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We show that developmental synapse elimination requires presynaptic Ube3a activity in\n            <jats:italic>Drosophila<\/jats:italic>\n            neurons. We further identified the domain of Ube3a that is required for its interaction with the kinesin motor. Angelman syndrome\u2013associated missense mutations in the interaction domain attenuate presynaptic targeting of Ube3a and prevent synapse elimination. Conversely, increased Ube3a activity in presynapses leads to precocious synapse elimination and impairs synaptic transmission. Our findings reveal the physiological role of Ube3a and suggest potential pathogenic mechanisms associated with Ube3a dysregulation.\n          <\/jats:p>","DOI":"10.1126\/science.ade8978","type":"journal-article","created":{"date-parts":[[2023,9,14]],"date-time":"2023-09-14T17:58:58Z","timestamp":1694714338000},"page":"1197-1205","update-policy":"https:\/\/doi.org\/10.34133\/aaas_crossmark","source":"Crossref","is-referenced-by-count":41,"title":["Presynaptic Ube3a E3 ligase promotes synapse elimination through down-regulation of BMP signaling"],"prefix":"10.1126","volume":"381","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-3707-1008","authenticated-orcid":true,"given":"Kotaro","family":"Furusawa","sequence":"first","affiliation":[{"name":"Department of Biological Sciences, Graduate School of Science, The University of Tokyo, 7-3-1 Hongo, Bunkyo-ku, Tokyo 113-0033, 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