{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,4]],"date-time":"2026-06-04T02:35:15Z","timestamp":1780540515040,"version":"3.54.1"},"reference-count":98,"publisher":"Wiley","issue":"5-6","license":[{"start":{"date-parts":[[2020,5,25]],"date-time":"2020-05-25T00:00:00Z","timestamp":1590364800000},"content-version":"vor","delay-in-days":24,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Developmental Neurobiology"],"published-print":{"date-parts":[[2020,5]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>Angelman syndrome (AS) is a rare neurodevelopmental disorder characterized by severe mental retardation, microcephaly, speech impairment, frequent epilepsy, EEG abnormalities, ataxic movements, tongue protrusion, bursts of laughter, sleep abruptions, and hyperactivity. AS results from loss of function of the imprinted UBE3A (ubiquitin\u2010protein ligase E3A) gene on chromosome 15q11\u2013q13, including a mutation on the maternal allele of Ube3a, a large deletion of the maternally inherited chromosomal region 15q11\u201313, paternal uniparental disomy of chromosome 15q11\u201313, or an imprinting defect. The Ube3a maternal deleted mouse model recaptured the major phenotypes of AS patients include seizure, learning and memory impairments, sleep disturbance, and motor problems. Owing to the activity\u2010dependent structural and functional plasticity, dendritic spines are believed as the basic subcellular compartment for learning and memory and the sites where LTP and LTD are induced. Defects of spine formation and dynamics are common among several neurodevelopmental disorders and neuropsychiatric disorders including AS and reflect the underlying synaptopathology, which drives clinically relevant behavioral deficits. This review will summarize the impaired spine density, morphology, and synaptic plasticity in AS and propose that future explorations on spine dynamics and synaptic plasticity may help develop novel interventions and therapy for neurodevelopmental disorders like AS.<\/jats:p>","DOI":"10.1002\/dneu.22757","type":"journal-article","created":{"date-parts":[[2020,5,7]],"date-time":"2020-05-07T09:07:53Z","timestamp":1588842473000},"page":"200-209","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":6,"title":["Characterizing spine issues: If offers novel therapeutics to Angelman syndrome"],"prefix":"10.1002","volume":"80","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-2153-256X","authenticated-orcid":false,"given":"Xin","family":"Yang","sequence":"first","affiliation":[{"name":"Division of Life Science Hong Kong University of Science and Technology Hong Kong China"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2020,5,25]]},"reference":[{"key":"e_1_2_5_2_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.1212-12.2012"},{"key":"e_1_2_5_3_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.conb.2017.06.001"},{"key":"e_1_2_5_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuron.2008.08.012"},{"key":"e_1_2_5_5_1","doi-asserted-by":"publisher","DOI":"10.1038\/nrneurol.2017.45"},{"key":"e_1_2_5_6_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuron.2015.03.009"},{"key":"e_1_2_5_7_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.20-01-00089.2000"},{"key":"e_1_2_5_8_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuron.2017.08.008"},{"key":"e_1_2_5_9_1","doi-asserted-by":"publisher","DOI":"10.1146\/annurev.physiol.010908.163140"},{"key":"e_1_2_5_10_1","doi-asserted-by":"publisher","DOI":"10.1891\/0730-0832.36.3.142"},{"key":"e_1_2_5_11_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.c.30273"},{"key":"e_1_2_5_12_1","doi-asserted-by":"publisher","DOI":"10.1038\/ejhg.2014.93"},{"key":"e_1_2_5_13_1","doi-asserted-by":"publisher","DOI":"10.1038\/nrneurol.2016.133"},{"key":"e_1_2_5_14_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.3353-13.2014"},{"key":"e_1_2_5_15_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.1004487107"},{"key":"e_1_2_5_16_1","doi-asserted-by":"publisher","DOI":"10.3389\/fnsys.2014.00221"},{"key":"e_1_2_5_17_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuron.2006.08.033"},{"key":"e_1_2_5_18_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.nbd.2017.10.016"},{"key":"e_1_2_5_19_