, Bellen HJ, Sesaki H, Sun KFASEB journal : official publication of the Federation of American Societies for Experimental BiologyNovel role of dynamin-related-protein 1 in dynamics of ER-lipid droplets in adipose tissue. FASEB J. 2020 06; 34(6):8265-8282.FASEB J2020-04-15T00:00:002020Novel role of dynamin-related-protein 1 in dynamics of ER-lipid droplets in adipose tissue.32330417Chung HL, Mao X, Wang H, Park YJ, Marcogliese PC, Rosenfeld JA, Burrage LC, Liu P, Murdock DR, Yamamoto S, Wangler MF, Undiagnosed Diseases Network, Chao HT, Long H, Feng L, Bacino CA, Bellen HJ, Xiao BAmerican journal of human geneticsDe Novo Variants in CDK19 Are Associated with a Syndrome Involving Intellectual Disability and Epileptic Encephalopathy. Am J Hum Genet. 2020 05 07; 106(5):717-725.Am J Hum Genet2020-04-23T00:00:002020De Novo Variants in CDK19 Are Associated with a Syndrome Involving Intellectual Disability and Epileptic Encephalopathy.Authorship 98815813Authorship 9884451832338762Huang Y, Mao X, van Jaarsveld RH, Shu L, Terhal PA, Jia Z, Xi H, Peng Y, Yan H, Yuan S, Li Q, Wang H, Bellen HJHuman molecular geneticsVariants in CAPZA2, a member of an F-actin capping complex, cause intellectual disability and developmental delay. Hum Mol Genet. 2020 06 03; 29(9):1537-1546.Hum Mol Genet2020-06-03T00:00:002020Variants in CAPZA2, a member of an F-actin capping complex, cause intellectual disability and developmental delay.32356556Dutta D, Briere LC, Kanca O, Marcogliese PC, Walker MA, High FA, Vanderver A, Krier J, Carmichael N, Callahan C, Taft RJ, Simons C, Helman G, Network UD, Wangler MF, Yamamoto S, Sweetser DA, Bellen HJHuman molecular geneticsDe novo mutations in TOMM70, a receptor of the mitochondrial import translocase, cause neurological impairment. Hum Mol Genet. 2020 06 03; 29(9):1568-1579.Hum Mol Genet2020-06-03T00:00:002020De novo mutations in TOMM70, a receptor of the mitochondrial import translocase, cause neurological impairment.Authorship 992605732579581Kao CY, Xu M, Wang L, Lin SC, Lee HJ, Duraine L, Bellen HJ, Goldstein DS, Tsai SY, Tsai MJPLoS geneticsElevated COUP-TFII expression in dopaminergic neurons accelerates the progression of Parkinson's disease through mitochondrial dysfunction. PLoS Genet. 2020 06; 16(6):e1008868.PLoS Genet2020-06-24T00:00:002020Elevated COUP-TFII expression in dopaminergic neurons accelerates the progression of Parkinson's disease through mitochondrial dysfunction.true1Postdoc Fellows and AssociatesPostdoc Fellows and AssociatesXueyangPanXueyang Pan15970Pan, XueyangPostdoc Fellows and AssociatesAuthorship 1116672Authorship 1170773Authorship 9969591332792353Ding X, Jo J, Wang CY, Cristobal CD, Zuo Z, Ye Q, Wirianto M, Lindeke-Myers A, Choi JM, Mohila CA, Kawabe H, Jung SY, Bellen HJ, Yoo SH, Lee HKGenes & developmentThe Daam2-VHL-Nedd4 axis governs developmental and regenerative oligodendrocyte differentiation. Genes Dev. 2020 09 01; 34(17-18):1177-1189.Genes Dev2020-08-13T00:00:002020The Daam2-VHL-Nedd4 axis governs developmental and regenerative oligodendrocyte differentiation.Authorship 999569932928889Ravenscroft TA, Janssens J, Lee PT, Tepe B, Marcogliese PC, Makhzami S, Holmes TC, Aerts S, Bellen HJThe Journal of neuroscience : the official journal of the Society for NeuroscienceDrosophila Voltage-Gated Sodium Channels Are Only Expressed in Active Neurons and Are Localized to Distal Axonal Initial Segment-like Domains. J Neurosci. 