{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,9,29]],"date-time":"2025-09-29T12:00:08Z","timestamp":1759147208561,"version":"3.40.5"},"reference-count":50,"publisher":"Wiley","issue":"12","license":[{"start":{"date-parts":[[2020,6,12]],"date-time":"2020-06-12T00:00:00Z","timestamp":1591920000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/501100003725","name":"National Research Foundation of Korea","doi-asserted-by":"publisher","award":["NRF\u20102017M3C1B607057","NRF\u20102018R1D1A1B07050206"],"award-info":[{"award-number":["NRF\u20102017M3C1B607057","NRF\u20102018R1D1A1B07050206"]}],"id":[{"id":"10.13039\/501100003725","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Glia"],"published-print":{"date-parts":[[2020,12]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:label\/><jats:p>NSD1 is a histone methyltransferase that methylates the lysine 36 at histone H3. <jats:italic>NSD<\/jats:italic> duplication is associated with short stature, microcephaly, intellectual disability, and behavioral defects in humans. Ectopic overexpression of <jats:italic>NSD<\/jats:italic>, an <jats:italic>NSD1<\/jats:italic> homolog in <jats:italic>Drosophila<\/jats:italic>, was shown to induce developmental abnormalities via apoptosis. In this study, to investigate the effects of <jats:italic>NSD<\/jats:italic> overexpression on <jats:italic>Drosophila<\/jats:italic> brain development, we first examined the typical <jats:italic>NSD<\/jats:italic> expression pattern in larval brains and found that endogenous <jats:italic>NSD<\/jats:italic> promoter activity was detected only in subsets of glial cells. Pan\u2010glial, but not pan\u2010neuronal, <jats:italic>NSD<\/jats:italic> overexpression induced apoptosis in larval brain cells. However, pan\u2010glial <jats:italic>NSD<\/jats:italic> overexpression also induced caspase\u20103 cleavage in neuronal cells. Among the various glial cell types, <jats:italic>NSD<\/jats:italic> overexpression in only astrocytic glia induced apoptosis and abnormal learning defects in the larval stage. Furthermore, <jats:italic>NSD<\/jats:italic> overexpression downregulated the expression of various astrocyte\u2010specific genes, including <jats:italic>draper<\/jats:italic> (<jats:italic>drpr<\/jats:italic>), possibly owing to an indirect effect of <jats:italic>NSD<\/jats:italic> overexpression\u2010induced astrocytic apoptosis. Since <jats:italic>drpr<\/jats:italic> plays a role in axon pruning during mushroom body (MB) formation in <jats:italic>Drosophila<\/jats:italic> astrocytes, we examined the effect of astrocytic <jats:italic>NSD<\/jats:italic> overexpression on this process and found that it disrupted the clearance of \u03b3\u2010neurons in the MB, subsequently inducing arrhythmic locomotor activity of the fly. Thus, these results suggest that aberrant <jats:italic>NSD<\/jats:italic> overexpression may cause neurodevelopmental disorders by interfering with crucial functions of astrocytes in the brain, underlining the importance of the tightly controlled astrocytic <jats:italic>NSD<\/jats:italic> expression for proper neurodevelopment.<\/jats:p><\/jats:sec>","DOI":"10.1002\/glia.23867","type":"journal-article","created":{"date-parts":[[2020,6,12]],"date-time":"2020-06-12T19:39:22Z","timestamp":1591990762000},"page":"2503-2516","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":12,"title":["Overexpression of <scp>H3K36<\/scp> methyltransferase <scp>NSD<\/scp> in glial cells affects brain development in <scp><i>Drosophila<\/i><\/scp>"],"prefix":"10.1002","volume":"68","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-4519-2555","authenticated-orcid":false,"given":"Taejoon","family":"Kim","sequence":"first","affiliation":[{"name":"Department of Biological Sciences, CHANS Research Center Konkuk University  Seoul South Korea"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5986-4941","authenticated-orcid":false,"given":"Hyewon","family":"Shin","sequence":"additional","affiliation":[{"name":"Department of Biological Sciences, CHANS Research Center Konkuk University  Seoul South Korea"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2967-9443","authenticated-orcid":false,"given":"Bokyeong","family":"Song","sequence":"additional","affiliation":[{"name":"Department of Biological Sciences, CHANS Research Center Konkuk University  Seoul South Korea"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4746-3734","authenticated-orcid":false,"given":"Chihyun","family":"Won","sequence":"additional","affiliation":[{"name":"Department of Biological Sciences, CHANS Research Center Konkuk University  Seoul South Korea"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hideki","family":"Yoshida","sequence":"additional","affiliation":[{"name":"Department of Applied Biology Kyoto Institute of Technology  Kyoto Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Masamitsu","family":"Yamaguchi","sequence":"additional","affiliation":[{"name":"Department of Applied Biology Kyoto Institute of Technology  Kyoto Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kyoung Sang","family":"Cho","sequence":"additional","affiliation":[{"name":"Department of Biological Sciences, CHANS Research Center Konkuk University  Seoul South Korea"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1392-8516","authenticated-orcid":false,"given":"Im\u2010Soon","family":"Lee","sequence":"additional","affiliation":[{"name":"Department of Biological Sciences, CHANS Research Center Konkuk University  Seoul South