{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,17]],"date-time":"2026-07-17T14:37:19Z","timestamp":1784299039866,"version":"3.55.0"},"reference-count":26,"publisher":"Elsevier BV","issue":"4","license":[{"start":{"date-parts":[[2020,10,1]],"date-time":"2020-10-01T00:00:00Z","timestamp":1601510400000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2020,10,1]],"date-time":"2020-10-01T00:00:00Z","timestamp":1601510400000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"},{"start":{"date-parts":[[2020,8,13]],"date-time":"2020-08-13T00:00:00Z","timestamp":1597276800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"funder":[{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","award":["DP1-DA044359"],"award-info":[{"award-number":["DP1-DA044359"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000054","name":"National Cancer Institute","doi-asserted-by":"publisher","award":["LMP400"],"award-info":[{"award-number":["LMP400"]}],"id":[{"id":"10.13039\/100000054","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000893","name":"Simons Foundation","doi-asserted-by":"publisher","award":["495112"],"award-info":[{"award-number":["495112"]}],"id":[{"id":"10.13039\/100000893","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100005280","name":"American Association of University Women","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100005280","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100007703","name":"North Carolina State University","doi-asserted-by":"publisher","award":["NSF-1830910"],"award-info":[{"award-number":["NSF-1830910"]}],"id":[{"id":"10.13039\/100007703","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100011082","name":"School of Medicine, University of North Carolina at Chapel Hill","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100011082","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["cell.com","elsevier.com","sciencedirect.com"],"crossmark-restriction":true},"short-container-title":["Stem Cell Reports"],"published-print":{"date-parts":[[2020,10]]},"DOI":"10.1016\/j.stemcr.2020.08.006","type":"journal-article","created":{"date-parts":[[2020,9,10]],"date-time":"2020-09-10T10:44:13Z","timestamp":1599734653000},"page":"845-854","update-policy":"https:\/\/doi.org\/10.1016\/elsevier_cm_policy","source":"Crossref","is-referenced-by-count":29,"title":["Human Cerebral Organoids Reveal Early Spatiotemporal Dynamics and Pharmacological Responses of UBE3A"],"prefix":"10.1016","volume":"15","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-5037-3014","authenticated-orcid":false,"given":"Dilara","family":"Sen","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alexis","family":"Voulgaropoulos","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zuzana","family":"Drobna","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8958-1232","authenticated-orcid":false,"given":"Albert J.","family":"Keung","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"78","reference":[{"key":"10.1016\/j.stemcr.2020.08.006_bib1","doi-asserted-by":"crossref","first-page":"1235","DOI":"10.1038\/s41593-019-0425-0","article-title":"Loss of nuclear UBE3A causes electrophysiological and behavioral deficits in mice and is associated with Angelman syndrome","volume":"22","author":"Avagliano Trezza","year":"2019","journal-title":"Nat. Neurosci."},{"key":"10.1016\/j.stemcr.2020.08.006_bib2","doi-asserted-by":"crossref","first-page":"233","DOI":"10.1002\/cne.24063","article-title":"Subcellular organization of UBE3A in neurons","volume":"525","author":"Burette","year":"2017","journal-title":"J.\u00a0Comp. Neurol."},{"key":"10.1016\/j.stemcr.2020.08.006_bib3","doi-asserted-by":"crossref","first-page":"15672","DOI":"10.1073\/pnas.1520760112","article-title":"Human cerebral organoids recapitulate gene expression programs of fetal neocortex development","volume":"112","author":"Camp","year":"2015","journal-title":"Proc. Natl. Acad. Sci. U S A"},{"key":"10.1016\/j.stemcr.2020.08.006_bib4","doi-asserted-by":"crossref","first-page":"17668","DOI":"10.1073\/pnas.1004487107","article-title":"Induced pluripotent stem cell models of the genomic imprinting disorders Angelman and Prader-Willi syndromes","volume":"107","author":"Chamberlain","year":"2010","journal-title":"Proc. Natl. Acad. Sci. U S A"},{"key":"10.1016\/j.stemcr.2020.08.006_bib5","doi-asserted-by":"crossref","first-page":"247","DOI":"10.1523\/JNEUROSCI.2899-04.2005","article-title":"Pax6, Tbr2, and Tbr1 are expressed sequentially by radial glia, intermediate progenitor cells, and postmitotic neurons in developing neocortex","volume":"25","author":"Englund","year":"2005","journal-title":"J.