{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,11,6]],"date-time":"2025-11-06T00:58:12Z","timestamp":1762390692242,"version":"3.37.3"},"reference-count":34,"publisher":"Springer Science and Business Media LLC","issue":"11","license":[{"start":{"date-parts":[[2016,7,9]],"date-time":"2016-07-09T00:00:00Z","timestamp":1468022400000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/501100001349","name":"National Medical Research Council","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100001349","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Hum Genet"],"published-print":{"date-parts":[[2016,11]]},"DOI":"10.1007\/s00439-016-1709-z","type":"journal-article","created":{"date-parts":[[2016,7,9]],"date-time":"2016-07-09T09:19:07Z","timestamp":1468055947000},"page":"1223-1232","update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":12,"title":["FUS-linked essential tremor associated with motor dysfunction in Drosophila"],"prefix":"10.1007","volume":"135","author":[{"given":"Murni","family":"Tio","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Rujing","family":"Wen","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yih Lin","family":"Lim","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Huashan","family":"Wang","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Shuo-Chien","family":"Ling","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yi","family":"Zhao","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Eng-King","family":"Tan","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"297","published-online":{"date-parts":[[2016,7,9]]},"reference":[{"key":"1709_CR1","doi-asserted-by":"crossref","first-page":"3157","DOI":"10.1093\/hmg\/ddu026","volume":"23","author":"DC Angeles","year":"2014","unstructured":"Angeles DC, Ho P, Chua LL, Wang C, Yap YW, Ng C, Zhou ZD, Lim KL, Wszolek ZK, Wang HY, Tan EK (2014) Thiol peroxidases ameliorate LRRK2 mutant-induced mitochondrial and dopaminergic neuronal degeneration in Drosophila. Hum Mol Genet 23:3157\u20133165","journal-title":"Hum Mol Genet"},{"key":"1709_CR2","doi-asserted-by":"crossref","first-page":"152","DOI":"10.1016\/j.neulet.2004.12.071","volume":"379","author":"A Belly","year":"2005","unstructured":"Belly A, Moreau-Gachelin F, Sadoul R, Goldberg Y (2005) Delocalization of the multifunctional RNA splicing factor TLS\/FUS in hippocampal neurones: exclusion from the nucleus and accumulation in dendritic granules and spine heads. Neurosci Lett 379:152\u2013157","journal-title":"Neurosci Lett"},{"key":"1709_CR3","doi-asserted-by":"crossref","first-page":"87","DOI":"10.3389\/fnagi.2014.00087","volume":"6","author":"A Benazzouz","year":"2014","unstructured":"Benazzouz A, Mamad O, Abedi P, Bouali-Benazzouz R, Chetrit J (2014) Involvement of dopamine loss in extrastriatal basal ganglia nuclei in the pathophysiology of Parkinson\u2019s disease. Front Aging Neurosci 6:87","journal-title":"Front Aging Neurosci"},{"key":"1709_CR4","doi-asserted-by":"crossref","first-page":"401","DOI":"10.1242\/dev.118.2.401","volume":"118","author":"AH Brand","year":"1993","unstructured":"Brand AH, Perrimon N (1993) Targeted gene expression as a means of altering cell fates and generating dominant phenotypes. Development 118:401\u2013415","journal-title":"Development"},{"key":"1709_CR5","doi-asserted-by":"crossref","first-page":"407","DOI":"10.1016\/j.parkreldis.2011.12.003","volume":"18","author":"V Chandran","year":"2012","unstructured":"Chandran V, Pal PK (2012) Essential tremor: beyond the motor features. Park Rel Disord 18:407\u2013413","journal-title":"Park Rel Disord"},{"key":"1709_CR6","doi-asserted-by":"crossref","first-page":"477","DOI":"10.1007\/s13238-011-1065-7","volume":"2","author":"Y Chen","year":"2011","unstructured":"Chen Y, Yang M, Deng J, Chen X, Ye Y, Zhu L, Liu J, Ye H, Shen Y, Li Y, Rao EJ, Fushimi K et al (2011) Expression of human FUS protein in Drosophila leads to progressive neurodegeneration. Protein Cell 