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.nbd.2016.08.002"},{"key":"e_1_2_5_20_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuropharm.2016.12.009"},{"key":"e_1_2_5_21_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.nbd.2005.04.003"},{"key":"e_1_2_5_22_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuron.2014.09.024"},{"key":"e_1_2_5_23_1","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/ddm288"},{"key":"e_1_2_5_24_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.2194-15.2015"},{"key":"e_1_2_5_25_1","doi-asserted-by":"publisher","DOI":"10.1007\/s12035-018-0871-9"},{"key":"e_1_2_5_26_1","doi-asserted-by":"publisher","DOI":"10.1038\/nn997"},{"key":"e_1_2_5_27_1","doi-asserted-by":"publisher","DOI":"10.1038\/ncomms15038"},{"key":"e_1_2_5_28_1","doi-asserted-by":"publisher","DOI":"10.1038\/nature13664"},{"key":"e_1_2_5_29_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.1067-10.2010"},{"key":"e_1_2_5_30_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.cell.2010.01.026"},{"key":"e_1_2_5_31_1","doi-asserted-by":"publisher","DOI":"10.1152\/jn.00913.2010"},{"key":"e_1_2_5_32_1","doi-asserted-by":"publisher","DOI":"10.1111\/ejn.12893"},{"key":"e_1_2_5_33_1","doi-asserted-by":"publisher","DOI":"10.1126\/science.1209236"},{"key":"e_1_2_5_34_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuron.2005.01.003"},{"key":"e_1_2_5_35_1","doi-asserted-by":"publisher","DOI":"10.1038\/nature10726"},{"key":"e_1_2_5_36_1","doi-asserted-by":"publisher","DOI":"10.1038\/ncomms5742"},{"key":"e_1_2_5_37_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.41.3.416"},{"key":"e_1_2_5_38_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.1700866114"},{"key":"e_1_2_5_39_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0896-6273(00)80596-6"},{"key":"e_1_2_5_40_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0012278"},{"key":"e_1_2_5_41_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.pcl.2015.03.004"},{"key":"e_1_2_5_42_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.biopsych.2012.01.021"},{"key":"e_1_2_5_43_1","doi-asserted-by":"publisher","DOI":"10.1098\/rstb.2016.0158"},{"key":"e_1_2_5_44_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.1511-17.2017"},{"key":"e_1_2_5_45_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.4204-15.2016"},{"key":"e_1_2_5_46_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pbio.1001900"},{"key":"e_1_2_5_47_1","doi-asserted-by":"publisher","DOI":"10.1152\/physrev.00010.2015"},{"key":"e_1_2_5_48_1","doi-asserted-by":"publisher","DOI":"10.1038\/35016089"},{"key":"e_1_2_5_49_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0092-8674(03)00122-3"},{"key":"e_1_2_5_50_1","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/ddy345"},{"key":"e_1_2_5_51_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.conb.2018.08.010"},{"key":"e_1_2_5_52_1","doi-asserted-by":"publisher","DOI":"10.1101\/cshperspect.a005710"},{"key":"e_1_2_5_53_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.0611698104"},{"key":"e_1_2_5_54_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.1504809112"},{"key":"e_1_2_5_55_1","doi-asserted-by":"publisher","DOI":"10.1038\/nature13975"},{"key":"e_1_2_5_56_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.2883-07.2007"},{"key":"e_1_2_5_57_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.2509-12.2013"},{"key":"e_1_2_5_58_1","doi-asserted-by":"publisher","DOI":"10.1006\/nbdi.2001.0463"},{"key":"e_1_2_5_59_1","doi-asserted-by":"publisher","DOI":"10.1038\/nature13294"},{"key":"e_1_2_5_60_1","doi-asserted-by":"publisher","DOI":"10.3389\/fnins.2018.00412"},{"key":"e_1_2_5_61_1","doi-asserted-by":"publisher","DOI":"10.1128\/MCB.19.2.1182"},{"key":"e_1_2_5_62_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.scr.2018.09.015"},{"key":"e_1_2_5_63_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.scr.2018.101363"},{"key":"e_1_2_5_64_1","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M509677200"},{"key":"e_1_2_5_65_1","doi-asserted-by":"publisher","DOI":"10.3389\/fnmol.2017.00234"},{"key":"e_1_2_5_66_1","doi-asserted-by":"publisher","DOI":"10.1038\/nn.2741"},{"key":"e_1_2_5_67_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.1846-13.2014"},{"key":"e_1_2_5_68_1","first-page":"3999","article-title":"Loss