\u00a0Neurosci."},{"key":"10.1016\/j.stemcr.2020.08.006_bib6","doi-asserted-by":"crossref","first-page":"15038","DOI":"10.1038\/ncomms15038","article-title":"Disrupted neuronal maturation in Angelman syndrome-derived induced pluripotent stem cells","volume":"8","author":"Fink","year":"2017","journal-title":"Nat. Commun."},{"key":"10.1016\/j.stemcr.2020.08.006_bib7","first-page":"41","article-title":"Dynamic expression of calretinin in embryonic and early fetal human cortex","volume":"8","author":"Gonzalez-Gomez","year":"2014","journal-title":"Front. Neuroanat."},{"key":"10.1016\/j.stemcr.2020.08.006_bib8","doi-asserted-by":"crossref","first-page":"1707","DOI":"10.1093\/hmg\/11.15.1707","article-title":"Allele-specific expression analysis by RNA-FISH demonstrates preferential maternal expression of UBE3A and imprint maintenance within 15q11-q13 duplications","volume":"11","author":"Herzing","year":"2002","journal-title":"Hum. Mol. Genet."},{"key":"10.1016\/j.stemcr.2020.08.006_bib9","doi-asserted-by":"crossref","first-page":"2181","DOI":"10.1073\/pnas.1815279116","article-title":"A bipartite boundary element restricts UBE3A imprinting to mature neurons","volume":"116","author":"Hsiao","year":"2019","journal-title":"Proc. Natl. Acad. Sci. U S A"},{"key":"10.1016\/j.stemcr.2020.08.006_bib10","doi-asserted-by":"crossref","first-page":"185","DOI":"10.1038\/nature10726","article-title":"Topoisomerase inhibitors unsilence the dormant allele of Ube3a in neurons","volume":"481","author":"Huang","year":"2012","journal-title":"Nature"},{"key":"10.1016\/j.stemcr.2020.08.006_bib11","doi-asserted-by":"crossref","first-page":"393","DOI":"10.1093\/hmg\/ddi456","article-title":"A human imprinting centre demonstrates conserved acquisition but diverged maintenance of imprinting in a mouse model for Angelman syndrome imprinting defects","volume":"15","author":"Johnstone","year":"2006","journal-title":"Hum. Mol. Genet."},{"key":"10.1016\/j.stemcr.2020.08.006_bib12","doi-asserted-by":"crossref","first-page":"1874","DOI":"10.1002\/cne.23507","article-title":"Allelic specificity of Ube3a expression in the mouse brain during postnatal Development","volume":"522","author":"Judson","year":"2014","journal-title":"J.\u00a0Comp. Neurol."},{"key":"10.1016\/j.stemcr.2020.08.006_bib13","doi-asserted-by":"crossref","first-page":"418","DOI":"10.1038\/s41586-019-1654-9","article-title":"Organoid single-cell genomic atlas uncovers human-specific features of brain development","volume":"574","author":"Kanton","year":"2019","journal-title":"Nature"},{"key":"10.1016\/j.stemcr.2020.08.006_bib14","doi-asserted-by":"crossref","first-page":"70","DOI":"10.1038\/ng0197-70","article-title":"UBE3A\/E6-AP mutations cause Angelman syndrome","volume":"15","author":"Kishino","year":"1997","journal-title":"Nat. Genet."},{"key":"10.1016\/j.stemcr.2020.08.006_bib15","doi-asserted-by":"crossref","first-page":"373","DOI":"10.1038\/nature12517","article-title":"Cerebral organoids model human brain development and microcephaly","volume":"501","author":"Lancaster","year":"2013","journal-title":"Nature"},{"key":"10.1016\/j.stemcr.2020.08.006_bib16","doi-asserted-by":"crossref","first-page":"1213","DOI":"10.2217\/epi.15.70","article-title":"Epigenetic regulation of UBE3A and roles in human neurodevelopmental disorders","volume":"7","author":"LaSalle","year":"2015","journal-title":"Epigenomics"},{"key":"10.1016\/j.stemcr.2020.08.006_bib17","doi-asserted-by":"crossref","first-page":"45","DOI":"10.1186\/s13229-018-0228-2","article-title":"Characterization and structure-activity relationships of indenoisoquinoline-derived topoisomerase i inhibitors in unsilencing the dormant Ube3a gene associated with Angelman syndrome","volume":"9","author":"Lee","year":"2018","journal-title":"Mol. Autism"},{"key":"10.1016\/j.stemcr.2020.08.006_bib18","doi-asserted-by":"crossref","first-page":"476","DOI":"10.3389\/fnmol.2018.00476","article-title":"UBE3A: an E3 ubiquitin ligase with genome-wide impact in neurodevelopmental disease","volume":"11","author":"Lopez","year":"2019","journal-title":"Front. Mol. Neurosci."