Ma M, Ganapathi M, Zheng Y, Tan KL, Kanca O, Bove KE, Quintanilla N, Sag SO, Temel SG, LeDuc CA, McPartland AJ, Pereira EM, Shen Y, Hagen J, Thomas CP, Galvan NTN, Pan X, Lu S, Rosenfeld JA, Calame DG, Wangler MF, Lupski JR, Pehlivan D, Hertel PM, Chung WK, Bellen HJ (2024) Homozygous missense variants in YKT6 result in loss of function and are associated with developmental delay, with or without severe infantile liver disease and risk for hepatocellular carcinoma. Genetics in Medicine :101125. [Abstract]
Pan X, Tao AM, Lu S, Ma M, Hannan SB, Slaugh R, Drewes Williams S, O'Grady L, Kanca O, Person R, Carter MT, Platzer K, Schnabel F, Abou Jamra R, Roberts AE, Newburger JW, Revah-Politi A, Granadillo JL, Stegmann APA, Sinnema M, Accogli A, Salpietro V, Capra V, Ghaloul-Gonzalez L, Brueckner M, Simon MEH, Sweetser DA, Glinton KE, Kirk SE; Baylor College of Medicine Center for Precision Medicine Models; Wangler MF, Yamamoto S, Chung WK, Bellen HJ (2024) De novo variants in FRYL are associated with developmental delay, intellectual disability, and dysmorphic features. American Journal of Human Genetics 111(4):742-760. [Abstract]
Martelli F, Lin J, Mele S, Imlach W, Kanca O, Barlow CK, Paril J, Schittenhelm RB, Christodoulou J, Bellen HJ, Piper MDW, Johnson TK (2024) Identifying potential dietary treatments for inherited metabolic disorders using Drosophila nutrigenomics. Cell Reports 43(3):113861. [Abstract]
Dutta D, Kanca O, Shridharan RV, Marcogliese PC, Steger B, Morimoto M, Frost FG, Macnamara E; Undiagnosed Diseases Network; Wangler MF, Yamamoto S, Jenny A, Adams D, Malicdan MC, Bellen HJ (2024) Loss of the endoplasmic reticulum protein Tmem208 affects cell polarity, development, and viability. Proceedings of the National Academy of Science Sci U S A 121(9):e232258212. [Abstract]
Ma M, Zheng Y, Lu S, Pan X, Worley KC, Burrage LC, Blieden LS, Allworth A, Chen WL, Merla G, Mandriani B, Rosenfeld JA, Li-Kroeger D, Dutta D, Yamamoto S, Wangler MF; Undiagnosed Diseases Network; Glass IA, Strohbehn S, Blue E, Prontera P, Lalani SR, Bellen HJ (2024) De novo variants in PLCG1 are associated with hearing impairment, ocular pathology, and cardiac defects. medRxiv :10.1101/2024.01.08.23300523. [Abstract]
Goodman LD, Moulton MJ, Bellen HJ (2024) Glial lipid droplets resolve ROS during sleep. Nat Neurosci 27(4):610-612. [Abstract]
Wilson KA, Bar S, Dammer EB, Carrera EM, Hodge BA, Hilsabeck TAU, Bons J, Brownridge GW 3rd, Beck JN, Rose J, Granath-Panelo M, Nelson CS, Qi G, Gerencser AA, Lan J, Afenjar A, Chawla G, Brem RB, Campeau PM, Bellen HJ, Schilling B, Seyfried NT, Ellerby LM, Kapahi P (2024) OXR1 maintains the retromer to delay brain aging under dietary restriction. Nat Commun 15(1):467. [Abstract]
Pan X, Alvarez AN, Ma M, Lu S, Crawford MW, Briere LC, Kanca O, Yamamoto S, Sweetser DA, Wilson JL, Napier RJ, Pruneda JN#, Bellen HJ# (2023) Allelic strengths of encephalopathy-associated UBA5 variants correlate between in vivo and in vitro assays.. eLife 12:RP89891. # corresponding author [Abstract]
Nil Z*, Deshwar AR*, Huang Y, Barish S, Zhang X, Choufani S, Stabej PLQ, Hayes I, Yap P, Haldeman-Englert C, Wilson C, Prescott T, Tveten K, Vollo A, Haynes D, Wheeler PG, Zon J, Cytrynbaum C, Jobling R, Blyth M, Banka S, Afenjar A, Mignot C, Robin-Renaldo F, Keren B, Kanca O, Mao X, Wegner DJ, Sisco K, Shinawi M, Undiagnosed Disease Network, Wangler MF, Weksberg R, Yamamoto S, Costain G#, Bellen HJ# (2023) Rare de novo gain-of-function missense variants in DOT1L are associated with developmental delay and congenital anomalies. American Journal of Human Genetics 110(11):1919-1937. *equal contribution, # corresponding authors [Abstract]
Dutta D, Kanca O, Byeon SK, Marcogliese PC, Zuo Z, Shridharan RV, Park JH, Undiagnosed Diseases Network, Lin G, Ge M, Heimer G, Kohler JN, Wheeler MT, Kaipparettu BA, Pandey A, Bellen HJ (2023) A defect in mitochondrial fatty acid synthesis impairs iron metabolism and causes elevated ceramide levels. Nature Metabolism 5(9):1595-1614. [Abstract]
Wang CY, Zuo Z, Jo J, Kim KI, Madamba C, Ye Q, Jung SY, Bellen HJ, Lee HK (2023) Daam2 phosphorylation by CK2alpha negatively regulates Wnt activity during white matter development and injury. Proceedings of the National Academy of Sciences 120(35):e2304112120. [Abstract]
Yamamoto S#, Kanca O#, Wangler MF *, Bellen HJ* (2023) Integrating non-mammaliam model organism in the diagnosis of rare genetic human disease. Nature Review Genetics :doi: 10.1038/s41576-023-00633-6. *equal contribution, # corresponding authors [Abstract]
Guichard A*, Lu S*, Kanca O*, Bressan D, Huang Y, Ma M, Sanz Juste S, Andrews JC, Jay K, Sneider M, Schwartz R, Huang MC, Bei D, Pan H, Ma L, Lin W-W, Auradkar A, Bhagwat P, Park S, Wan KH, Ohsako T, Takano-Shimizu T, Celniker SE, Wangler MF, Yamamoto S#, Bellen HJ#, Bier E# (2023) A comprehensive Drosophila resource to identify key functional interactions between SARS-CoV-2 factors and host proteins. Cell Reports 42(8):112842. *equal contribution, # corresponding authors [Abstract]
Lu TC, Brbic M, Park YJ, Jackson T, Chen J, Kolluru SS, Qi Y, Katheder NS, Cai XT, Lee S, Chen YC, Auld N, Liang CY, Ding SH, Welsch D, D'Souza S, Pisco AO, Jones RC, Leskovec J, Lai EC, Bellen HJ, Luo L, Jasper H, Quake SR, Li H (2023) Aging Fly Cell Atlas identifies exhaustive aging features at cellular resolution. Science 380(6650):eadg0934. [Abstract]
Mao D, Liu C, Wang L, AI-Ouran R, Deisseroth C, Pasupuleti S, Kim SY, Li L, Rosenfeld JA, Meng L, Burrage LC, Wangler MF, Yamamoto S, Undiagnosed Diseases Network, Santana M, Perez V, Shukla P, Lee B, Yuan B, Xia F, Bellen HJ*, Liu P*, Liu Z* (2023) Enhancing Molecular Diagnostics of Mendelian Disorders with AI-MARRVEL: A Knowledge-Driven Artificial Intelligence Approach. Preprints with The Lancet :https://papers.ssrn.com/sol3/papers.cfm?abstract_id=4465963. *corresponding authors
Ralhan I, Chang J, Moulton MJ, Goodman LD, Lee NYJ, Plummer G, Pasolli HA, Matthies D, Bellen HJ , Ioannou MS (2023) Autolysosomal exocytosis of lipids protect neurons from ferroptosis. Journal of Cell Biology 222 :doi: 10.1083/jcb.202207130. [Abstract]
Andrews JC, Mok JW, Kanca O, Jangam S, Tifft C, Macnamara EF, Russell B, Wang LK, Undiagnosed Disease Network (UDN), Nelson SF, Bellen HJ, Yamamoto S, Malicdan M#, Wangler MF# (2023) De Novo Variants in MRTFB have gain of function activity in Drosophila and are associated with a novel neurodevelopmental phenotype with dysmorphic features. Genetics in Medicine 100833:doi: 10.1016/j.gim.2023.100833. #co-corresponding authors [Abstract]
Chung HL, Ye Q, Park YJ, Zuo Z, Kanca O, Mok J-W, Tattikota SG, Lu S, Perrimon N, Lee HK, Bellen HJ (2023) Very long-chain fatty acids induce glial-derived Sphingosine-1-Phosphate synthesis, secretion, and neuroinflammation. Cell Metabolism 35:855-874. [Abstract]
Ravenscroft TA, Jacobs A, Gu M, Eberl DF, Bellen HJ (2023) The Voltage-gated sodium channel in Drosophila, Para, localizes to dendrites as well as axons in mechanosensitive chordotonal neurons. eNeuro 10(6):ENEURO.0105-23.2023. [Abstract]
Tepe B, Macke EL, Niceta M, Hubsham MW, Kanca O, Schultz-Rogers L, Zarate YA, Schaefer B, Granadillo De Luque JL, Wegner DJ, Cogne B, Gilbert-Dussardier B, Guillou XL, Wagner EJ, Pais LS, Neil JE, Mochida GH, Walsh CA, Magal N, Drasinover V, Shohat M, Schwab T, Schmitz C, Clark K, Fine A, Lanpher B, Gavrilova R, Blanc P, Burglen L, Afenjar A, Steel D, Kurian MA, Prabhakar P. Gößwein S, Donato ND, Bertini ES, Undiagnosed Diseases Network(UDN), Wangler MF, Yamamoto S, Tartaglia M, Klee EW, Bellen HJ (2023) Biallelic variants in INTS11 are associated with a novel complex neurological disorder. American Journal of Human Genetics 110:774-789. [Abstract]
Pan X, Dutta D, Lu S, Bellen HJ (2023) Sphingolipids in neurodegenerative diseases. Frontiers in Neuroscience 17:1137893. DOI: 10.3389/fnins.2023.1137893. [Abstract]
Lin G, Tepe B, McGrane G, Tipon RC, Croft G, Panwala L, Hope A, Liang AJH, Zuo Z, Wang L, Bellen HJ (2023) Exploring therapeutic strategies for Infantile Neuronal Axonal Dystrophy (INAD/PARK14). eLife :doi:10.7554/eLife.82555. [Abstract]
Mele S, Martelli F, Lin J, Kanca O, Christodoulou J, Bellen HJ, Piper M, Johnson T (2022) Drosophila as a diet discovery tool for treating amino acid disorders. Trends in Endocrinology and Metabolism S1043-2760(22)00220-X:DOI: 10.1016/j.tem.2022.12.004 . [Abstract]
Ray SS, Dutta D, Dennys C, Powers S, Roussel F, Lisowski P, Glazar P, Zhang X, Biswas P, Caporale JR, Rajewsky N, Bickle M, Wein N, Bellen HJ, Likhite S, Marcogliese PC, Meyer KC (2022) Mechanisms of IRF2BPL-related disorders and identification of a potential therapeutic strategy. Cell Reports 41:111751.