2:477\u2013486","journal-title":"Protein Cell"},{"key":"1709_CR7","doi-asserted-by":"crossref","first-page":"424","DOI":"10.1111\/j.1460-9568.2007.05655.x","volume":"26","author":"S Dasari","year":"2007","unstructured":"Dasari S, Viele K, Turner AC, Cooper RL (2007) Influence of PCPA and MDMA (ecstasy) on physiology, development and behavior in Drosophila melanogaster. Eur J Neurosci 26:424\u2013438","journal-title":"Eur J Neurosci"},{"key":"1709_CR8","doi-asserted-by":"crossref","first-page":"148","DOI":"10.1016\/S1474-4422(10)70322-7","volume":"10","author":"G Deuschl","year":"2011","unstructured":"Deuschl G, Raethjen J, Hellriegel H, Elble R (2011) Treatment of patients with essential tremor. Lancet Neurol 10:148\u2013161","journal-title":"Lancet Neurol"},{"key":"1709_CR9","doi-asserted-by":"crossref","first-page":"21087","DOI":"10.1073\/pnas.1218850110","volume":"109","author":"AV Devineni","year":"2012","unstructured":"Devineni AV, Heberlein U (2012) Acute ethanol responses in Drosophila are sexually dimorphic. PNAS 109:21087\u201321092","journal-title":"PNAS"},{"key":"1709_CR10","doi-asserted-by":"crossref","first-page":"5677","DOI":"10.1093\/hmg\/ddv281","volume":"24","author":"H Hor","year":"2015","unstructured":"Hor H, Francescatto L, Bartesaghi L, Ortega-Cubero S, Kousi M, Lorenzo-Betancor O, Jimenez-Jimenez FJ, Gironell A, Clarimon J, Drechsel O, Agundez JA, Kenzelmann Broz D et al (2015) Missense mutations in TENM4, a regulator of axon guidance and central myelination, cause essential tremor. Hum Mol Genet 24:5677\u20135686","journal-title":"Hum Mol Genet"},{"key":"1709_CR11","doi-asserted-by":"crossref","first-page":"645","DOI":"10.1212\/WNL.45.4.645","volume":"45","author":"J Jankovic","year":"1995","unstructured":"Jankovic J, Beach J, Schwartz K, Contant C (1995) Tremor and longevity in relatives of patients with Parkinson\u2019s disease, essential tremor and control subjects. Neurology 45:645\u2013648","journal-title":"Neurology"},{"key":"1709_CR12","doi-asserted-by":"crossref","first-page":"3897","DOI":"10.1242\/dev.01255","volume":"131","author":"K Jia","year":"2004","unstructured":"Jia K, Chen D, Riddle DL (2004) The TOR pathway interacts with the insulin signaling pathway to regulate C elegans larval development, metabolism and life span. Development 131:3897\u20133906","journal-title":"Development"},{"key":"1709_CR13","doi-asserted-by":"crossref","first-page":"359","DOI":"10.1111\/ane.12148","volume":"128","author":"FJ Jimenez-Jimenez","year":"2013","unstructured":"Jimenez-Jimenez FJ, Alonso-Navarro H, Garcia-Martin E, Lorenzo- Betancor O, Pastor P, Agundez JA (2013) Update on genetics of essential tremor. Acta Neurol Scand 128:359\u2013371","journal-title":"Acta Neurol Scand"},{"key":"1709_CR14","doi-asserted-by":"crossref","first-page":"421","DOI":"10.1093\/gerona\/glu044","volume":"70","author":"AJ Kahn","year":"2015","unstructured":"Kahn AJ (2015) FOXO3 and related transcription factors in development, aging and exceptional longevity. J Gerontol A Biol Sci Med Sci 70:421\u2013425","journal-title":"J Gerontol A Biol Sci Med Sci"},{"key":"1709_CR15","doi-asserted-by":"crossref","first-page":"21","DOI":"10.1016\/S0163-7258(01)00122-X","volume":"90","author":"P Kwan","year":"2001","unstructured":"Kwan P, Sills GJ, Brodie MJ (2001) The mechanisms of action of commonly used antiepileptic drugs. Pharmacol Ther 90:21\u201334","journal-title":"Pharmacol Ther"},{"key":"1709_CR16","doi-asserted-by":"crossref","first-page":"35","DOI":"10.1016\/j.bbrc.2013.05.036","volume":"436","author":"Z Lei","year":"2013","unstructured":"Lei Z, Chen K, Li H, Liu H, Guo A (2013) The GABA system regulates the sparse coding of odors in the mushroom bodies of Drosophila. Biochem Biophys Res Commun 436:35\u201340","journal-title":"Biochem Biophys Res Commun"},{"key":"1709_CR17","doi-asserted-by":"crossref","first-page":"402","DOI":"10.1006\/meth.2001.1262","volume":"25","author":"KJ Livak","year":"2001","unstructured":"Livak KJ, Schmittgen