of hierarchical imprinting regulation at the Prader\u2013Willi\/Angelman syndrome locus in human iPSCs","volume":"27","author":"P\u00f3lvora\u2010Brand\u00e3o D.","year":"2018","journal-title":"Human Molecular Genetics"},{"key":"e_1_2_5_69_1","doi-asserted-by":"publisher","DOI":"10.1186\/s12915-016-0261-6"},{"key":"e_1_2_5_70_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.2376-15.2015"},{"key":"e_1_2_5_71_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.0083-18.2018"},{"key":"e_1_2_5_72_1","doi-asserted-by":"publisher","DOI":"10.1152\/physrev.00012.2013"},{"key":"e_1_2_5_73_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.1001281107"},{"key":"e_1_2_5_74_1","first-page":"625418","article-title":"Deleting a UBE3A substrate rescues impaired hippocampal physiology and learning in Angelman syndrome mice","author":"Sell G. L.","year":"2019","journal-title":"bioRxiv"},{"key":"e_1_2_5_75_1","doi-asserted-by":"publisher","DOI":"10.1146\/annurev.cellbio.23.090506.123516"},{"key":"e_1_2_5_76_1","doi-asserted-by":"publisher","DOI":"10.1172\/JCI80554"},{"key":"e_1_2_5_77_1","doi-asserted-by":"publisher","DOI":"10.1126\/scitranslmed.3002627"},{"key":"e_1_2_5_78_1","doi-asserted-by":"publisher","DOI":"10.1186\/s13229-019-0277-1"},{"key":"e_1_2_5_79_1","doi-asserted-by":"publisher","DOI":"10.1186\/s13229-018-0231-7"},{"key":"e_1_2_5_80_1","doi-asserted-by":"publisher","DOI":"10.7554\/eLife.37993"},{"key":"e_1_2_5_81_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.4276-14.2015"},{"key":"e_1_2_5_82_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00018-016-2269-z"},{"key":"e_1_2_5_83_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.celrep.2015.06.023"},{"key":"e_1_2_5_84_1","doi-asserted-by":"publisher","DOI":"10.1126\/science.aag3260"},{"key":"e_1_2_5_85_1","doi-asserted-by":"publisher","DOI":"10.1098\/rstb.2013.0288"},{"key":"e_1_2_5_86_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10354-015-0408-z"},{"key":"e_1_2_5_87_1","doi-asserted-by":"publisher","DOI":"10.1038\/nn.3996"},{"key":"e_1_2_5_88_1","doi-asserted-by":"publisher","DOI":"10.1038\/nn1845"},{"key":"e_1_2_5_89_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuron.2012.03.036"},{"key":"e_1_2_5_90_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.0878-15.2016"},{"key":"e_1_2_5_91_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.1820131116"},{"key":"e_1_2_5_92_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.nbd.2019.104585"},{"key":"e_1_2_5_93_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuron.2008.05.014"},{"key":"e_1_2_5_94_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.0805291105"},{"key":"e_1_2_5_95_1","doi-asserted-by":"publisher","DOI":"10.1038\/cr.2017.132"},{"key":"e_1_2_5_96_1","doi-asserted-by":"publisher","DOI":"10.1038\/nn.2327"},{"key":"e_1_2_5_97_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.cell.2015.06.045"},{"key":"e_1_2_5_98_1","doi-asserted-by":"publisher","DOI":"10.1126\/scitranslmed.aar4338"},{"key":"e_1_2_5_99_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuron.2005.04.001"}],"container-title":["Developmental Neurobiology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1002%2Fdneu.22757","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/dneu.22757","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1002\/dneu.22757","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/dneu.22757","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,9,5]],"date-time":"2023-09-05T09:52:14Z","timestamp":1693907534000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/dneu.22757"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2020,5]]},"references-count":98,"journal-issue":{"issue":"5-6","published-print":{"date-parts":[[2020,5]]}},"alternative-id":["10.1002\/dneu.22757"],"URL":"https:\/\/doi.org\/10.1002\/dneu.22757","archive":["Portico"],"relation":{},"ISSN":["1932-8451","1932-846X"],"issn-type":[{"value":"1932-8451","type":"print"},{"value":"1932-846X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2020,5]]},"assertion":[{"value":"2019-09-27","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2020-05-01","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2020-05-25","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}