Srivastava S, Shaked HM, Gable K, Gupta SD, Pan X, Somashekarappa N, Han G, Mohassel P, Gotkin3 M, Doney E, Goldenberg P, Tan QKG, Gong Y, Kleinstiver B, Wishart B, Cope H, Pires CB, Stutzman H, Spillmann RC, Undiagnosed Disease Network (UDN), Sadjadi R, Elpeleg O, Lee CH, Bellen HJ, Edvardon S, Eichler F, Dunn TM (2022) SPTSSA variants alter sphingolipid synthesis and cause a complex hereditary spastic paraplegia. Brain :awac460. doi: 10.1093/brain/awac460. [Abstract]
Bosch JA, Ugur B, Pichardo-Casas I, Rabasco J, Escobedo F, Zuo Z, Brown B, Celniker S, Sinclair DA, Bellen HJ, Perrimon N (2022) Two neuronal peptides encoded from a single transcript regulate mitochondrial complex III in Drosophila. eLife 11:DOI:10.7554/eLife.82709. [Abstract]
Karagas N, Gupta R, Rastegari E, Tan KL, Leung HH, Bellen HJ, Venkatachalam K, Wong CO (2022) Loss of activity-induced mitochondrial ATP production underlies the synaptic defects in a Drosophila model of ALS. Journal of Neuroscience 42(42):8019-8037. [Abstract]
Ma M*, Zhang X*, Zheng Y, Lu S, Pan X, Mao X, Pan H, Chung HL, Wang H, Hong Guo H#, Bellen HJ# (2022) The fly homolog of SUPT16H, a gene associated with neurodevelopmental disorders, is required in a cell-autonomous fashion for cell survival. Human Molecular Genetics :DOI:10.1093/hmg/ddac259. *equal contribution. #co-corresponding authors [Abstract]
Lu S, Ma M, Mao X, Bacino CA, Jankovic J, Sutton VR, Bartley JA, Wang X, Rosenfeld JA, Beleza-Meireles A, Chauhan J, Pan X, Li M, Liu P, Prescott K, Amin S, Davies G, Wangler MF, Dai Y#, Bellen HJ# (2022) De novo variants in FRMD5 are associated with developmental delay, intellectual disability, ataxia and abnormalities of eye movement. American Journal of Human Genetics 109(10):1932-1943. #co-corresponding authors [Abstract]
Huang Y*, Lemire G*, Briere LC*, Liu F, Wessels MW, Wang X, Osmond M, Kanca O, Lu S, High FA, Walker MA, Rodan LH, Undiagnosed Diseases Network (UDN), Care4Rare Canada Consortium, Wangler MF, Yamamoto S, Kernohan KD, Sweetser DA, Boycott KM, Bellen HJ (2022) The recurrent de novo c.2011C>T missense variant in MTSS2 causes syndromic intellectual disability. American Journal of Human Genetics :1923-1931. *contributed equally [Abstract]
Cheng KC, Burdine RD, Dickinson ME, Ekker SC, Lin AY, Kent Lloyd KC, Lutz CM, MacRae CA, Morrison JH, O'Connor D, Postlethwait JH, Rogers CD, Sanchez S, Simpson JH, Talbot WS, Wallace DC, Weimer JM, Bellen HJ (2022) Promoting validation and cross-phylogenetic integration in model organism research. Disease Models & Mechanisms 15(9):DOI: 10.1242/dmm.049600. [Abstract]
Barish S, Senturk M, Schoch K, Minogue AL, Lopergolo D, Fallerini C, Harland J, Seemann JH, Stong N, Kranz PG, Kansagra S, Mikati MA, Jasien J, El-Dairi M, Galluzzi P, Undiagnosed Diseases Network (UDN), Ariani F, Renieri A, Mari F, Wangler MF, Arur S, Jiang YH, Yamamoto S, Shashi V, Bellen HJ (2022) The microRNA processor DROSHA is a candidate gene for a severe progressive neurological disorder. Human Molecular Genetics 31(17):2934-2950. [Abstract]
Thomas AX, Link N, Robak LA, Demmler-Harrison G, Pao EC, Squire AE, Michels S, Cohen JS, Comi A, Prontera P, Verrotti di Pianella A, Cara GD, Garavelli L, Caraffi SG, Fusco C, Zuntini R, Parks KC, Sherr EH, Hashem MO, Maddirevula S, Alkuraya FS, Contractar IAF, Neil JE, Walsh CA, Bellen HJ, Chao HT, Clark RD, Mirzaa GM (2022) ANKLE2-related microcephaly: a variable disorder resembling Zika infection. Annals of Clinical and Translational Neurology 9(8):1276-1288. [Abstract]
Wang L, Lin G, Zuo Z, Li Y, Byeon SK, Pandey A, Bellen HJ (2022) Neuronal activity induces Glucosylceramide that is secreted via exosomes for lysosomal degradation in glia. Science Advances 8(28):eabn3326. [Abstract]
Deshpande SA, Rohrbach EW, Asuncion JD, Harrigan J, Eamani A, Schlingmann EH, Suto DJ, Lee PT, Schweizer FE, Bellen HJ, Krantz DE (2022) Regulation of Drosophila oviduct muscle contractility by octopamine. iScience 25(8):104697. [Abstract]
Kanca O, Zirin J, Hu Y, Tepe B, Dutta D, Lin WW, Ma L, Ge M, Zuo Z, Liu LP, Levis RW, Perrimon N, Bellen HJ (2022) An expanded toolkit for Drosophila gene tagging using synthesized homology donor constructs for CRISPR mediated homologous recombination. eLife 11:e76077. [Abstract]
Manivannan 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 HJ (2022) De novo FZR1 loss-of-function variants cause developmental and epileptic encephalopathy. Brain 145(5):1684-1697. [Abstract]
Wang Y, Lobb-Rabe M, Ashley J, Chatterjee P, Anand V, Bellen HJ, Kanca O, Carrillo R (2022) Systematic expression profiling of dprs and DIPs reveals cell surface codes in Drosophila larval motor and sensory neurons. Development 149(10):dev200355. [Abstract]
Ma M*, Moulton MJ*, Lu S, Bellen HJ (2022) 'Fly-ing' from rare to common neurodegenerative disease mechanisms. Trends in Genetics 38(9):972-984. *contributed equally [Abstract]
Huang 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 HJ (2022) Novel dominant and recessive variants in human ROBO1 cause distinct neurodevelopmental defects through different mechanisms. Human Molecular Genetics 31(16):2751-2765. [Abstract]
Lu 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 HJ# (2022) Loss of function variants in TIAM1 are associated with developmental delay, intellectual disability and seizures . American Journal of Human Genetics 109(4):571-586. #co-corresponding authors [Abstract]
Accogli A*, Lu S*, Musante I*, Scudieri P, Rosenfeld JA, Severino M, Baldassari S, Iacomino M, Riva A, Balagura A, Piccolo G, Minetti C, Roberto D, Xia F, Razak R, Lawrence E, Hussein M, Chang EY, Holick M, Cali E, Aliberto E, Gambardella A, Undiagnosed Diseases Network (UDN), SYNaPS Study Group, Emrick L, McCaffery PJA, Clagett-Dame M, Marcogliese PC, Bellen HJ, Lalani SR, Zara F, Striano P, Salpietro V (2022) Loss of Neuron Navigator 2 impairs brain and cerebellar development. The Cerebellum :doi: 10.1007/s12311-022-01379-3. Online ahead of print. *contributed equally [Abstract]
Chung 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 HJ#, Harel T# (2022) De novo variants in EMC1 lead to neurodevelopmental delay and cerebellar degeneration and affect glial function in Drosophila. Human Molecular Genetics :doi: 10.1093/hmg/ddac053. #co-corresponding authors [Abstract]
Cristobal CD, Wang CY, Zuo Z, Smith JA, Lindeke-Myers A, Bellen HJ, Lee HK (2022) Daam2 regulates myelin structure and the oligodendrocyte actin cytoskeleton through Rac1 and Gelsolin. Journal of Neuroscience 42(9):1679-1691. [Abstract]
Bellen HJ (2022) Lord of the fruit flies: an interview with Hugo Bellen. Disease Models & Mechanisms :doi: 10.1242/dmm.049500. [Abstract]
Harnish JM, Li LK, Rogic S, Piorier-Morency G, Kim SY, Undiagnosed Diseases Network (UDN), Boycott KM, Wangler MF, Bellen HJ, Hieter P, Pavlidis P, Liu Z, Yamamoto S (2022) ModelMatcher: A scientist-centric online platform to facilitate collaborations between stakeholders of rare and undiagnosed disease research. Human Mutation :doi: 10.1002/humu.24364. [Abstract]
Martelli F, Hernandes NH, Zuo Z, Wang J, Wong CO, Karagas NE, Roessner U, Rupasinhe T, Robin C, Venkatachalam K, Perry T, Batterham P, Bellen HJ (2022) Low doses of the organic insecticide spinosad trigger lysosomal defects, ROS driven lipid dysregulation and neurodegeneration in flies. eLife :doi: 10.7554/eLife.73812. [Abstract]
Marcogliese PC*#, Dutta D*, Sinha-Ray S, Dang NDP, Zuo Z, Wang Y, Lu D, Fazal F, Ravenscroft TA, Chung HL, Kanca O, Wan JJ, Douine ED, Undiagnosed Diseases Network (UDN), Pena LDM, Yamamoto S, Nelson SF, Might M, Meyer KC, Yeo NC, Bellen HJ# (2022) Loss of IRF2BPL impairs neuronal maintenance through excess Wnt signaling. Science Advances 8:doi: 10.1126/sciadv.abl5613. *co-first authors and #co-corresponding authors [Abstract]
Moulton MJ, Barish S, Ralhan I, Chang J, Goodman LD, Harland J, Marcogliese PC, Johansson JO, Ioannou M, Bellen HJ (2021) Neuronal ROS-Induced Glial Lipid Droplet Formation is Altered by Loss of Alzheimer's Disease-associated Genes. Proceedings of the National Academy of Sciences 118(52):e2112095118. [Abstract]
Ravenscroft TA, Phillips JB, Fieg E, Bajikar SS, Peirce J, Wegner J, Luna AA, Fox EJ, Yan YL, Rosenfeld JA, Zirin J, Kanca O, Undiagnosed Disease Network (UDN), Benke PJ, Cameron ES, Strehlow V, Platzer K, Jamra RA, Klockner C, Osmond M, Licata T, Rojas S, Dyment D, Chong JSC, Lincoln S, Stoler JM, Postlethwait J, Wangler MF, Yamamoto S, Krier J, Westerfield M, Bellen HJ (2021) Heterozygous loss-of-function variants significantly expand the phenotypes associated with loss of GDF11 . Genetics in Medicine 23(10):1889-1900. [Abstract]
Bellen HJ, Hubbard EJA, Lehmann R, Madhani HD, Solnica-Krezel L, Sothard-Smith EM (2021) Model organism databases are in jeopardy. Development 148(19):dev200193 Article | Twitter.
Park 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 SH (2021) Phosphatidylserine synthase plays an essential role in glia and affects development, as well as the maintenance of neuronal function. iScience 24(8):102899. [Abstract]
Goodman 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, Cogne 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 (UDN), Wangler MF, Yamamoto S, Bellen HJ#, Tan QKG# (2021) TNPO2 variants associate with human developmental delays, neurologic deficits, and dysmorphic features and alter TNPO2 activity in Drosophila. American Journal of Human Genetics 108(9):1669-1691. # co-corresponding authors [Abstract]
Baldridge D*, Wangler MF*, Bowman AN, Yamamoto S, Undiagnosed Diseases Network (UDN), Schedl T, Pak SC, Postlethwait JH, Shin J, Solnica-Krezel L, Bellen HJ, Westerfield M (2021) Model Organisms Contribute to Diagnosis and Discovery in the Undiagnosed Diseases Network: Current State and a Future Vision. Orphanet Journal of Rare Diseases 16(1):206. *co-first authors and co-corresponding authors [Abstract]
Luo X, Schoch K, Jangam SV, Bhavana VH, Graves HK, Kansagra S, Jasien JM, Stong N, keren B, Mignot C, Ravelli C, Undiagnosed Diseases Network (UDN), Bellen HJ, Wangler MF, Shashi V, Yamamoto S (2021) Rare deleterious de novo missense variants in Rnf2/Ring2 are associated with a neurodevelopmental disorder with unique clinical features. Human Molecular Genetics 30(14):1283-1292. [Abstract]
Cunningham K, Zhang K, Maulding K, Senturk M, Grima J, Sung H, Zuo Z, Song H, Rothstein J, Bellen HJ, Lloyd T (2020) TFEB/Mitf links impaired nuclear import to autophagolysosomal dysfunction in C9-ALS. eLife 9:e59419. [Abstract]
Barish S, Barakat TS, Michel BC, Mashtalir N, Phillips J, Valencia AM, Ugur B, Wegner J, Scott TM, Bostwick B, Members of Undiagnosed Diseases Network, Murdock DR, Dai H, Perenthaler E, Nikoncuk A, Slegtenhorst MV, 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, Cooley L, Gass J, Louie R, Stolerman E, Washington C, Laar IVD, Vetrini F, Otsubo A, Pratt VM, Conboy E, Treat K, Shannon N, Rosenfeld JA, Westerfield M, Wangler M, Kadoch C, Yamamoto S, Scott DA, Bellen HJ (2020) BICRA, a SWI/SNF complex member, is associated with BAF-disorder related phenotypes in humans and model organisms. American Journal of Human Genetics 107(6):1096-1112. [Abstract]
Martelli F, Zuo Z, Wang J, Wong CO, Karagas NE, Roessner U, Rupasinghe T, Venkatachalam K, Perry T, Bellen HJ, Batterham P (2020) Low doses of the neonicotinoid insecticide imidacloprid induce ROS triggering neurological and metabolic impairments in Drosophila. Proceedings of the National Academy of Sciences 117:25840-25850. [Abstract]
Link N, Bellen HJ (2020) Using Drosophila to drive the diagnosis and understand the mechanisms of rare human diseases. Development 147(21):1-7. [Abstract]
Ravenscroft TA, Janssens J, Lee PT, Tepe B, Marcogliese PC, Makhzami S, Holmes T, Aerts S, Bellen HJ (2020) Drosophila voltage-gated sodium channels are only expressed in active neurons and are localized to distal axonal initial segment-like domains. Journal of Neuroscience 40:7999-8024 Article. [Abstract]
Ding X, Jo J, Wang CY, Cristobal CD, Zuo Z, Ye Q, Wirianto M, Lindeke-Myers A, Choi JM, Mohila CA, Kawabe H, Jun SY, Bellen HJ, Yoo SH, Lee HK (2020) The Daam2-VHL-Nedd4 Axis 1 Governs Developmental and Regenerative Oligodendrocyte Differentiation. Genes and Development 34(17-18):1177-1189. [Abstract]
Kao CY, Xu M, Wang L, Lin SC, Lee HJ, Duraine L, Bellen HJ, Goldetein DS, Tsai SY, Tsai MJ (2020) Elevated COUP-TFII expression in dopaminergic neurons accelerates the progression of Parkinson's disease through mitochondrial dysfunction. PloS Genetics 16(6):e1008868.