2020 10 14; 40(42):7999-8024.J Neurosci2020-09-14T00:00:002020Drosophila Voltage-Gated Sodium Channels Are Only Expressed in Active Neurons and Are Localized to Distal Axonal Initial Segment-like Domains.http://flypush.imgen.bcm.tmc.eduBellen Lab WebsiteAuthorship 10010402Authorship 10010411032988995Link N, Bellen HJDevelopment (Cambridge, England)Using Drosophila to drive the diagnosis and understand the mechanisms of rare human diseases. Development. 2020 09 28; 147(21).Development2020-09-28T00:00:002020Using Drosophila to drive the diagnosis and understand the mechanisms of rare human diseases.32989137Martelli F, Zhongyuan Z, Wang J, Wong CO, Karagas NE, Roessner U, Rupasinghe T, Venkatachalam K, Perry T, Bellen HJ, Batterham PProceedings of the National Academy of Sciences of the United States of AmericaLow doses of the neonicotinoid insecticide imidacloprid induce ROS triggering neurological and metabolic impairments in Drosophila. Proc Natl Acad Sci U S A. 2020 10 13; 117(41):25840-25850.Proc Natl Acad Sci U S A2020-09-28T00:00:002020Low doses of the neonicotinoid insecticide imidacloprid induce ROS triggering neurological and metabolic impairments in Drosophila.Authorship 10056334833232675Barish S, Barakat TS, Michel BC, Mashtalir N, Phillips JB, Valencia AM, Ugur B, Wegner J, Scott TM, Bostwick B, Undiagnosed Diseases Network, Murdock DR, Dai H, Perenthaler E, Nikoncuk A, van Slegtenhorst M, Brooks AS, Keren B, Nava C, Mignot C, Douglas J, Rodan L, Nowak C, Ellard S, Stals K, Lynch SA, Faoucher M, Lesca G, Edery P, Engleman KL, Zhou D, Thiffault I, Herriges J, Gass J, Louie RJ, Stolerman E, Washington C, Vetrini F, Otsubo A, Pratt VM, Conboy E, Treat K, Shannon N, Camacho J, Wakeling E, Yuan B, Chen CA, Rosenfeld JA, Westerfield M, Wangler M, Yamamoto S, Kadoch C, Scott DA, Bellen HJAmerican journal of human geneticsBICRA, a SWI/SNF Complex Member, Is Associated with BAF-Disorder Related Phenotypes in Humans and Model Organisms. Am J Hum Genet. 2020 12 03; 107(6):1096-1112.Am J Hum Genet2020-11-23T00:00:002020BICRA, a SWI/SNF Complex Member, Is Associated with BAF-Disorder Related Phenotypes in Humans and Model Organisms.Authorship 10066821333300868Cunningham KM, Maulding K, Ruan K, Senturk M, Grima JC, Sung H, Zuo Z, Song H, Gao J, Dubey S, Rothstein JD, Zhang K, Bellen HJ, Lloyd TEeLifeTFEB/Mitf links impaired nuclear import to autophagolysosomal dysfunction in C9-ALS. Elife. 2020 12 10; 9.Elife2020-12-10T00:00:002020TFEB/Mitf links impaired nuclear import to autophagolysosomal dysfunction in C9-ALS.true1InstructorInstructorAuthorship 114374true1ProfessorProfessorAuthorship 10164421033864376Luo X, Schoch K, Jangam SV, Bhavana VH, Graves HK, Kansagra S, Jasien JM, Stong N, Keren B, Mignot C, Ravelli C, Undiagnosed Diseases Network, Bellen HJ, Wangler MF, Shashi V, Yamamoto SHuman molecular geneticsRare deleterious de novo missense variants in Rnf2/Ring2 are associated with a neurodevelopmental disorder with unique clinical features. Hum Mol Genet. 