TD (2001) Analysis of relative gene expression data using real-time quantitative PCR and the 2(-Delta Delta C(T)) method. Methods 25:402\u2013408","journal-title":"Methods"},{"key":"1709_CR18","doi-asserted-by":"crossref","first-page":"235","DOI":"10.1007\/s12311-015-0692-6","volume":"15","author":"ED Louis","year":"2016","unstructured":"Louis ED (2016) Linking essential tremor to the cerebellum: neuropathological evidence. Cerebellum 15:235\u2013242","journal-title":"Cerebellum"},{"key":"1709_CR19","doi-asserted-by":"crossref","first-page":"534","DOI":"10.1002\/mds.22838","volume":"25","author":"ED Louis","year":"2010","unstructured":"Louis ED, Ferreira JJ (2010) How common is the most common adult movement disorder? Update on the worldwide prevalence of essential tremor. Mov Disord 25:534\u2013541","journal-title":"Mov Disord"},{"key":"1709_CR20","doi-asserted-by":"crossref","first-page":"1982","DOI":"10.1212\/01.wnl.0000279339.87987.d7","volume":"69","author":"ED Louis","year":"2007","unstructured":"Louis ED, Benito-Leon J, Ottman R, Bermejo-Pareja F (2007) A population-based study of mortality in essential tremor. Neurology 69:1982\u20131989","journal-title":"Neurology"},{"key":"1709_CR21","doi-asserted-by":"crossref","first-page":"471","DOI":"10.1007\/s00018-011-0789-0","volume":"69","author":"J Luo","year":"2012","unstructured":"Luo J, Becnel J, Nichols CD, Nassel DR (2012) Insulin-producing cells in the brain of adult Drosophila are regulated by the serotonin 5-HT1A receptor. Cell Mol Life Sci 69:471\u2013484","journal-title":"Cell Mol Life Sci"},{"key":"1709_CR22","doi-asserted-by":"crossref","first-page":"3810","DOI":"10.1093\/hmg\/ddu094","volume":"23","author":"JB Machamer","year":"2014","unstructured":"Machamer JB, Collins SE, Lloyd TE (2014) The ALS gene FUS regulates synaptic transmission at the Drosophila neuromuscular junction. Hum Mol Genet 23:3810\u20133822","journal-title":"Hum Mol Genet"},{"key":"1709_CR23","doi-asserted-by":"crossref","first-page":"313","DOI":"10.1016\/j.ajhg.2012.07.002","volume":"91","author":"ND Merner","year":"2012","unstructured":"Merner ND, Girard SL, Catoire H, Bourassa CV, Belzil VV, Riviere JB, Hince P, Levert A, Dionne-Laporte A, Spiegelman D, Noreau A, Diab S et al (2012) Exome sequencing identifies FUS mutations as a cause of essential tremor. Am J Hum Genet 91:313\u2013319","journal-title":"Am J Hum Genet"},{"key":"1709_CR24","doi-asserted-by":"crossref","first-page":"273","DOI":"10.1007\/s00401-012-1043-z","volume":"125","author":"JC Mitchell","year":"2013","unstructured":"Mitchell JC, McGoldrick P, Vance C, Hortobagyi T, Sreedharan J, Rogelj B, Tudor EL, Smith BN, Klasen C et al (2013) Overexpression of human wild-type FUS causes progressive motor neuron degeneration in an age- and dose-dependent fashion. Acta Neuropathol 125:273\u2013288","journal-title":"Acta Neuropathol"},{"key":"1709_CR25","doi-asserted-by":"crossref","first-page":"5","DOI":"10.1186\/s13293-015-0023-0","volume":"6","author":"EH Morrow","year":"2015","unstructured":"Morrow EH (2015) The evolution of sex differences in disease. Biol Sex Differ 6:5","journal-title":"Biol Sex Differ"},{"key":"1709_CR26","doi-asserted-by":"crossref","first-page":"105","DOI":"10.1093\/brain\/awr301","volume":"135","author":"S Paris-Robidas","year":"2012","unstructured":"Paris-Robidas S, Brochu E, Sintes M, Emond V, Bousquet M, Vandal M, Pilote M, Tremblay C, Di Paolo T, Rajput AH, Rajput A, Calon F (2012) Defective dentate nucleus GABA receptors in essential tremor. Brain 135:105\u2013116","journal-title":"Brain"},{"key":"1709_CR27","first-page":"3717","volume":"9","author":"DD Prasad","year":"1994","unstructured":"Prasad DD, Ouchida M, Lee L, Rao VN, Reddy ES (1994) TLS\/FUS fusion domain of TLS\/FUS-erg chimeric protein resulting from the t(16;21) chromosomal translocation in human myeloid leukemia functions as a transcriptional activation domain. Oncogene 