Dutta 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, Undiagnosed Diseases Network, Wangler MF, Yamamoto S, Sweetser DA, Bellen HJ (2020) De novo mutations in TOMM70, a receptor of the mitochondrial import translocase, cause neurological impairment. Human Molecular Genetics 29(9):1568-1579. [Abstract]
Huang Y, Mao X, van Jaarsveld RH, Shu L, Terhal PA, Jia Z, Xi H, Peng Y, Yan H, Yuan S, Li O, Wang H, Bellen HJ (2020) Variants in CAPZA2, a member of a F-actin capping complex, cause intellectual disability and developmental delay. Human Molecular Genetics 29(9):1537-1546. [Abstract]
Chung 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 HJ (2020) Loss- or gain-of-function mutations in ACOX1 cause axonal loss via different mechanisms. Neuron 106:589-606. Recommended by F1000. Neuron 106:551-3, Preview. [Abstract]
Chung HL, Mao X, Wang H, Park YJ, Marcogliese PC, Rosenfels 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 B* (2020) De novo variants in CDK19 are associated with a new syndrome with intellectual disability and epileptic encephalopathy. American Journal of Human Genetics 106:717-725. *co-corresponding authors [Abstract]
Li 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 K (2020) Novel role of dynamin-related-protein 1 in dynamics of ER-lipid droplets in adipose tissue. The FASEB Journal 34(6):8265-8282. [Abstract]
Ye H, Ojelade SA, Li-Kroger D, Zuo Z, Wang L, Y Li, Gu JYJ, Tepass U, Rodal AA, Bellen HJ, Shulman JM (2020) Retromer subunit, VPS29, regulates synaptic transmission and is required for endolysosomal function in the aging brain. eLife 9:e91577.
Mao 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, Küry 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 HT* (2020) De novo EIF2AK1 and EIF2AK2 variants are associated with developmental delay, leukoencephalopathy, and neurologic decompensation. American Journal of Human Genetics 106:570-583. *co-corresponding authors [Abstract]
Bosch JA, Knight S, Kanca O, Zirin J, Yang-Zhou D, Hu Y, Rodiger J, Amador G, Bellen HJ, Perrimon N, Mohr SE (2020) Use of the CRISPR-Cas9 system in Drosophila cultured cells to introduce fluorescent tags into endogenous genes. Current Protocols in Molecular Biology 130:e112. [Abstract]
Link 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 HJ (2019) Mutations in ANKLE2, a ZIKA virus target, disrupt an asymmetric cell division pathway in Drosophila neuroblasts to cause microcephaly. Developmental Cell 51:713-729. [Abstract]
Wang 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 PM* (2019) Loss of Oxidation Resistance 1, OXR1, is associated with an autosomal-recessive neurological disease with cerebellar atrophy and lysosomal dysfunction. American Journal of Human Genetics 105:1237-1253. *co-corresponding authors [Abstract]
Bellen HJ, Wangler MF, Yamamoto S (2019) The fruit fly at the interface of diagnosis and pathogenic mechanisms of rare and common human diseases. Human Molecular Genetics 28:R207-R214. [Abstract]
Ansar 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 SE* (2019) Bi-allelic variants in IQSEC1 cause intellectual disability, developmental delay and short stature. American Journal of Human Genetics 105:907-920. *co-corresponding authors [Abstract]
Kanca O, Zirin J, Garcia-Marques J, Knight S, Yang-Zhou D, Amador G, Chung HL, 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 HJ (2019) An efficient CRISPR-based strategy to insert small and large fragments of DNA using short homology arms. eLife 8:e51539. [Abstract]
Kanca O, Bellen HJ (2019) La CaSSA da Drosophila: a versatile expansion of the tool box. Neuron 23:177-179. [Abstract]
Guo 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, Büttner 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 EE (2019) Disruptive mutations in TANC2 define a neurodevelopmental syndrome associated with psychiatric disorders. Nature Communications 10:4679. [Abstract]
Wang J, Mao D, Fazal F, Kim SY, Yamamoto S, Bellen H, Liu Z (2019) Using MARRVEL v1.2 for bioinformatics analysis of human genes and variant pathogenicity. Current Protocols in Bioinformatics 67:e85. [Abstract]
Wang J, Liu Z, Bellen HJ, Yamamoto S (2019) Navigating MARRVEL, a web-based tool that integrates human genomics and model organism genetics information. Journal of Visualized Experiments: JoVE 150:59542. video [Abstract]
Ojelade SA, Lee TV, Giagtzoglou N, Yu L, Ugur B, Li Y, Duraine L, Zuo Z, Petyuk V, De Jager PL, Bennett DS, Arenkiel BR, Bellen HJ, Shulman JM (2019) cindr, the Drosophila homolog of the CD2AP Alzheimer's disease susceptibility gene, is required for synaptic transmission and proteostasis. Cell Reports 28:1799-1813. [Abstract]
Kanca 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 MCV* (2019) De novo variants in WDR37 are associated with epilepsy, colobomas, dysmorphism, developmental delay, intellectual disability, and cerebellar hypoplasia. American Journal of Human Genetics 105:413-424. *co-corresponding authors [Abstract]
Şentürk M, Mao D, Bellen HJ (2019) Loss of proteins associated with amyotrophic lateral sclerosis affects lysosomal acidification via different routes. Autophagy 28:1-3. [Abstract]
Şentürk M, Lin G, Mao D, Zuo Z, Watson E, Mikos AG, Bellen HJ (2019) Ubiquilins regulate autophagic flux through mTOR signaling and lysosomal acidification. Nature Cell Biology 21:384-396. [Abstract]
Mao D, Lin G, Tepe B, Zuo Z, Tan KL, Senturk M, Zhang S, Arenkiel BR, Sardiello M, Bellen HJ (2019) VAMP associated proteins are required for autophagic and lysosomal degradation by promoting a PtdIns4P-mediated endosomal pathway. Autophagy 11:1-20. [Abstract]
Lin G, Wang L, Marcogliese PC, Bellen HJ (2019) Sphingolipids in the pathogenesis of Parkinson's disease and parkinsonism. Trends in Endocrinology & Metabolism 30:106-117. [Abstract]
Cosmanescu F, Katsamba PS, Sergeeva AP, Ahlsen G, Patel SD, Brewer JJ, Tan L, Xu S, Xiao Q, Nagarkar-Jaiswal S, Nern A, Bellen HJ, Zipursky SL, Honig B, Shapiro L (2018) Neuron-subtype-specific expression, interaction affinities, and specificity determinants of DIP/Dpr cell recognition proteins. Neuron 100:1385-1400. Neuron 100:1270-2, Preview. [Abstract]
Xu S, Xiao Q, Cosmanescu F, Sergeeva AP, Yoo J, Lin Y, Katsamba PS, Ahlsen G, Kaufman J, Linaval NT, Lee PT, Bellen HJ, Shapiro L, Honig B, Tan L, Zipursky SL (2018) Interactions between the Ig-superfamily proteins DIP-alpha and Dpr6/10 regulate assembly of neural circuits. Neuron 100:1369-1384. [Abstract]
Shah PS, Link N, Jang GM, Sharp PP, Zhu T, Swaney DL, Johnson JR, Von Dollen J, Ramage HR, Satkamp L, Newton B, Hüttenhain R, Petit MJ, Baum T, Everitt A, Laufman O, Tassetto M, Shales M, Stevenson E, Iglesias GN, Shokat L, Tripathi S, Balasubramaniam V, Webb LG, Aguirre S, Willsey AJ, Garcia-Sastre A, Pollard KS, Cherry S, Gamarnik AV, Marazzi I, Taunton J, Fernandez-Sesma A, Bellen HJ*, Andino R*, Krogan NJ* (2018) Comparative flavivirus-host protein interaction mapping reveals mechanisms of dengue and Zika virus pathogenesis. Cell 175:1931-1945. Recommended by F1000. Cell 175:1728-9, Preview. *co-corresponding authors [Abstract]
Splinter K, Adams DR, Bacino CA, Bellen HJ, Bernstein JA, Cheatle-Jarvela AM, Eng CM, Esteves C, Gahl WA, Hamid R, Jacob HJ, Kikani B, Koeller DM, Kohane IS, Lee BH, Loscalzo J, Luo X, McCray AT, Metz TO, Mulvihill JJ, Nelson SF, Palmer CGS, Phillips JA 3rd, Pick L, Postlethwait JH, Reuter C, Shashi V, Sweetser DA, Tifft CJ, Walley NM, Wangler MF, Westerfield M, Wheeler MT, Wise AL, Worthey EA, Yamamoto S, Ashley EA; Undiagnosed Diseases Network (2018) Effect of genetic diagnosis on patients with previously undiagnosed disease. New England Journal of Medicine 379:2131-2139. BMJ 363:k4272, Research News. [Abstract]
Ansar M, Chung HL, Taylor RL, Nazir A, Imtiaz S, Sarwar MT, Manousopoulou A, Makrythanasis P, Saeed S, Falconnet E, Guipponi M, Pournaras CJ, Ansari MA, Ranza E, Santoni FA, Ahmed J, Shah I, Gul K, Black GC, Bellen HJ*, Antonarakis SE* (2018) Bi-allelic loss-of-function variants in DNMBP cause infantile cataracts. American Journal of Human Genetics 103:568-578. *co-corresponding authors [Abstract]
Lin G, Lee PT, Chen K, Mao D, Tan KL, Zuo Z, Lin WW, Wang L, Bellen HJ (2018) Phospholipase PLA2G6, a Parkinsonism-associated gene, affects Vps26 and Vps35, retromer function, and ceramide levels, similar to alpha-Synuclein gain. Cell Metabolism 28:605-618. [Abstract]
Li-Kroeger D, Kanca O, Lee PT, Cowan S, Lee MT, Jaiswal M, Salazar JL, He Y, Zuo Z, Bellen HJ (2018) An expanded toolkit for gene tagging based on MiMIC and scarless CRISPR tagging in Drosophila. eLife 7:e38709. [Abstract]
Marcogliese PC, Shashi V, Spillmann RC, Stong N, Rosenfeld JA, Koenig MK, Martínez-Agosto JA, Herzog M, Chen AH, Dickson PI, Lin HJ, Vera MU, Salamon N, Graham JM Jr, Ortiz D, Infante E, Steyaert W, Dermaut B, Poppe B, Chung HL, Zuo Z, Lee PT, Kanca O, Xia F, Yang Y, Smith EC, Jasien J, Kansagra S, Spiridigliozzi G, El-Dairi M, Lark R, Riley K, Koeberl DD, Golden-Grant K; Program for Undiagnosed Diseases (UD-PrOZA); Undiagnosed Diseases Network, Yamamoto S, Wangler MF, Mirzaa G, Hemelsoet D, Lee B, Nelson SF, Goldstein DB, Bellen HJ*, Pena LDM* (2018) IRF2BPL is associated with neurological phenotypes. American Journal of Human Genetics 103:245-260. *co-corresponding authors [Abstract]
Willsey AJ, Morris MT, Wang S, Willsey HR, Sun N, Teerikorpi N, Baum TB, Cagney G, Bender KJ, Desai TA, Srivastava D, Davis GW, Doudna J, Chang E, Sohal V, Lowenstein DH, Li H, Agard D, Keiser MJ, Shoichet B, von Zastrow M, Mucke L, Finkbeiner S, Gan L, Sestan N, Ward ME, Huttenhain R, Nowakowski TJ, Bellen HJ, Frank LM, Khokha MK, Lifton RP, Kampmann M, Ideker T, State MW, Krogan NJ (2018) The Psychiatric Cell Map Initiative: A convergent systems biological approach to illuminating key molecular pathways in neuropsychiatric disorders. Cell 174:505-520. [Abstract]
Liu N, Schoch K, Luo X, Pena LDM, Bhavana VH, Kukolich MK, Stringer S, Powis Z, Radtke K, Mroske C, Deak KL, McDonald MT, McConkie-Rosell A, Markert ML, Kranz PG, Stong N, Need AC, Bick D, Amaral MD, Worthey EA, Levy S; Undiagnosed Diseases Network (UDN), Wangler MF, Bellen HJ, Shashi V, Yamamoto S (2018) Functional variants in TBX2 are associated with a syndromic cardiovascular and skeletal developmental disorder. Human Molecular Genetics 27:2454-2465. [Abstract]
Şentürk M, Bellen HJ (2018) Genetic strategies to tackle neurological diseases in fruit flies. Current Opinion in Neurobiology 50:24-32. [Abstract]
Li T, Bellen HJ, Groves AK (2018) Using Drosophila to study mechanisms of hereditary hearing loss. Disease Models & Mechanisms 11:dmm031492. [Abstract]
Ansar M, Chung H, Waryah YM, Makrythanasis P, Falconnet E, Rao AR, Guipponi M, Narsani AK, Fingerhut R, Santoni FA, Ranza E, Waryah AM, Bellen HJ*, Antonarakis SE* (2018) Visual impairment and progressive phthisis bulbi caused by recessive pathogenic variant in MARK3. Human Molecular Genetics 27:2703-2711. *co-corresponding authors [Abstract]