2021 06 26; 30(14):1283-1292.Hum Mol Genet2021-06-26T00:00:002021Rare deleterious de novo missense variants in Rnf2/Ring2 are associated with a neurodevelopmental disorder with unique clinical features.Authorship 10179121133962631Baldridge D, Wangler MF, Bowman AN, Yamamoto S, Undiagnosed Diseases Network, Schedl T, Pak SC, Postlethwait JH, Shin J, Solnica-Krezel L, Bellen HJ, Westerfield MOrphanet journal of rare diseasesModel organisms contribute to diagnosis and discovery in the undiagnosed diseases network: current state and a future vision. Orphanet J Rare Dis. 2021 05 07; 16(1):206.Orphanet J Rare Dis2021-05-07T00:00:002021Model organisms contribute to diagnosis and discovery in the undiagnosed diseases network: current state and a future vision.Authorship 10208243134113007Ravenscroft TA, Phillips JB, Fieg E, Bajikar SS, Peirce J, Wegner J, Luna AA, Fox EJ, Yan YL, Rosenfeld JA, Zirin J, Kanca O, Undiagnosed Diseases Network, Benke PJ, Cameron ES, Strehlow V, Platzer K, Jamra RA, Kl?ckner C, Osmond M, Licata T, Rojas S, Dyment D, Chong JSC, Lincoln S, Stoler JM, Postlethwait JH, Wangler MF, Yamamoto S, Krier J, Westerfield M, Bellen HJGenetics in medicine : official journal of the American College of Medical GeneticsHeterozygous loss-of-function variants significantly expand the phenotypes associated with loss of GDF11. Genet Med. 2021 10; 23(10):1889-1900.Genet Med2021-06-10T00:00:002021Heterozygous loss-of-function variants significantly expand the phenotypes associated with loss of GDF11.2026-06-30NIHBELLEN, HUGO J;BOTAS, JUAN ;MILOSAVLJEVIC, ALEKSANDAR ;SHULMAN, JOSHUA M2021-07-01Functional Genomic Dissection of Alzheimer's Disease in Humans and Drosophila ModelsU01AG0724392025-03-31NIHBELLEN, HUGO J2021-07-01A Comprehensive Resource for Manipulating the Drosophila GenomeR24OD031447Co-Principal InvestigatorPrincipal Investigator2026-04-30NIHBELLEN, HUGO J ;WANGLER, MICHAEL FRANCIS2021-07-13Genomic medicine and gene function implementation for an underserved populationR01HG011795Principal InvestigatorAuthorship 10262784534314705Goodman LD, Cope H, Nil Z, Ravenscroft TA, Charng WL, Lu S, Tien AC, Pfundt R, Koolen DA, Haaxma CA, Veenstra-Knol HE, Wassink-Ruiter JSK, Wevers MR, Jones M, Walsh LE, Klee VH, Theunis M, Legius E, Steel D, Barwick KES, Kurian MA, Mohammad SS, Dale RC, Terhal PA, van Binsbergen E, Kirmse B, Robinette B, Cogn? B, Isidor B, Grebe TA, Kulch P, Hainline BE, Sapp K, Morava E, Klee EW, Macke EL, Trapane P, Spencer C, Si Y, Begtrup A, Moulton MJ, Dutta D, Kanca O, Undiagnosed Diseases Network, Wangler MF, Yamamoto S, Bellen HJ, Tan QKAmerican journal of human geneticsTNPO2 variants associate with human developmental delays, neurologic deficits, and dysmorphic features and alter TNPO2 activity in Drosophila. Am J Hum Genet. 2021 09 02; 108(9):1669-1691.Am J Hum Genet2021-07-26T00:00:002021TNPO2 variants associate with human developmental delays, neurologic deficits, and dysmorphic features and alter TNPO2 activity in Drosophila.Authorship 10279111334401677Park YJ, Kim S, Shim HP, Park JH, Lee G, Kim TY, Jo MC, Kwon AY, Lee M, Lee S, Yeo J, Chung HL, Bellen HJ, Kwon SH, Jeon SHiSciencePhosphatidylserine synthase plays an essential role in glia and affects development, as well as the maintenance of neuronal function. iScience. 