Korea"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2020,6,12]]},"reference":[{"key":"e_1_2_9_2_1","doi-asserted-by":"publisher","DOI":"10.1038\/nrg.2016.59"},{"key":"e_1_2_9_3_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuron.2008.10.013"},{"key":"e_1_2_9_4_1","doi-asserted-by":"publisher","DOI":"10.1002\/mrdd.20009"},{"key":"e_1_2_9_5_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.0906831106"},{"key":"e_1_2_9_6_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10048-012-0315-z"},{"key":"e_1_2_9_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuron.2007.10.001"},{"key":"e_1_2_9_8_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.31329"},{"key":"e_1_2_9_9_1","doi-asserted-by":"publisher","DOI":"10.2741\/4782"},{"key":"e_1_2_9_10_1","doi-asserted-by":"publisher","DOI":"10.1038\/nature12776"},{"key":"e_1_2_9_11_1","doi-asserted-by":"publisher","DOI":"10.1038\/s42003-018-0031-9"},{"key":"e_1_2_9_12_1","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.201306099"},{"key":"e_1_2_9_13_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajpath.2014.06.018"},{"key":"e_1_2_9_14_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.conb.2019.07.001"},{"key":"e_1_2_9_15_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.nbscr.2019.01.003"},{"key":"e_1_2_9_16_1","doi-asserted-by":"publisher","DOI":"10.1101\/cshperspect.a020552"},{"key":"e_1_2_9_17_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.tins.2005.12.002"},{"key":"e_1_2_9_18_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0086178"},{"key":"e_1_2_9_19_1","doi-asserted-by":"publisher","DOI":"10.1002\/glia.21092"},{"key":"e_1_2_9_20_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.bbrc.2012.01.122"},{"key":"e_1_2_9_21_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.3004-14.2015"},{"key":"e_1_2_9_22_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.yexcr.2017.12.010"},{"key":"e_1_2_9_23_1","doi-asserted-by":"publisher","DOI":"10.3390\/ijms10020407"},{"key":"e_1_2_9_24_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.bbrc.2018.01.156"},{"key":"e_1_2_9_25_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.conb.2010.04.001"},{"key":"e_1_2_9_26_1","doi-asserted-by":"publisher","DOI":"10.1002\/dvdy.21094"},{"key":"e_1_2_9_27_1","doi-asserted-by":"publisher","DOI":"10.3390\/ijms18071404"},{"key":"e_1_2_9_28_1","doi-asserted-by":"publisher","DOI":"10.1007\/s13258-018-0649-5"},{"key":"e_1_2_9_29_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pbio.1000569"},{"key":"e_1_2_9_30_1","doi-asserted-by":"publisher","DOI":"10.1002\/glia.23115"},{"key":"e_1_2_9_31_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.chembiol.2013.10.016"},{"key":"e_1_2_9_32_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.cell.2008.02.052"},{"key":"e_1_2_9_33_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng863"},{"key":"e_1_2_9_34_1","doi-asserted-by":"publisher","DOI":"10.1242\/dmm.018069"},{"key":"e_1_2_9_35_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.126.18.4065"},{"key":"e_1_2_9_36_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.pediatrneurol.2008.11.013"},{"key":"e_1_2_9_37_1","doi-asserted-by":"publisher","DOI":"10.1111\/ejn.12646"},{"key":"e_1_2_9_38_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.0912493107"},{"key":"e_1_2_9_39_1","doi-asserted-by":"publisher","DOI":"10.1101\/gad.188326.112"},{"key":"e_1_2_9_40_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.cub.2011.03.027"},{"key":"e_1_2_9_41_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.devcel.2016.09.028"},{"key":"e_1_2_9_42_1","doi-asserted-by":"publisher","DOI":"10.1159\/000345578"},{"key":"e_1_2_9_43_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM196407162710301"},{"key":"e_1_2_9_44_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.cell.2007.10.036"},{"key":"e_1_2_9_45_1","doi-asserted-by":"publisher","DOI":"10.1101\/pdb.top067587"},{"key":"e_1_2_9_46_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuron.2014.06.026"},{"key":"e_1_2_9_47_1","doi-asserted-by":"publisher","DOI":"10.1111\/bpa.12537"},{"key":"e_1_2_9_48_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.celrep.2013.12.016"},{"key":"e_1_2_9_49_1","doi-asserted-by":"publisher","DOI":"10.1002\/glia.23459"},{"key":"e_1_2_9_50_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.conb.2014.04.005"},{"key":"e_1_2_9_51_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.33769"}],"container-title":["Glia"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1002%2Fglia.23867","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/glia.23867","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1002\/glia.23867","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/glia.23867","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,9,3]],"date-time":"2023-09-03T01:17:55Z","timestamp":1693703875000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/glia.23867"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2020,6,12]]},"references-count":50,"journal-issue":{"issue":"12","published-print":{"date-parts":[[2020,12]]}},"alternative-id":["10.1002\/glia.23867"],"URL":"https:\/\/doi.org\/10.1002\/glia.23867","archive":["Portico"],"relation":{},"ISSN":["0894-1491","1098-1136"],"issn-type":[{"type":"print","value":"0894-1491"},{"type":"electronic","value":"1098-1136"}],"subject":[],"published":{"date-parts":[[2020,6,12]]},"assertion":[{"value":"2019-11-05","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2020-05-16","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2020-06-12","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}