Tan KL, Haelterman NA, Kwartler CS, Regalado ES, Lee PT, Nagarkar-Jaiswal S, Guo DC, Duraine L, Wangler MF; University of Washington Center for Mendelian Genomics, Bamshad MJ, Nickerson DA, Lin G, Milewicz DM, Bellen HJ (2018) Ari-1 regulates myonuclear organization together with Parkin and is associated with aortic aneurysms. Developmental Cell 45:226-244. Recommended by F1000. Developmental Cell 45:149-50, Preview. [Abstract]
Wangler MF, Assia Batzir N, Robak LA, Koenig MK, Bacino CA, Scaglia F, Bellen HJ (2018) The expanding neurological phenotype of DNM1L-related disorders. Brain 141:e28. [Abstract]
Lee PT, Zirin J, Kanca O, Lin WW, Schulze KL, Li-Kroeger D, Tao R, Devereaux C, Hu Y, Chung V, Fang Y, He Y, Pan H, Ge M, Zuo Z, Housden BE, Mohr SE, Yamamoto S, Levis RW, Spradling AC, Perrimon N, Bellen HJ (2018) A gene-specific T2A-GAL4 library for Drosophila. eLife 7:e35574. [Abstract]
Oláhová M, Yoon WH, Thompson K, Jangam S, Fernandez L, Davidson JM, Kyle JE, Grove ME, Fisk DG, Kohler JN, Holmes M, Dries AM, Huang Y, Zhao C, Contrepois K, Zappala Z, Frésard L, Waggott D, Zink EM, Kim YM, Heyman HM, Stratton KG, Webb-Robertson BM; Undiagnosed Diseases Network, Snyder M, Merker JD, Montgomery SB, Fisher PG, Feichtinger RG, Mayr JA, Hall J, Barbosa IA, Simpson MA, Deshpande C, Waters KM, Koeller DM, Metz TO, Morris AA, Schelley S, Cowan T, Friederich MW, McFarland R, Van Hove JLK, Enns GM, Yamamoto S, Ashley EA, Wangler MF, Taylor RW, Bellen HJ, Bernstein JA, Wheeler MT (2018) Biallelic mutations in ATP5F1D, which encodes a subunit of ATP synthase, cause a metabolic disorder. American Journal of Human Genetics 102:494-504. [Abstract]
Lee PT, Lin G, Lin WW, Diao F, White BH, Bellen HJ (2018) A kinase-dependent feedforward loop affects novel CREBB stability and long-term memory formation. eLife 7:e33007. [Abstract]
Landrock KK, Sullivan P, Martini-Stoica H, Goldstein DS, Graham BH, Yamamoto S, Bellen HJ, Gibbs RA, Chen R, D'Amelio M, Stoica G (2018) Pleiotropic neuropathological and biochemical alterations associated with Myo5a mutation in a rat model. Brain Research 1679:155-170. [Abstract]
Ugur B, Bao H, Stawarski M, Duraine LR, Zuo Z, Lin YQ, Neely GG, Macleod GT, Chapman ER, Bellen HJ (2017) The Krebs cycle enzyme Isocitrate Dehydrogenase 3A couples mitochondrial metabolism to synaptic transmission. Cell Reports 21:3794-3806. [Abstract]
Kanca O, Bellen HJ, Schnorrer F (2017) Gene tagging strategies to assess protein expression, localization, and function in Drosophila. Genetics 207:389-412. [Abstract]
Chao HT, Liu L, Bellen HJ (2017) Building dialogues between clinical and biomedical research through cross-species collaborations. Seminars in Cell & Developmental Biology 70:49-57. [Abstract]
Liu L, MacKenzie KR, Putluri N, Maletić-Savatić M, Bellen HJ (2017) The glia-neuron lactate shuttle and elevated ROS promote lipid synthesis in neurons and lipid droplet accumulation in glia via APOE/D. Cell Metabolism 26:719-737. Cell Metabolism 26:701-2, Preview. Science Translational Medicine 9(412):eaap8170, Editor's Choice. [Abstract]
Wangler MF, Yamamoto S, Chao HT, Posey JE, Westerfield M, Postlethwait J, Members of the Undiagnosed Diseases Network (UDN), Hieter P, Boycott KM, Campeau PM, Bellen HJ (2017) Model organisms facilitate rare disease diagnosis and therapeutic research. Genetics 207:9-27. [Abstract]
Luo X, Rosenfeld JA, Yamamoto S, Harel T, Zuo Z, Hall M, Wierenga KJ, Pastore MT, Bartholomew D, Delgado MR, Rotenberg J, Lewis RA, Emrick L, Bacino CA, Eldomery MK, Coban Akdemir Z, Xia F, Yang Y, Lalani SR, Lotze T, Lupski JR, Lee B, Bellen HJ, Wangler MF; Members of the UDN (2017) Clinically severe CACNA1A alleles affect synaptic function and neurodegeneration differentially. PLoS Genetics 13:e1006905. [Abstract]
Wangler MF, Chao YH, Bayat V, Giagtzoglou N, Shinde AB, Putluri N, Coarfa C, Donti T, Graham BH, Faust JE, McNew JA, Moser A, Sardiello M, Baes M, Bellen HJ (2017) Peroxisomal biogenesis is genetically and biochemically linked to carbohydrate metabolism in Drosophila and mouse. PLoS Genetics 13:e1006825. [Abstract]
Wong CO, Gregory S, Hu H, Chao Y, Sepúlveda VE, He Y, Li-Kroeger D, Goldman WE, Bellen HJ, Venkatachalam K (2017) Lysosomal degradation is required for sustained phagocytosis of bacteria by macrophages. Cell Host Microbe 21:719-730. Cell Host Microbe 21:657-9, Preview. [Abstract]
Wang J, Al-Ouran R, Hu Y, Kim SY, Wan YW, Wangler MF, Yamamoto S, Chao HT, Comjean A, Mohr SE, UDN, Perrimon N, Liu Z*, Bellen HJ* (2017) MARRVEL: integration of human and model organism genetic resources to facilitate functional annotation of the human genome. American Journal of Human Genetics 100:843-853. Selected among Best of AJHG 2016-2017. *co-corresponding author [Abstract]
Nagarkar-Jaiswal S, Manivannan SN, Zuo Z, Bellen HJ (2017) A cell cycle-independent, conditional gene inactivation strategy for differentially tagging wild-type and mutant cells. eLife 31:e26420. [Abstract]
Malmanche N, Dourlen P, Gistelinck M, Demiautte F, Link N, Dupont C, Vanden Broeck L, Werkmeister E, Amouyel P, Bongiovanni A, Bauderlique H, Moechars D, Royou A, Bellen HJ, Lafont F, Callaerts P, Lambert JC, Dermaut B (2017) Developmental expression of 4-Repeat-Tau induces neuronal aneuploidy in Drosophila tauopathy models. Scientific Reports 7:40764. [Abstract]
Chao HT, Davids M, Burke E, Pappas JG, Rosenfeld JA, McCarty AJ, Davis T, Wolfe L, Toro C, Tifft C, Xia F, Stong N, Johnson TK, Warr CG, Undiagnosed Diseases Network, Yamamoto S, Adams DR, Markello TC, Gahl WA, Bellen HJ, Wangler MF, Malicdan MC (2017) A syndromic neurodevelopmental disorder caused by de novo variants in EBF3. American Journal of Human Genetics 100:128-137. [Abstract]
Yoon WH, Sandoval H, Nagarkar-Jaiswal S, Jaiswal M, Yamamoto S, HaeltermanNA, Putluri N, Putluri V, Sreekumar A, Tos T, Aksoy A, Donti T, Graham BH, OhnoM, Nishi E, Hunter J, Muzny DM, Carmichael J, Shen J, Arboleda VA, Nelson SF,Wangler MF, Karaca E, Lupski JR, Bellen HJ (2017) Loss of Nardilysin, a mitochondrial co-chaperone for α-ketoglutarate dehydrogenase, promotes mTORC1 activation and neurodegeneration. Neuron 93:115-131. [Abstract]
Lin G, Mao D, Bellen HJ (2017) Amyotrophic Lateral Sclerosis pathogenesis converges on defects in protein homeostasis associated with TDP-43 mislocalization and proteasome-mediated degradation overload. Current Topics in Developmental Biology 121:111-171. [Abstract]
Chen K, Ho TS, Lin G, Tan KL, Rasband MN, Bellen HJ (2016) Loss of Frataxin activates the iron/sphingolipid/PDK1/Mef2 pathway in mammals. eLife 5:e20732. [Abstract]
Jakobsdottir J, van der Lee SJ, Bis JC, Chouraki V, Li-Kroeger D, Yamamoto S, Grove ML, Naj A, Vronskaya M, Salazar JL, DeStefano AL, Brody JA, Smith AV, Amin N, Sims R, Ibrahim-Verbaas CA, Choi SH, Satizabal CL, Lopez OL, Beiser A, Ikram MA, Garcia ME, Hayward C, Varga TV, Ripatti S, Franks PW, Hallmans G, Rolandsson O, Jansson JH, Porteous DJ, Salomaa V, Eiriksdottir G, Rice KM, Bellen HJ, Levy D, Uitterlinden AG, Emilsson V, Rotter JI, Aspelund T, Cohorts for Heart and Aging Research in Genomic Epidemiology consortium, Alzheimer's Disease Genetic Consortium, Genetic and Environmental Risk in Alzheimer's Disease consortium, O'Donnell CJ, Fitzpatrick AL, Launer LJ, Hofman A, Wang LS, Williams J, Schellenberg GD, Boerwinkle E, Psaty BM, Seshadri S, Shulman JM, Gudnason V, van Duijn CM (2016) Rare functional variant in TM2D3 is associated with late-onset Alzheimer's disease. PLoS Genetics 12:e1006327. [Abstract]
Harel 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 JR (2016) Recurrent de novo and biallelic variation of ATAD3A, encoding a mitochondrial membrane protein, results in distinct neurological syndromes. American Journal of Human Genetics 99:831-845. [Abstract]
Chen K, Lin G, Haelterman NA, Ho TS, Li T, Li Z, Duraine L, Graham BH, Jaiswal M, Yamamoto S, Rasband MN, Bellen HJ (2016) Loss of Frataxin induces iron toxicity, sphingolipid synthesis, and Pdk1/Mef2 activation, leading to neurodegeneration. eLife 5:e16043. [Abstract]
Chouhan AK, Guo C, Hsieh YC, Ye H, Senturk M, Zuo Z, Li Y, Chatterjee S, Botas J, Jackson GR, Bellen HJ, Shulman JM (2016) Uncoupling neuronal death and dysfunction in Drosophila models of neurodegenerative disease. Acta Neuropathologica Communications 4:62. [Abstract]
Li T, Giagtzoglou N, Eberl DF, Jaiswal SN, Cai T, Godt D, Groves AK, Bellen HJ (2016) The E3 ligase Ubr3 regulates Usher syndrome and MYH9 disorder proteins in the auditory organs of Drosophila and mammals. eLife 5:e15258. [Abstract]
Ali 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 HC (2016) NMNAT2:HSP90 complex mediates proteostasis in proteinopathies. PLoS Biology 14:e1002472. PLoS Biology 14:e1002522, Primer. [Abstract]
Li T, Fan J, Blanco-Sánchez B, Giagtzoglou N, Lin G, Yamamoto S, Jaiswal M, Chen K, Zhang J, Wei W, Lewis MT, Groves AK, Westerfield M, Jia J, Bellen HJ (2016) Ubr3, a novel modulator of Hh signaling affects the degradation of Costal-2 and Kif7 through poly-ubiquitination. PLoS Genetics 12:e1006054. [Abstract]
Chao YH, Robak LA, Xia F, Koenig MK, Adesina A, Bacino CA, Scaglia F, Bellen HJ, Wangler MF (2016) Missense variants in the middle domain of DNM1L in cases of infantile encephalopathy alter peroxisomes and mitochondria when assayed in Drosophila. Human Molecular Genetics 25:1846-1856. [Abstract]
Ugur B, Chen K, Bellen HJ (2016) Drosophila tools and assays for the study of human diseases. Disease Models & Mechanisms 9:235-244. [Abstract]
David-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 HJ (2016) WAC regulates mTOR activity by acting as an adaptor for the TTT and Pontin/Reptin complexes. Developmental Cell 36:139-151. Developmental Cell 36:129-30, Preview. [Abstract]
Carrillo RA, Özkan E, Menon KP, Nagarkar-Jaiswal S, Lee PT, Jeon M, Birnbaum ME, Bellen HJ, Garcia KC, Zinn K (2015) Control of synaptic connectivity by a network of Drosophila IgSF cell surface proteins. Cell 163:1770-1782. Recommended by F1000. [Abstract]
Tan 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 SL (2015) Ig superfamily ligand and receptor pairs expressed in synaptic partners in Drosophila. Cell 163:1756-1769. Recommended by F1000. [Abstract]
Bellen HJ, Yamamoto S (2015) Morgan's legacy: fruit flies and the functional annotation of conserved genes. Cell 163:12-14. [Abstract]
Zanet J, Benrabah E, Li T, Pélissier-Monier A, Chanut-Delalande H, Ronsin B, Bellen HJ, Payre F, Plaza S (2015) Pri sORF peptides induce selectiveproteasome-mediated protein processing. Science 349:1356-1358. Recommended by F1000. Science Signaling 8(395):ec274, Editor's Choice. [Abstract]
Wu 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 SY (2015) Increased COUP-TFII expression in adult hearts induces mitochondrial dysfunction resulting in heart failure. Nature Communications 6:8245. [Abstract]
Akbari 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 J (2015) Safeguarding gene drive experiments in the laboratory. Science 349:927-929. [Abstract]
Wang F, Jiang L, Chen Y, Haelterman NA, Bellen HJ, Chen R (2015) FlyVar: a database for genetic variation in Drosophila melanogaster. Database 2015:bav079. [Abstract]