2021 Aug 20; 24(8):102899.iScience2021-07-24T00:00:002021Phosphatidylserine synthase plays an essential role in glia and affects development, as well as the maintenance of neuronal function.true1Distinguished Service ProfessorDistinguished Service Professor2Distinguished Service ProfessorDistinguished Service Professor34418791Goodman LD, Bellen HJCurrent opinion in neurobiologyRecent insights into the role of glia and oxidative stress in Alzheimer's disease gained from Drosophila. Curr Opin Neurobiol. 2022 02; 72:32-38.Curr Opin Neurobiol2021-08-18T00:00:002021Recent insights into the role of glia and oxidative stress in Alzheimer's disease gained from Drosophila.Authorship 102859722026-05-31NIHBELLEN, HUGO J2021-09-01IMPACTS OF GLIAL LIPID DROPLETS ON OXIDATIVE STRESS AND NEURODEGENERATION IN ALZHEIMER'S DISEASER01AG073260Principal InvestigatorAuthorship 10342801934788397Manivannan SN, Roovers J, Smal N, Myers CT, Turkdogan D, Roelens F, Kanca O, Chung HL, Scholz T, Hermann K, Bierhals T, Caglayan HS, Stamberger H, MAE Working Group of EuroEPINOMICS RES Consortium, Mefford H, de Jonghe P, Yamamoto S, Weckhuysen S, Bellen HJBrain : a journal of neurologyDe novo FZR1 loss-of-function variants cause developmental and epileptic encephalopathies. Brain. 2022 06 03; 145(5):1684-1697.Brain2022-06-03T00:00:002022De novo FZR1 loss-of-function variants cause developmental and epileptic encephalopathies.Authorship 1181253Authorship 1173463Authorship 1184002Authorship 10359671034949639Moulton MJ, Barish S, Ralhan I, Chang J, Goodman LD, Harland JG, Marcogliese PC, Johansson JO, Ioannou MS, Bellen HJProceedings of the National Academy of Sciences of the United States of AmericaNeuronal ROS-induced glial lipid droplet formation is altered by loss of Alzheimer's disease-associated genes. Proc Natl Acad Sci U S A. 2021 12 28; 118(52).Proc Natl Acad Sci U S A2021-12-28T00:00:002021Neuronal ROS-induced glial lipid droplet formation is altered by loss of Alzheimer's disease-associated genes.Authorship 10385351Authorship 10392066Authorship 104010221Authorship 104089713Authorship 104115529Authorship 10411989Authorship 10412891Authorship 104132416Authorship 104139117Authorship 10416321535218524Accogli A, Lu S, Musante I, Scudieri P, Rosenfeld JA, Severino M, Baldassari S, Iacomino M, Riva A, Balagura G, Piccolo G, Minetti C, Roberto D, Xia F, Razak R, Lawrence E, Hussein M, Chang EY, Holick M, Cal? E, Aliberto E, De-Sarro R, Gambardella A, Network UD, Group SS, Emrick L, McCaffery PJA, Clagett-Dame M, Marcogliese PC, Bellen HJ, Lalani SR, Zara F, Striano P, Salpietro VCerebellum (London, England)Loss of Neuron Navigator 2 Impairs Brain and Cerebellar Development. Cerebellum. 2023 Apr; 22(2):206-222.Cerebellum2022-02-26T00:00:002022Loss of Neuron Navigator 2 Impairs Brain and Cerebellar Development.35224820Harnish JM, Li L, Rogic S, Poirier-Morency G, Kim SY, Undiagnosed Diseases Network, Boycott KM, Wangler MF, Bellen HJ, Hieter P, Pavlidis P, Liu Z, Yamamoto SHuman mutationModelMatcher: A scientist-centric online platform to facilitate collaborations between stakeholders of rare and undiagnosed disease research. Hum Mutat. 