9:3717\u20133729","journal-title":"Oncogene"},{"key":"1709_CR28","doi-asserted-by":"crossref","first-page":"503","DOI":"10.1002\/neu.10297","volume":"58","author":"ER Reynolds","year":"2004","unstructured":"Reynolds ER, Stauffer EA, Feeney L, Rojahn E, Jacobs B, McKeever C (2004) Treatment with the antiepileptic drugs phenytoin and gabapentin ameliorates seizure and paralysis of Drosophila bang-sensitive mutants. J Neurobiol 58:503\u2013513","journal-title":"J Neurobiol"},{"key":"1709_CR29","doi-asserted-by":"crossref","first-page":"1361","DOI":"10.1152\/jn.90750.2008","volume":"101","author":"F Saitow","year":"2009","unstructured":"Saitow F, Murano M, Suzuki H (2009) Modulatory effects of serotonin on GABAergic synaptic transmission and membrane properties in the deep cerebellar nuclei. J Neurophysiol 101:1361\u20131374","journal-title":"J Neurophysiol"},{"key":"1709_CR30","doi-asserted-by":"crossref","first-page":"835","DOI":"10.1093\/nar\/23.5.835","volume":"23","author":"DT Stolow","year":"1995","unstructured":"Stolow DT, Haynes SR (1995) Cabeza, a Drosophila gene encoding a novel RNA binding protein, shares homology with EWS and TLS, two genes involved in human sarcoma formation. Nucleic Acids Res 23:835\u2013843","journal-title":"Nucleic Acids Res"},{"key":"1709_CR31","doi-asserted-by":"crossref","first-page":"1208","DOI":"10.1126\/science.1165942","volume":"323","author":"C Vance","year":"2009","unstructured":"Vance C, Rogelj B, Hortobagyi T, De Vos KJ, Nishimura AL, Sreedharan J, Hu X, Smith B, Ruddy D, Wright P, Ganesalingam J, William KL et al (2009) Mutations in FUS, an RNA processing protein, cause familial amyotrophic lateral sclerosis type 6. Science 323:1208\u20131211","journal-title":"Science"},{"key":"1709_CR32","doi-asserted-by":"crossref","first-page":"242","DOI":"10.1016\/j.cub.2013.12.015","volume":"24","author":"AC Philipsborn Von","year":"2014","unstructured":"Von Philipsborn AC, Jorchel S, Tirian L, Demir E, Morita T, Stern DL, Dickson BJ (2014) Cellular and behavioural functions of fruitless isoforms in Drosophila courtship. Curr Biol 24:242\u2013251","journal-title":"Curr Biol"},{"key":"1709_CR33","doi-asserted-by":"crossref","first-page":"13987","DOI":"10.1073\/pnas.0801030105","volume":"105","author":"BJ Willcox","year":"2008","unstructured":"Willcox BJ, Donlon TA, He Q, Chen R, Grove JS, Yano K, Masaki KH, Willcox DC, Rodriguez B, Curb JD (2008) FOXO3A genotype is strongly associated with human longevity. Proc Natl Acad Sci USA 105:13987\u201313992","journal-title":"Proc Natl Acad Sci USA"},{"key":"1709_CR34","doi-asserted-by":"crossref","first-page":"2897","DOI":"10.1152\/jn.00287.2010","volume":"105","author":"JG Xue","year":"2011","unstructured":"Xue JG, Masuoka T, Gong XD, Chen KS, Yanagawa Y, Law SK, Konishi S (2011) NMDA receptor activation enhances inhibitory GABAergic transmission onto hippocampal pyramidal neurons via presynaptic and postsynaptic mechanisms. J Neurophysiol 105:2897\u20132906","journal-title":"J Neurophysiol"}],"container-title":["Human Genetics"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/link.springer.com\/content\/pdf\/10.1007\/s00439-016-1709-z.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/link.springer.com\/article\/10.1007\/s00439-016-1709-z\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/link.springer.com\/content\/pdf\/10.1007\/s00439-016-1709-z.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2022,7,2]],"date-time":"2022-07-02T22:24:46Z","timestamp":1656800686000},"score":1,"resource":{"primary":{"URL":"http:\/\/link.springer.com\/10.1007\/s00439-016-1709-z"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2016,7,9]]},"references-count":34,"journal-issue":{"issue":"11","published-print":{"date-parts":[[2016,11]]}},"alternative-id":["1709"],"URL":"https:\/\/doi.org\/10.1007\/s00439-016-1709-z","relation":{},"ISSN":["0340-6717","1432-1203"],"issn-type":[{"type":"print","value":"0340-6717"},{"type":"electronic","value":"1432-1203"}],"subject":[],"published":{"date-parts":[[2016,7,9]]}}}