Wang S, Bellen HJ (2015) The retromer complex in development and disease. Development 142:2392-2396. [Abstract]
Jaiswal M, Haelterman NA, Sandoval H, Xiong B, Donti T, Kalsotra A, Yamamoto S, Cooper TA, Graham BH, Bellen HJ (2015) Impaired mitochondrial energy production causes light-induced photoreceptor degeneration independent of oxidative stress. PLoS Biology 13:e1002197. [Abstract]
Nagarkar-Jaiswal S, DeLuca SZ, Lee PT, Lin WW, Pan H, Zuo Z, Lv J, Spradling AC, Bellen HJ (2015) A genetic toolkit for tagging intronic MiMIC containing genes. eLife 4:e08469. [Abstract]
Nagarkar-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 HJ (2015) A library of MiMICs allows tagging of genes and reversible, spatial and temporal knockdown of proteins in Drosophila. eLife 4:e05338. [Abstract]
Tian 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 C* (2015) A voltage-gated calcium channel regulates lysosomal fusion with endosomes and autophagosomes and is required for neuronal homeostasis. PLoS Biology 13:e1002103. Recommended by F1000. *co-corresponding author [Abstract]
Rui YN, Xu Z, Patel B, Chen Z, Chen D, Tito A, David G, Sun Y, Stimming EF, Bellen HJ, Cuervo AM, Zhang S (2015) Huntingtin functions as a scaffold for selective macroautophagy. Nature Cell Biology 17:262-275. Nature Cell Biology 17:214-5, News & Views. [Abstract]
Wangler MF, Yamamoto S, Bellen HJ (2015) Fruit flies in biomedical research. Genetics 199:639-653. [Abstract]
Liu L, Zhang K, Sandoval H, Yamamoto S, Jaiswal M, Sanz E, Li Z, Hui J, Graham BH, Quintana A, Bellen HJ (2015) Glial lipid droplets and ROS induced by mitochondrial defects promote neurodegeneration. Cell 160:177-190. [Abstract]
Wong 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 K (2014) A TRPV channel in Drosophila motor neurons regulates presynaptic resting Ca2+ levels, synapse growth, and synaptic transmission. Neuron 84:764-777. Neuron 84:659-61, Preview. [Abstract]
Sandoval 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 HJ (2014) Mitochondrial fusion but not fission regulates larval growth and synaptic development through steroid hormone production. eLife 3:e03558. [Abstract]
Bellen HJ (2014) Survival of the fittest tools. Genetics 198:427-428. [Abstract]
Haelterman NA, Jiang L, Li Y, Bayat V, Sandoval H, Ugur B, Tan KL, Zhang K, Bei D, Xiong B, Charng WL, Busby T, Jawaid A, David G, Jaiswal M, Venken KJ, Yamamoto S, Chen R, Bellen HJ (2014) Large-scale identification of chemically induced mutations in Drosophila melanogaster. Genome Research 24:1707-1718. [Abstract]
Yamamoto S, Jaiswal M, Charng WL, Gambin T, Karaca E, Mirzaa G, Wiszniewski W, Sandoval H, Haelterman NA, Xiong B, Zhang K, Bayat V, David G, Li T, Chen K, Gala U, Harel T, Pehlivan D, Penney S, Vissers LELM, de Ligt J, Jhangiani SN, Xie Y, Tsang SH, Parman Y, Sivaci M, Battaloglu E, Muzny D, Wan YW, Liu Z, Lin-Moore AT, Clark RD, Curry CJ, Link N, Schulze KL, Boerwinkle E, Dobyns WB, Allikmets R, Gibbs RA, Chen R, Lupski JR, Wangler MF, Bellen HJ (2014) A Drosophila genetic resource of mutants to study mechanisms underlying human genetic diseases. Cell 159:200-214. Recommended by F1000. Nature Methods 11:1197, Comment. [Abstract]
Halstead JM, Lin YQ, Duraine L, Hamilton RS, Ball G, Neely GG, Bellen HJ, Davis I (2014) Syncrip/hnRNP Q influences synaptic transmission and regulates BMP signaling at the Drosophila neuromuscular synapse. Biology Open 3:839-849. [Abstract]
Venken KJ, Bellen HJ (2014) Chemical mutagens, transposons, and transgenes to interrogate gene function in Drosophila melanogaster. Methods 68:15-28. [Abstract]
Haelterman NA, Yoon WH, Sandoval H, Jaiswal M, Shulman JM, Bellen HJ (2014) A mitocentric view of Parkinson's disease. Annual Review of Neuroscience 37:137-159. [Abstract]
Wang 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 HJ (2014) The retromer complex is required for rhodopsin recycling and its loss leads to photoreceptor degeneration. PLoS Biology 12:e1001847. [Abstract]
Charng WL, Yamamoto S, Bellen HJ (2014) Shared mechanisms between Drosophila peripheral nervous system development and human neurodegenerative diseases. Current Opinion in Neurobiology 27:158-164. [Abstract]
Moustaqim-Barrette A, Lin YQ, Pradhan S, Neely GG, Bellen HJ, Tsuda H (2014) The amyotrophic lateral sclerosis 8 protein, VAP, is required for ER protein quality control. Human Molecular Genetics 23:1975-1989. [Abstract]
Charng WL, Yamamoto S, Jaiswal M, Bayat V, Xiong B, Zhang K, Sandoval H, David G, Gibbs S, Lu HC, Chen K, Giagtzoglou N, Bellen HJ (2014) Drosophila Tempura, a novel protein prenyltransferase α subunit, regulates Notch signaling via Rab1 and Rab11. PLoS Biology 12:e1001777. PLoS Biology 12:e1001778, Synopsis. [Abstract]
Yamamoto S, Bellen HJ (2014) Preface: Notch signaling. Methods in Molecular Biology 1187:v. [Abstract]
Yamamoto S, Schulze KL, Bellen HJ (2014) Introduction to Notch signaling. Methods in Molecular Biology 1187:1-14. [Abstract]
Cassidy JJ, Jha AR, Posadas DM, Giri R, Venken KJ, Ji J, Jiang H, Bellen HJ, White KP, Carthew RW (2013) miR-9a minimizes the phenotypic impact of genomic diversity by buffering a transcription factor. Cell 155:1556-1567. [Abstract]
Xiong B, Bellen HJ (2013) Rhodopsin homeostasis and retinal degeneration: lessons from the fly. Trends in Neurosciences 36:652-660. [Abstract]
Ali YO, Li-Kroeger D, Bellen HJ, Zhai RG, Lu HC (2013) NMNATs, evolutionarily conserved neuronal maintenance factors. Trends in Neurosciences 36:632-640. [Abstract]
Han SM, El Oussini H, Scekic-Zahirovic J, Vibbert J, Cottee P, Prasain JK, Bellen HJ, Dupuis L, Miller MA (2013) VAPB/ALS8 MSP ligands regulate striated muscle energy metabolism critical for adult survival in Caenorhabditis elegans. PLoS Genetics 9:e1003738. [Abstract]
Giagtzoglou N, Li T, Yamamoto S, Bellen HJ (2013) Drosophila EHBP1 regulates Scabrous secretion during Notch-mediated lateral inhibition. Journal of Cell Science 126:3686-3696. [Abstract]
Yamamoto S, Bayat V, Bellen HJ, Tan C (2013) Protein phosphatase 1Β limits ring canal constriction during Drosophila germline cyst formation. PLoS One 8:e70502. [Abstract]
Zhang K, Li Z, Jaiswal M, Bayat V, Xiong B, Sandoval H, Charng WL, David G, Haueter C, Yamamoto S, Graham BH, Bellen HJ (2013) The C8ORF38 homologue Sicily is a cytosolic chaperone for a mitochondrial complex I subunit. Journal of Cell Biology 200:807-820. [Abstract]
Winther ÅM, Jiao W, Vorontsova O, Rees KA, Koh TW, Sopova E, Schulze KL, Bellen HJ, Shupliakov O (2013) The dynamin-binding domains of Dap160/intersectin affect bulk membrane retrieval in synapses. Journal of Cell Science 126:1021-1031. [Abstract]
Xiong B, Bayat V, Jaiswal M, Zhang K, Sandoval H, Charng WL, Li T, David G, Duraine L, Lin YQ, Neely GG, Yamamoto S, Bellen HJ (2012) Crag is a GEF for Rab11 required for rhodopsin trafficking and maintenance of adult photoreceptor cells. PLoS Biology 10:e1001438. [Abstract]
Yamamoto S, Charng WL, Rana NA, Kakuda S, Jaiswal M, Bayat V, Xiong B, Zhang K, Sandoval H, David G, Wang H, Haltiwanger RS, Bellen HJ (2012) A mutation in EGF repeat-8 of Notch discriminates between Serrate/Jagged and Delta family ligands. Science 338:1229-1232. Recommended by F1000. Science Signaling 6(276), pe19, Perspective. [Abstract]
Chen YC, Lin YQ, Banerjee S, Venken K, Li J, Ismat A, Chen K, Duraine L, Bellen HJ, Bhat MA (2012) Drosophila neuroligin 2 is required presynaptically and postsynaptically for proper synaptic differentiation and synaptic transmission. Journal of Neuroscience 32:16018-16030. [Abstract]
Szabad J, Bellen HJ, Venken KJ (2012) An assay to detect in vivo Y chromosome loss in Drosophila wing disc cells. G3 2:1095-1102. [Abstract]
Alves-Silva J, Sánchez-Soriano N, Beaven R, Klein M, Parkin J, Millard TH, Bellen HJ, Venken KJ, Ballestrem C, Kammerer RA, Prokop A (2012) Spectraplakins promote microtubule-mediated axonal growth by functioning as structural microtubule-associated proteins and EB1-dependent +TIPs (tip interacting proteins). Journal of Neuroscience 32:9143-9158. [Abstract]
Bayat V, Thiffault I, Jaiswal M, Tétreault M, Donti T, Sasarman F, Bernard G, Demers-Lamarche J, Dicaire M, Mathieu V, Rioux M, Lesage J, Lourenco CM, Li Z, Haueter C, Shoubridge E, Graham BH, Brais B, Bellen HJ (2012) Mutations in the mitochondrial methionyl-tRNA synthetase cause a neurodegenerative phenotype in flies and a recessive ataxia (ARSAL) in humans. PLoS Biology 10:e1001288. [Abstract]
Venken KJ, Bellen HJ (2012) Genome-wide manipulations of Drosophila melanogaster with transposons, Flp recombinase, and ΦC31 integrase. Methods in Molecular Biology 859:203-228. [Abstract]
Neumüller RA, Wirtz-Peitz F, Lee S, Kwon Y, Buckner M, Hoskins RA, Venken KJ, Bellen HJ, Mohr SE, Perrimon N (2012) Stringent analysis of gene function and protein-protein interactions using fluorescently tagged genes. Genetics 190:931-940. [Abstract]
Han SM, Tsuda H, Yang Y, Vibbert J, Cottee P, Lee SJ, Winek J, Haueter C, Bellen HJ, Miller MA (2012) Secreted VAPB/ALS8 major sperm protein domains modulate mitochondrial localization and morphology via growth cone guidance receptors. Developmental Cell 22:348-362. Recommended by F1000. Developmental Cell 22:238-9. Preview. [Abstract]
Giagtzoglou N, Yamamoto S, Zitserman D, Graves HK, Schulze KL, Wang H, Klein H, Roegiers F, Bellen HJ (2012) dEHBP1 controls exocytosis and recycling of Delta during asymmetric divisions. Journal of Cell Biology 196:65-83. [Abstract]
Jaiswal M, Sandoval H, Zhang K, Bayat V, Bellen HJ (2012) Probing mechanisms that underlie human neurodegenerative disease in Drosophila. Annual Review of Genetics 46:371-396. [Abstract]
Xue M, Giagtzoglou N, Bellen HJ (2011) Dueling Ca2+ sensors in neurotransmitter release. Cell 147:491-493. [Abstract]
Venken KJ, Simpson JH, Bellen HJ (2011) Genetic manipulation of genes and cells in the nervous system of the fruit fly. Neuron 72:202-230. [Abstract]
Spradling AC, Bellen HJ, Hoskins RA (2011) Drosophila P elements preferentially transpose to replication origins. Proceedings of the National Academy of Sciences USA 108:15948-15953. [Abstract]
Mitne-Neto M, Machado-Costa M, Marchetto MC, Bengtson MH, Joazeiro CA, Tsuda H, Bellen HJ, Silva HC, Oliveira AS, Lazar M, Muotri AR, Zatz M (2011) Downregulation of VAPB expression in motor neurons derived from induced pluripotent stem cells of ALS8 patients. Human Molecular Genetics 20:3642-3652. [Abstract]
Venken KJ, Schulze KL, Haelterman NA, Pan H, He Y, Evans-Holm M, Carlson JW, Levis RW, Spradling AC, Hoskins RA, Bellen HJ (2011) MiMIC: a highly versatile transposon insertion resource for engineering Drosophila melanogaster genes. Nature Methods 8:737-743. Nature Methods 8:728-9, News & Views. [Abstract]
Tong C, Ohyama T, Tien AC, Rajan A, Haueter CM, Bellen HJ (2011) rich regulates target specificity of photoreceptor cells and N-cadherin trafficking in the Drosophila visual system via Rab6. Neuron 71:447-459. [Abstract]
Zheng L, Michelson Y, Freger V, Avraham Z, Venken KJ, Bellen HJ, Justice MJ, Wides R (2011) Drosophila Ten-m and filamin affect motor neuron growth cone guidance. PLoS One 6:e22956. [Abstract]