2022 06; 43(6):743-759.Hum Mutat2022-03-24T00:00:002022ModelMatcher: A scientist-centric online platform to facilitate collaborations between stakeholders of rare and undiagnosed disease research.35231122Bellen HJ, Hubbard EJA, Lehmann R, Madhani HD, Solnica-Krezel L, Southard-Smith EMDevelopment (Cambridge, England)Model organism databases are in jeopardy. Development. 2021 10 01; 148(19).Development2021-09-27T00:00:002021Model organism databases are in jeopardy.35302163Bellen HJDisease models & mechanismsLord of the fruit flies: an interview with Hugo Bellen. Dis Model Mech. 2022 03 01; 15(3).Dis Model Mech2022-03-01T00:00:002022Lord of the fruit flies: an interview with Hugo Bellen.35191376Martelli F, Hernandes NH, Zuo Z, Wang J, Wong CO, Karagas NE, Roessner U, Rupasinghe T, Robin C, Venkatachalam K, Perry T, Batterham P, Bellen HJeLifeLow doses of the organic insecticide spinosad trigger lysosomal defects, elevated ROS, lipid dysregulation, and neurodegeneration in flies. Elife. 2022 02 22; 11.Elife2022-02-22T00:00:002022Low doses of the organic insecticide spinosad trigger lysosomal defects, elevated ROS, lipid dysregulation, and neurodegeneration in flies.35101966Cristobal CD, Wang CY, Zuo Z, Smith JA, Lindeke-Myers A, Bellen HJ, Lee HKThe Journal of neuroscience : the official journal of the Society for NeuroscienceDaam2 Regulates Myelin Structure and the Oligodendrocyte Actin Cytoskeleton through Rac1 and Gelsolin. J Neurosci. 2022 03 02; 42(9):1679-1691.J Neurosci2022-01-31T00:00:002022Daam2 Regulates Myelin Structure and the Oligodendrocyte Actin Cytoskeleton through Rac1 and Gelsolin.35348658Huang Y, Ma M, Mao X, Pehlivan D, Kanca O, Un-Candan F, Shu L, Akay G, Mitani T, Lu S, Candan S, Wang H, Xiao B, Lupski JR, Bellen HJHuman molecular geneticsNovel dominant and recessive variants in human ROBO1 cause distinct neurodevelopmental defects through different mechanisms. Hum Mol Genet. 2022 08 23; 31(16):2751-2765.Hum Mol Genet2022-08-23T00:00:002022Novel dominant and recessive variants in human ROBO1 cause distinct neurodevelopmental defects through different mechanisms.35234901Chung HL, Rump P, Lu D, Glassford MR, Mok JW, Fatih J, Basal A, Marcogliese PC, Kanca O, Rapp M, Fock JM, Kamsteeg EJ, Lupski JR, Larson A, Haninbal MC, Bellen H, Harel THuman molecular geneticsDe novo variants in EMC1 lead to neurodevelopmental delay and cerebellar degeneration and affect glial function in Drosophila. Hum Mol Genet. 2022 09 29; 31(19):3231-3244.Hum Mol Genet2022-09-29T00:00:002022De novo variants in EMC1 lead to neurodevelopmental delay and cerebellar degeneration and affect glial function in Drosophila.35240055Lu S, Hernan R, Marcogliese PC, Huang Y, Gertler TS, Akcaboy M, Liu S, Chung HL, Pan X, Sun X, Oguz MM, Oztoprak U, de Baaij JHF, Ivanisevic J, McGinnis E, Guillen Sacoto MJ, Chung WK, Bellen HJAmerican journal of human geneticsLoss-of-function variants in TIAM1 are associated with developmental delay, intellectual disability, and seizures. 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