Bellen HJ, Levis RW, He Y, Carlson JW, Evans-Holm M, Bae E, Kim J, Metaxakis A, Savakis C, Schulze KL, Hoskins RA, Spradling AC (2011) The Drosophila gene disruption project: progress using transposons with distinctive site specificities. Genetics 188:731-743. [Abstract]
Nègre N, Brown CD, Ma L, Bristow CA, Miller SW, Wagner U, Kheradpour P, Eaton ML, Loriaux P, Sealfon R, Li Z, Ishii H, Spokony RF, Chen J, Hwang L, Cheng C, Auburn RP, Davis MB, Domanus M, Shah PK, Morrison CA, Zieba J, Suchy S, Senderowicz L, Victorsen A, Bild NA, Grundstad AJ, Hanley D, MacAlpine DM, Mannervik M, Venken K, Bellen H, White R, Gerstein M, Russell S, Grossman RL, Ren B, Posakony JW, Kellis M, White KP (2011) A cis-regulatory map of the Drosophila genome. Nature 471:527-531. [Abstract]
Bayat V, Jaiswal M, Bellen HJ (2011) The BMP signaling pathway at the Drosophila neuromuscular junction and its links to neurodegenerative diseases. Current Opinion in Neurobiology 21:182-188. [Abstract]
modENCODE Consortium, Roy S, Ernst J, Kharchenko PV, Kheradpour P, et al. (2010) Identification of functional elements and regulatory circuits by Drosophila modENCODE. Science 330:1787-1797. [Abstract]
Venken KJ, Popodi E, Holtzman SL, Schulze KL, Park S, Carlson JW, Hoskins RA, Bellen HJ, Kaufman TC (2010) A molecularly defined duplication set for the X chromosome of Drosophila melanogaster. Genetics 186:1111-1125. [Abstract]
Neely GG, Hess A, Costigan M, Keene AC, Goulas S, Langeslag M, Griffin RS, Belfer I, Dai F, Smith SB, Diatchenko L, Gupta V, Xia CP, Amann S, Kreitz S, Heindl-Erdmann C, Wolz S, Ly CV, Arora S, Sarangi R, Dan D, Novatchkova M, et al. (2010) A genome-wide Drosophila screen for heat nociception identifies α2δ3 as an evolutionarily conserved pain gene. Cell 143:628-638. [Abstract]
Yamamoto S, Charng WL, Bellen HJ (2010) Endocytosis and intracellular trafficking of Notch and its ligands. Current Topics in Developmental Biology 92:165-200. [Abstract]
Bellen HJ, Tong C, Tsuda H (2010) 100 years of Drosophila research and its impact on vertebrate neuroscience: a history lesson for the future. Nature Reviews Neuroscience 11:514-522. [Abstract]
Giagtzoglou N, Mahoney T, Yao CK, Bellen HJ (2009) Rab3 GTPase lands Bruchpilot. Neuron 64:595-597. [Abstract]
Xue M, Lin YQ, Pan H, Reim K, Deng H, Bellen HJ, Rosenmund C (2009) Tilting the balance between facilitatory and inhibitory functions of mammalian and Drosophila Complexins orchestrates synaptic vesicle exocytosis. Neuron 64:367-380. Recommended by F1000. Neuron 64:295-7, Preview. [Abstract]
Giagtzoglou N, Ly CV, Bellen HJ (2009) Cell adhesion, the backbone of the synapse: vertebrate and invertebrate perspectives. Cold Spring Harbor Perspectives in Biology 1:a003079. [Abstract]
Yao CK, Lin YQ, Ly CV, Ohyama T, Haueter CM, Moiseenkova-Bell VY, Wensel TG, Bellen HJ (2009) A synaptic vesicle-associated Ca2+ channel promotes endocytosis and couples exocytosis to endocytosis. Cell 138:947-960. [Abstract]
Fotowat H, Fayyazuddin A, Bellen HJ, Gabbiani F (2009) A novel neuronal pathway for visually guided escape in Drosophila melanogaster. Journal of Neurophysiology 102:875-885. [Abstract]
Verstreken P, Ohyama T, Haueter C, Habets RL, Lin YQ, Swan LE, Ly CV, Venken KJ, De Camilli P, Bellen HJ (2009) Tweek, an evolutionarily conserved protein, is required for synaptic vesicle recycling. Neuron 63:203-215. Recommended by F1000. Nature Chemical Biology 5:614-5, News & Views. [Abstract]
Rajan A, Tien AC, Haueter CM, Schulze KL, Bellen HJ (2009) The Arp2/3 complex and WASp are required for apical trafficking of Delta into microvilli during cell fate specification of sensory organ precursors. Nature Cell Biology 11:815-824. Recommended by F1000. Nature Cell Biology 11:791-3, News & Views. [Abstract]
Patel PH, Costa-Mattioli M, Schulze KL, Bellen HJ (2009) The Drosophila deoxyhypusine hydroxylase homologue nero and its target eIF5A are required for cell growth and the regulation of autophagy. Journal of Cell Biology 185:1181-1194. [Abstract]
Venken KJ, Carlson JW, Schulze KL, Pan H, He Y, Spokony R, Wan KH, Koriabine M, de Jong PJ, White KP, Bellen HJ, Hoskins RA (2009) Versatile P[acman] BAC libraries for transgenesis studies in Drosophila melanogaster. Nature Methods 6:431-434. Recommended by F1000. [Abstract]
Andrews HK, Giagtzoglou N, Yamamoto S, Schulze KL, Bellen HJ (2009) Sequoia regulates cell fate decisions in the external sensory organs of adult Drosophila. EMBO Reports 10:636-641. [Abstract]
Giagtzoglou N, Lin YQ, Haueter C, Bellen HJ (2009) Importin 13 regulates neurotransmitter release at the Drosophila neuromuscular junction. Journal of Neuroscience 29:5628-5639. [Abstract]
Tien AC, Rajan A, Bellen HJ (2009) A Notch updated. Journal of Cell Biology 184:621-629. [Abstract]
Giagtzoglou N, Xue M, Bellen HJ (2009) An old dog learns a new trick: Synaptotagmin, a Ca2+ sensor for spontaneous release at central synapses. Cell Science Reviews 6(2):.
Tien AC, Rajan A, Schulze KL, Ryoo HD, Acar M, Steller H, Bellen HJ (2008) Ero1L, a thiol oxidase, is required for Notch signaling through cysteine bridge formation of the Lin12-Notch repeats in Drosophila melanogaster. Journal of Cell Biology 182:1113-1125. [Abstract]
Tsuda H, Han SM, Yang Y, Tong C, Lin YQ, Mohan K, Haueter C, Zoghbi A, Harati Y, Kwan J, Miller MA, Bellen HJ (2008) The amyotrophic lateral sclerosis 8 protein VAPB is cleaved, secreted, and acts as a ligand for Eph receptors. Cell 133:963-977. Recommended by F1000. Cell 133:949, Preview. [Abstract]
Zhai RG, Zhang F, Hiesinger PR, Cao Y, Haueter CM, Bellen HJ (2008) NAD synthase NMNAT acts as a chaperone to protect against neurodegeneration. Nature 452:887-891. Recommended by F1000. Nature Reviews Neuroscience 9:323, Featured Highlight. [Abstract]
Ly CV, Yao CK, Verstreken P, Ohyama T, Bellen HJ (2008) straightjacket is required for the synaptic stabilization of cacophony, a voltage-gated calcium channel alpha1 subunit. Journal of Cell Biology 181:157-170. [Abstract]
Verstreken P, Ohyama T, Bellen HJ. (2008) FM 1-43 labeling of synaptic vesicle pools at the Drosophila neuromuscular junction. Methods in Molecular Biology 440:349-369. [Abstract]
Acar M, Jafar-Nejad H, Takeuchi H, Rajan A, Ibrani D, Rana NA, Pan H, Haltiwanger RS, Bellen HJ (2008) Rumi is a CAP10 domain glycosyltransferase that modifies Notch and is required for Notch signaling. Cell 132:247-258. Recommended by F1000. Cell 132:177-9, Preview. Nature Reviews Molecular Cell Biology 9:186-7, Research Highlight. [Abstract]
Romero E, Cha GH, Verstreken P, Ly CV, Hughes RE, Bellen HJ, Botas J (2008) Suppression of neurodegeneration and increased neurotransmission caused by expanded full-length huntingtin accumulating in the cytoplasm. Neuron 57:27-40. [Abstract]
Zhang W, Wang D, Volk E, Bellen HJ, Hiesinger PR, Quiocho FA (2008) V-ATPase V0 sector subunit a1 in neurons is a target of calmodulin. Journal of Biological Chemistry 283:294-300. [Abstract]
Ohyama T, Verstreken P, Ly CV, Rosenmund T, Rajan A, Tien AC, Haueter C, Schulze KL, Bellen HJ (2007) Huntingtin-interacting protein 14, a palmitoyl transferase required for exocytosis and targeting of CSP to synaptic vesicles. Journal of Cell Biology 179:1481-1496. [Abstract]
Venken KJ, Bellen HJ (2007) Transgenesis upgrades for Drosophila melanogaster. Development 134:3571-3584. [Abstract]
Koh TW, Korolchuk VI, Wairkar YP, Jiao W, Evergren E, Pan H, Zhou Y, Venken KJ, Shupliakov O, Robinson IM, O'Kane CJ, Bellen HJ (2007) Eps15 and Dap160 control synaptic vesicle membrane retrieval and synapse development. Journal of Cell Biology 178:309-322. [Abstract]
Zhang J, Schulze KL, Hiesinger PR, Suyama K, Wang S, Fish M, Acar M, Hoskins RA, Bellen HJ, Scott MP (2007) Thirty-one flavors of Drosophila rab proteins. Genetics 176:1307-1322. [Abstract]
Venken KJ, He Y, Hoskins RA, Bellen HJ (2006) P[acman]: a BAC transgenic platform for targeted insertion of large DNA fragments in D. melanogaster. Science 314:1747-1751. Recommended by F1000. Nature Methods 4:114, Research Highlight. [Abstract]
Zhai RG, Cao Y, Hiesinger PR, Zhou Y, Mehta SQ, Schulze KL, Verstreken P, Bellen HJ (2006) Drosophila NMNAT maintains neural integrity independent of its NAD synthesis activity. PLoS Biology 4:e416. [Abstract]
Hiesinger PR, Zhai RG, Zhou Y, Koh TW, Mehta SQ, Schulze KL, Cao Y, Verstreken P, Clandinin TR, Fischbach KF, Meinertzhagen IA, Bellen HJ (2006) Activity-independent prespecification of synaptic partners in the visual map of Drosophila. Current Biology 16:1835-1843. [Abstract]
Acar M, Jafar-Nejad H, Giagtzoglou N, Yallampalli S, David G, He Y, Delidakis C, Bellen HJ (2006) Senseless physically interacts with proneural proteins and functions as a transcriptional co-activator. Development 133:1979-1989. [Abstract]
Jafar-Nejad H, Tien AC, Acar M, Bellen HJ (2006) Senseless and Daughterless confer neuronal identity to epithelial cells in the Drosophila wing margin. Development 133:1683-1692. [Abstract]
Seto ES, Bellen HJ (2006) Internalization is required for proper Wingless signaling in Drosophila melanogaster. Journal of Cell Biology 173:95-106. [Abstract]
Fayyazuddin A, Zaheer MA, Hiesinger PR, Bellen HJ (2006) The nicotinic acetylcholine receptor Dalpha7 is required for an escape behavior in Drosophila. PLoS Biology 4:e63. [Abstract]
Giagtzoglou N, Bellen HJ (2006) Fighting anthrax with flies. Proceedings of the National Academy of Sciences USA 103:3013-3014. [Abstract]
Shroyer NF, Wallis D, Venken KJ, Bellen HJ, Zoghbi HY (2005) Gfi1 functions downstream of Math1 to control intestinal secretory cell subtype allocation and differentiation. Genes and Development 19:2412-2417. [Abstract]
Wu S, Mehta SQ, Pichaud F, Bellen HJ, Quiocho FA (2005) Sec15 interacts with Rab11 via a novel domain and affects Rab11 localization in vivo. Nature Structural and Molecular Biology 12:879-885. [Abstract]
Jafar-Nejad H, Andrews HK, Acar M, Bayat V, Wirtz-Peitz F, Mehta SQ, Knoblich JA, Bellen HJ (2005) Sec15, a component of the exocyst, promotes notch signaling during the asymmetric division of Drosophila sensory organ precursors. Developmental Cell 9:351-363. Developmental Cell 9:312-3, Review. [Abstract]
Tsuda H, Jafar-Nejad H, Patel AJ, Sun Y, Chen HK, Rose MF, Venken KJ, Botas J, Orr HT, Bellen HJ, Zoghbi HY (2005) The AXH domain of Ataxin-1 mediates neurodegeneration through its interaction with Gfi-1/Senseless proteins. Cell 122:633-644. Nature Neuroscience 8:1422-4, News & Views. [Abstract]
Verstreken P, Ly CV, Venken KJ, Koh TW, Zhou Y, Bellen HJ (2005) Synaptic mitochondria are critical for mobilization of reserve pool vesicles at Drosophila neuromuscular junctions. Neuron 47:365-378. Recommended by F1000. Neuron 47:331-3, Minireview. Journal of Cell Biology 170:699.3, Research Roundup. [Abstract]
Dermaut B, Norga KK, Kania A, Verstreken P, Pan H, Zhou Y, Callaerts P, Bellen HJ (2005) Aberrant lysosomal carbohydrate storage accompanies endocytic defects and neurodegeneration in Drosophila benchwarmer. Journal of Cell Biology 170:127-139. [Abstract]
Hiesinger PR, Fayyazuddin A, Mehta SQ, Rosenmund T, Schulze KL, Zhai RG, Verstreken P, Cao Y, Zhou Y, Kunz J, Bellen HJ (2005) The v-ATPase V0 subunit a1 is required for a late step in synaptic vesicle exocytosis in Drosophila. Cell 121:607-620. Cell 121:496-7, Preview. [Abstract]
Mehta SQ, Hiesinger PR, Beronja S, Zhai RG, Schulze KL, Verstreken P, Cao Y, Zhou Y, Tepass U, Crair MC, Bellen HJ (2005) Mutations in Drosophila sec15 reveal a function in neuronal targeting for a subset of exocyst components. Neuron 46:219-232. Neuron 46:164-6, Preview. [Abstract]
Venken KJ, Bellen HJ (2005) Emerging technologies for gene manipulation in Drosophila melanogaster. Nature Reviews Genetics 6:167-178. [Abstract]
Escudero LM, Caminero E, Schulze KL, Bellen HJ, Modolell J (2005) Charlatan, a Zn-finger transcription factor, establishes a novel level of regulation of the proneural achaete/scute genes of Drosophila. Development 132:1211-1222. [Abstract]
Beckstead RB, Ner SS, Hales KG, Grigliatti TA, Baker BS, Bellen HJ (2005) Bonus, a Drosophila TIF1 homolog, is a chromatin-associated protein that acts as a modifier of position-effect variegation. Genetics 169:783-794. [Abstract]
Kazanjian A, Wallis D, Au N, Nigam R, Venken KJ, Cagle PT, Dickey BF, Bellen HJ, Gilks CB, Grimes HL (2004) Growth factor independence-1 is expressed in primary human neuroendocrine lung carcinomas and mediates the differentiation of murine pulmonary neuroendocrine cells. Cancer Research 64:6874-6882. [Abstract]
Zhai RG, Bellen HJ (2004) Hauling t-SNAREs on the microtubule highway. Nature Cell Biology 6:918-919. [Abstract]
Jafar-Nejad H, Bellen HJ (2004) Gfi/Pag-3/senseless zinc finger proteins: a unifying theme?. Molecular and Cellular Biology 24:8803-8812. [Abstract]
Zhai RG, Bellen HJ (2004) The architecture of the active zone in the presynaptic nerve terminal. Physiology 19:262-270. [Abstract]
Koh TW, Verstreken P, Bellen HJ (2004) Dap160/intersectin acts as a stabilizing scaffold required for synaptic development and vesicle endocytosis. Neuron 43:193-205. Recommended by F1000. Current Biology 14:R853-5, Dispatch. [Abstract]
Bellen HJ, Levis RW, Liao G, He Y, Carlson JW, Tsang G, Evans-Holm M, Hiesinger PR, Schulze KL, Rubin GM, Hoskins RA, Spradling AC (2004) The BDGP gene disruption project: single transposon insertions associated with 40% of Drosophila genes. Genetics 167:761-781. [Abstract]
Pesah Y, Pham T, Burgess H, Middlebrooks B, Verstreken P, Zhou Y, Harding M, Bellen H, Mardon G (2004) Drosophila parkin mutants have decreased mass and cell size and increased sensitivity to oxygen radical stress. Development 131:2183-2194. [Abstract]
Bellen HJ (2004) Hugo J. Bellen. Current Biology 14:R218. [Abstract]
Hiesinger PR, Bellen HJ (2004) Flying in the face of total disruption. Nature Genetics 36:211-212. [Abstract]
Seto ES, Bellen HJ (2004) The ins and outs of Wingless signaling. Trends in Cell Biology 14:45-53. [Abstract]
Jafar-Nejad H, Acar M, Nolo R, Lacin H, Pan H, Parkhurst SM, Bellen HJ (2003) Senseless acts as a binary switch during sensory organ precursor selection. Genes and Development 17:2966-2978. [Abstract]
Fabian-Fine R, Verstreken P, Hiesinger PR, Horne JA, Kostyleva R, Zhou Y, Bellen HJ, Meinertzhagen IA (2003) Endophilin promotes a late step in endocytosis at glial invaginations in Drosophila photoreceptor terminals. Journal of Neuroscience 23:10732-10744. [Abstract]
Verstreken P, Koh TW, Schulze KL, Zhai RG, Hiesinger PR, Zhou Y, Mehta SQ, Cao Y, Roos J, Bellen HJ (2003) Synaptojanin is recruited by endophilin to promote synaptic vesicle uncoating. Neuron 40:733-748. Recommended by F1000. Neuron 40:665-7, Preview. Nature Reviews Neuroscience 5:4-5, News & Views. [Abstract]
Zhai RG, Hiesinger PR, Koh TW, Verstreken P, Schulze KL, Cao Y, Jafar-Nejad H, Norga KK, Pan H, Bayat V, Greenbaum MP, Bellen HJ (2003) Mapping Drosophila mutations with molecularly defined P element insertions. Proceedings of the National Academy of Sciences USA 100:10860-10865. Recommended by F1000. Nature Reviews Genetics 4:849, Highlight. [Abstract]
Norga KK, Gurganus MC, Dilda CL, Yamamoto A, Lyman RF, Patel PH, Rubin GM, Hoskins RA, Mackay TF, Bellen HJ (2003) Quantitative analysis of bristle number in Drosophila mutants identifies genes involved in neural development. Current Biology 13:1388-1396. [Abstract]
Koh TW, Bellen HJ (2003) Synaptotagmin I, a Ca2+ sensor for neurotransmitter release. Trends in Neurosciences 26:413-422. [Abstract]
Wallis D, Hamblen M, Zhou Y, Venken KJ, Schumacher A, Grimes HL, Zoghbi HY, Orkin SH, Bellen HJ (2003) The zinc finger transcription factor Gfi1, implicated in lymphomagenesis, is required for inner ear hair cell differentiation and survival. Development 130:221-232. [Abstract]
Kango-Singh M, Nolo R, Tao C, Verstreken P, Hiesinger PR, Bellen HJ, Halder G (2002) Shar-pei mediates cell proliferation arrest during imaginal disc growth in Drosophila. Development 129:5719-5730. [Abstract]
Kjaerulff O, Verstreken P, Bellen HJ (2002) Synaptic vesicle retrieval: still time for a kiss. Nature Cell Biology 4:E245-E248. [Abstract]
Wang VY, Hassan BA, Bellen HJ, Zoghbi HY (2002) Drosophila atonal fully rescues the phenotype of Math1 null mice: new functions evolve in new cellular contexts. Current Biology 12:1611-1616. [Abstract]
Jafar-Nejad H, Norga K, Bellen HJ (2002) Numb: "Adapting" Notch for endocytosis. Developmental Cell 3:155-156. [Abstract]
Verstreken P, Bellen HJ (2002) Meaningless minis? Mechanisms of neurotransmitter-receptor clustering. Trends in Neurosciences 25:383-385. [Abstract]
Pennetta G, Hiesinger PR, Fabian-Fine R, Meinertzhagen IA, Bellen HJ (2002) Drosophila VAP-33A directs bouton formation at neuromuscular junctions in a dosage-dependent manner. Neuron 35:291-306. [Abstract]
Seto ES, Bellen HJ, Lloyd TE (2002) When cell biology meets development: endocytic regulation of signaling pathways. Genes and Development 16:1314-1336. [Abstract]
Verstreken P, Kjaerulff O, Lloyd TE, Atkinson R, Zhou Y, Meinertzhagen IA, Bellen HJ (2002) Endophilin mutations block clathrin-mediated endocytosis but not neurotransmitter release. Cell 109:101-112. Recommended by F1000. Nature Cell Biology 4:E123, News & Views. [Abstract]
Izaddoost S, Nam SC, Bhat MA, Bellen HJ, Choi KW (2002) Drosophila Crumbs is a positional cue in photoreceptor adherens junctions and rhabdomeres. Nature 416:178-183. [Abstract]
Chen S, Zheng X, Schulze KL, Morris T, Bellen H, Stanley EF (2002) Enhancement of presynaptic calcium current by cysteine string protein. Journal of Physiology 538:383-389. [Abstract]
Lloyd TE, Atkinson R, Wu MN, Zhou Y, Pennetta G, Bellen HJ (2002) Hrs regulates endosome membrane invagination and tyrosine kinase receptor signaling in Drosophila. Cell 108:261-269. Recommended by F1000. Nature Medicine 8:218, News & Views. [Abstract]
Peter A, Schöttler P, Werner M, Beinert N, Dowe G, Burkert P, Mourkioti F, Dentzer L, He Y, Deák 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, Miñana B, Kafatos FC, Bolshakov S, Sidén-Kiamos I, Papagiannakis G, Spanos L, Louis C, Madueño 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, Jäckle H, Schäfer U (2002) Mapping and identification of essential gene functions on the X chromosome of Drosophila. EMBO Reports 3:34-38. [Abstract]
Fergestad T, Wu MN, Schulze KL, Lloyd TE, Bellen HJ, Broadie K (2001) Targeted mutations in the syntaxin H3 domain specifically disrupt SNARE complex function in synaptic transmission. Journal of Neuroscience 21:9142-9150. [Abstract]
Frankfort BJ, Nolo R, Zhang Z, Bellen HJ, Mardon G (2001) senseless repression of rough is required for R8 photoreceptor differentiation in the developing Drosophila eye. Neuron 32:403-414. [Abstract]
Lloyd TE, Bellen HJ (2001) pRIMing synaptic vesicles for fusion. Nature Neuroscience 4:965-966. [Abstract]
Verstreken P, Bellen HJ (2001) The meaning of a mini. Science 293:443-444. [Abstract]
Bermingham NA, Hassan BA, Wang VY, Fernandez M, Banfi S, Bellen HJ, Fritzsch B, Zoghbi HY (2001) Proprioceptor pathway development is dependent on Math1. Neuron 30:411-422. [Abstract]
Bhat MA, Rios JC, Lu Y, Garcia-Fresco GP, Ching W, St Martin M, Li J, Einheber S, Chesler M, Rosenbluth J, Salzer JL, Bellen HJ (2001) Axon-glia interactions and the domain organization of myelinated axons requires neurexin IV/Caspr/Paranodin. Neuron 30:369-383. Current Biology 11:R555-7, Dispatch. [Abstract]
Beckstead R, Ortiz JA, Sanchez C, Prokopenko SN, Chambon P, Losson R, Bellen HJ (2001) Bonus, a Drosophila homolog of TIF1 proteins, interacts with nuclear receptors and can inhibit betaFTZ-F1-dependent transcription. Molecular Cell 7:753-765. [Abstract]
Steffensen S, Coelho PA, Cobbe N, Vass S, Costa M, Hassan B, Prokopenko SN, Bellen H, Heck MM, Sunkel CE (2001) A role for Drosophila SMC4 in the resolution of sister chromatids in mitosis. Current Biology 11:295-307. [Abstract]
Wu MN, Schulze KL, Lloyd TE, Bellen HJ (2001) The ROP-syntaxin interaction inhibits neurotransmitter release. European Journal of Cell Biology 80:196-199. [Abstract]
Nolo R, Abbott LA, Bellen HJ (2001) Drosophila Lyra mutations are gain-of-function mutations of senseless. Genetics 157:307-315. [Abstract]
Prokopenko SN, He Y, Lu Y, Bellen HJ (2000) Mutations affecting the development of the peripheral nervous system in Drosophila: a molecular screen for novel proteins. Genetics 156:1691-1715. [Abstract]
Nolo R, Abbott LA, Bellen HJ (2000) Senseless, a Zn finger transcription factor, is necessary and sufficient for sensory organ development in Drosophila. Cell 102:349-362. Recommended by F1000. [Abstract]
Hassan BA, Bellen HJ (2000) Doing the MATH: is the mouse a good model for fly development?. Genes and Development 14:1852-1865. [Abstract]
Lloyd TE, Verstreken P, Ostrin EJ, Phillippi A, Lichtarge O, Bellen HJ (2000) A genome-wide search for synaptic vesicle cycle proteins in Drosophila. Neuron 26:45-50. [Abstract]
Prokopenko SN, Saint R, Bellen HJ (2000) Untying the Gordian knot of cytokinesis: Role of small G proteins and their regulators. Journal of Cell Biology 148:843-848. [Abstract]
Hassan BA, Bermingham NA, He Y, Sun Y, Jan YN, Zoghbi HY, Bellen HJ (2000) atonal regulates neurite arborization but does not act as a proneural gene in the Drosophila brain. Neuron 25:549-561. [Abstract]
Ben-Arie N, Hassan BA, Bermingham NA, Malicki DM, Armstrong D, Matzuk M, Bellen HJ, Zoghbi HY (2000) Functional conservation of atonal and Math1 in the CNS and PNS. Development 127:1039-1048. [Abstract]
Mao Y, Nickitenko A, Duan X, Lloyd TE, Wu MN, Bellen H, Quiocho FA (2000) Crystal structure of the VHS and FYVE tandem domains of Hrs, a protein involved in membrane trafficking and signal transduction. Cell 100:447-456. [Abstract]
Prokopenko SN, Saint R, Bellen HJ (2000) Tissue distribution of PEBBLE RNA and pebble protein during Drosophila embryonic development. Mechanisms of Development 90:269-273. [Abstract]
Bellen HJ, Budnik V (2000) The neuromuscular junction. Drosophila protocols Cold Spring Harbor Laboratory Press, New York: pp. 175-200.
Bellen HJ (2000) The fruitfly neuromuscular junction flexes its muscle. Cell 101:141-143.
Pennetta G, Wu MN, Bellen HJ (1999) Dissecting the molecular mechanisms of neurotransmitter release in Drosophila. Neurotransmitter release. Frontiers in Molecular Biology. Oxford University Press, New York: pp. 304-351.
Lloyd T, Bellen HJ (1999) Genetic analysis of neurotransmitter release in mice and humans. Neurotransmitter release. Frontiers in Molecular Biology. Oxford University Press, New York: pp. 352-388.
Bellen HJ (1999) Ten years of enhancer detection: lessons from the fly. Plant Cell 11:2271-2281. [Abstract]
Prokopenko SN, Brumby A, O'Keefe L, Prior L, He Y, Saint R, Bellen HJ (1999) A putative exchange factor for Rho1 GTPase is required for initiation of cytokinesis in Drosophila. Genes and Development 13:2301-2314. [Abstract]
Wu MN, Fergestad T, Lloyd TE, He Y, Broadie K, Bellen HJ (1999) Syntaxin 1A interacts with multiple exocytic proteins to regulate neurotransmitter release in vivo. Neuron 23:593-605. [Abstract]
Zhang B, Ganetzky B, Bellen HJ, Murthy VN (1999) Tailoring uniform coats for synaptic vesicles during endocytosis. Neuron 23:419-422. [Abstract]
Bermingham NA, Hassan BA, Price SD, Vollrath MA, Ben-Arie N, Eatock RA, Bellen HJ, Lysakowski A, Zoghbi HY (1999) Math1: an essential gene for the generation of inner ear hair cells. Science 284:1837-1841. [Abstract]
Bhat MA, Izaddoost S, Lu Y, Cho KO, Choi KW, Bellen HJ (1999) Discs Lost, a novel multi-PDZ domain protein, establishes and maintains epithelial polarity. Cell 96:833-845. [Abstract]
Zhang B, Koh YH, Beckstead RB, Budnik V, Ganetzky B, Bellen HJ (1998) Synaptic vesicle size and number are regulated by a clathrin adaptor protein required for endocytosis. Neuron 21:1465-1475. [Abstract]
Hassan BA, Prokopenko SN, Breuer S, Zhang B, Paululat A, Bellen HJ (1998) skittles, a Drosophila phosphatidylinositol 4-phosphate 5-kinase, is required for cell viability, germline development and bristle morphology, but not for neurotransmitter release. Genetics 150:1527-1537. [Abstract]
Bellen HJ, Lu Y, Beckstead R, Bhat MA (1998) Neurexin IV, caspr and paranodin--novel members of the neurexin family: encounters of axons and glia. Trends in Neurosciences 21:444-449. [Abstract]
Bellen HJ (1998) The fruit fly: a model organism to study the genetics of alcohol abuse and addiction?. Cell 93:909-912. [Abstract]
Adams RR, Tavares AA, Salzberg A, Bellen HJ, Glover DM (1998) pavarotti encodes a kinesin-like protein required to organize the central spindle and contractile ring for cytokinesis. Genes and Development 12:1483-1494. [Abstract]
Dye CA, Lee JK, Atkinson RC, Brewster R, Han PL, Bellen HJ (1998) The Drosophila sanpodo gene controls sibling cell fate and encodes a tropomodulin homolog, an actin/tropomyosin-associated protein. Development 125:1845-1856. [Abstract]
Wu MN, Littleton JT, Bhat MA, Prokop A, Bellen HJ (1998) ROP, the Drosophila Sec1 homolog, interacts with syntaxin and regulates neurotransmitter release in a dosage-dependent manner. EMBO Journal 17:127-139. [Abstract]
Cabello OA, Baldini A, Bhat M, Bellen H, Belmont JW (1997) Localization of BRRN1, the human homologue of Drosophila barr, to 2q11.2. Genomics 46:311-313. [Abstract]
Salzberg A, Prokopenko SN, He Y, Tsai P, Pál M, Maróy P, Glover DM, Deák P, Bellen HJ (1997) P-element insertion alleles of essential genes on the third chromosome of Drosophila melanogaster: mutations affecting embryonic PNS development. Genetics 147:1723-1741. [Abstract]
Ben-Arie N, Bellen HJ, Armstrong DL, McCall AE, Gordadze PR, Guo Q, Matzuk MM, Zoghbi HY (1997) Math1 is essential for genesis of cerebellar granule neurons. Nature 390:169-172. [Abstract]
Wu MN, Bellen HJ (1997) Genetic dissection of synaptic transmission in Drosophila. Current Opinion in Neurobiology 7:624-630. [Abstract]
Littleton JT, Bhat MA, Bellen HJ (1997) Deciphering the function of neurexins at cellular junctions. Journal of Cell Biology 137:793-796. [Abstract]
Bhat MA, Philp AV, Glover DM, Bellen HJ (1996) Chromatid segregation at anaphase requires the barren product, a novel chromosome-associated protein that interacts with Topoisomerase II. Cell 87:1103-1114. [Abstract]
Baumgartner S, Littleton JT, Broadie K, Bhat MA, Harbecke R, Lengyel JA, Chiquet-Ehrismann R, Prokop A, Bellen HJ (1996) A Drosophila neurexin is required for septate junction and blood-nerve barrier formation and function. Cell 87:1059-1068. [Abstract]
Schulze KL, Bellen HJ (1996) Drosophila syntaxin is required for cell viability and may function in membrane formation and stabilization. Genetics 144:1713-1724. [Abstract]
Salzberg A, Golden K, Bodmer R, Bellen HJ (1996) gutfeeling, a Drosophila gene encoding an antizyme-like protein, is required for late differentiation of neurons and muscles. Genetics 144:183-196. [Abstract]
Ben-Arie N, McCall AE, Berkman S, Eichele G, Bellen HJ, Zoghbi HY (1996) Evolutionary conservation of sequence and expression of the bHLH protein Atonal suggests a conserved role in neurogenesis. Human Molecular Genetics 5:1207-1216. [Abstract]
Rodriguez A, Zhou Z, Tang ML, Meller S, Chen J, Bellen HJ, Kimbrell DA (1996) Identification of immune system and response genes, and novel mutations causing melanotic tumor formation in Drosophila melanogaster. Genetics 143:929-940. [Abstract]
Salzberg A, Bellen HJ (1996) Invertebrate versus vertebrate neurogenesis: variations on the same theme?. Developmental Genetics 18:1-10. [Abstract]
Bellen HJ, Smith RF (1995) FlyBase: a virtual Drosophila cornucopia. Trends in Genetics 11:456-457. [Abstract]
Broadie K, Prokop A, Bellen HJ, O'Kane CJ, Schulze KL, Sweeney ST (1995) Syntaxin and synaptobrevin function downstream of vesicle docking in Drosophila. Neuron 15:663-673. [Abstract]
Littleton JT, Bellen HJ (1995) Presynaptic proteins involved in exocytosis in Drosophila melanogaster: a genetic analysis. Invertebrate Neuroscience 1:3-13. [Abstract]
Kania A, Salzberg A, Bhat M, D'Evelyn D, He Y, Kiss I, Bellen HJ (1995) P-element mutations affecting embryonic peripheral nervous system development in Drosophila melanogaster. Genetics 139:1663-1678. [Abstract]
Littleton JT, Bellen HJ (1995) Synaptotagmin controls and modulates synaptic-vesicle fusion in a Ca2+-dependent manner. Trends in Neurosciences 18:177-183. [Abstract]
Kania A, Bellen HJ (1995) Mutations in neuromusculin, a gene encoding a cell adhesion molecule, cause nervous system defects. Roux's Archives of Developmental Biology 204:259-269. [Abstract]
Schulze KL, Broadie K, Perin MS, Bellen HJ (1995) Genetic and electrophysiological studies of Drosophila syntaxin-1A demonstrate its role in nonneuronal secretion and neurotransmission. Cell 80:311-320. [Abstract]
Littleton JT, Stern M, Perin M, Bellen HJ (1994) Calcium dependence of neurotransmitter release and rate of spontaneous vesicle fusions are altered in Drosophila synaptotagmin mutants. Proceedings of the National Academy of Sciences USA 91:10888-10892. Current Biology 5:257-9, Dispatch. [Abstract]
Schulze KL, Littleton JT, Salzberg A, Halachmi N, Stern M, Lev Z, Bellen HJ (1994) rop, a Drosophila homolog of yeast Sec1 and vertebrate n-Sec1/Munc-18 proteins, is a negative regulator of neurotransmitter release in vivo. Neuron 13:1099-1108. [Abstract]
Broadie K, Bellen HJ, DiAntonio A, Littleton JT, Schwarz TL (1994) Absence of synaptotagmin disrupts excitation-secretion coupling during synaptic transmission. Proceedings of the National Academy of Sciences USA 91:10727-10731. [Abstract]
Littleton JT, Bellen HJ. (1994) Genetic and phenotypic analysis of thirteen essential genes in cytological interval 22F1-2; 23B1-2 reveals novel genes required for neural development in Drosophila. Genetics 138:111-123. [Abstract]
Salzberg A, D'Evelyn D, Schulze KL, Lee JK, Strumpf D, Tsai L, Bellen HJ (1994) Mutations affecting the pattern of the PNS in Drosophila reveal novel aspects of neuronal development. Neuron 13:269-287. [Abstract]
Eldon E, Kooyer S, D'Evelyn D, Duman M, Lawinger P, Botas J, Bellen HJ (1994) The Drosophila 18 wheeler is required for morphogenesis and has striking similarities to Toll. Development 120:885-899. [Abstract]
Kania A, Han PL, Kim YT, Bellen HJ (1993) neuromusculin, a Drosophila gene expressed in peripheral neuronal precursors and muscles, encodes a cell adhesion molecule. Neuron 11:673-687. [Abstract]
Littleton JT, Stern M, Schulze K, Perin M, Bellen HJ (1993) Mutational analysis of Drosophila synaptotagmin demonstrates its essential role in Ca2+-activated neurotransmitter release. Cell 74:1125-1134. Cell 74:947-50, Minireview. [Abstract]
Littleton JT, Bellen HJ, Perin MS (1993) Expression of synaptotagmin in Drosophila reveals transport and localization of synaptic vesicles to the synapse. Development 118:1077-1088. [Abstract]
Bellen HJ, Kooyer S, D'Evelyn D, Pearlman J (1992) The Drosophila Couch potato protein is expressed in nuclei of peripheral neuronal precursors and shows homology to RNA-binding proteins. Genes and Development 6:2125-2136. [Abstract]
Bellen HJ, Vaessin H, Bier E, Kolodkin A, D'Evelyn D, Kooyer S, Jan YN (1992) The Drosophila couch potato gene: an essential gene required for normal adult behavior. Genetics 131:365-375. [Abstract]
Bellen HJ, D'Evelyn D, Harvey M, Elledge SJ (1992) Isolation of temperature-sensitive diphtheria toxins in yeast and their effects on Drosophila cells. Development 114:787-796. [Abstract]