},{"key":"10.1016\/j.stemcr.2020.08.006_bib19","doi-asserted-by":"crossref","first-page":"48","DOI":"10.1038\/nature22047","article-title":"Cell diversity and network dynamics in photosensitive human brain organoids","volume":"545","author":"Quadrato","year":"2017","journal-title":"Nature"},{"key":"10.1016\/j.stemcr.2020.08.006_bib20","doi-asserted-by":"crossref","first-page":"8011","DOI":"10.1523\/JNEUROSCI.0083-18.2018","article-title":"Adult Ube3a gene reinstatement restores the electrophysiological deficits of prefrontal cortex layer 5 neurons in a mouse model of angelman syndrome","volume":"38","author":"Rotaru","year":"2018","journal-title":"J.\u00a0Neurosci."},{"key":"10.1016\/j.stemcr.2020.08.006_bib21","doi-asserted-by":"crossref","first-page":"588","DOI":"10.1093\/cercor\/bhq125","article-title":"Neocortical layer formation of human developing brains and lissencephalies: consideration of layer-specific marker expression","volume":"21","author":"Saito","year":"2011","journal-title":"Cereb. Cortex"},{"key":"10.1016\/j.stemcr.2020.08.006_bib22","doi-asserted-by":"crossref","first-page":"2069","DOI":"10.1172\/JCI80554","article-title":"Ube3a reinstatement identifies distinct developmental windows in a murine Angelman syndrome model","volume":"125","author":"Silva-Santos","year":"2015","journal-title":"J.\u00a0Clin. Invest."},{"key":"10.1016\/j.stemcr.2020.08.006_bib26","doi-asserted-by":"crossref","DOI":"10.1093\/hmg\/ddaa191","article-title":"Abundance and localization of human UBE3A protein isoforms","author":"Sirois","year":"2020","journal-title":"Hum. Mol. Genet."},{"key":"10.1016\/j.stemcr.2020.08.006_bib23","doi-asserted-by":"crossref","first-page":"23","DOI":"10.1186\/s13229-019-0277-1","article-title":"Delayed loss of UBE3A reduces the expression of Angelman syndrome-associated phenotypes","volume":"10","author":"Sonzogni","year":"2019","journal-title":"Mol. Autism"},{"key":"10.1016\/j.stemcr.2020.08.006_bib24","doi-asserted-by":"crossref","first-page":"30792","DOI":"10.1038\/srep30792","article-title":"Angelman syndrome-derived neurons display late onset of paternal UBE3A silencing","volume":"6","author":"Stanurova","year":"2016","journal-title":"Sci. Rep."},{"key":"10.1016\/j.stemcr.2020.08.006_bib25","doi-asserted-by":"crossref","first-page":"1486","DOI":"10.1126\/science.aav5386","article-title":"Potassium channel dysfunction in human neuronal models of Angelman syndrome","volume":"366","author":"Sun","year":"2019","journal-title":"Science"}],"container-title":["Stem Cell Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S2213671120303374?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S2213671120303374?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"}],"deposited":{"date-parts":[[2025,11,1]],"date-time":"2025-11-01T09:45:22Z","timestamp":1761990322000},"score":1,"resource":{"primary":{"URL":"https:\/\/linkinghub.elsevier.com\/retrieve\/pii\/S2213671120303374"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2020,10]]},"references-count":26,"journal-issue":{"issue":"4","published-print":{"date-parts":[[2020,10]]}},"alternative-id":["S2213671120303374"],"URL":"https:\/\/doi.org\/10.1016\/j.stemcr.2020.08.006","relation":{"has-preprint":[{"id-type":"doi","id":"10.1101\/742213","asserted-by":"object"}]},"ISSN":["2213-6711"],"issn-type":[{"value":"2213-6711","type":"print"}],"subject":[],"published":{"date-parts":[[2020,10]]},"assertion":[{"value":"Elsevier","name":"publisher","label":"This article is maintained by"},{"value":"Human Cerebral Organoids Reveal Early Spatiotemporal Dynamics and Pharmacological Responses of UBE3A","name":"articletitle","label":"Article Title"},{"value":"Stem Cell Reports","name":"journaltitle","label":"Journal Title"},{"value":"https:\/\/doi.org\/10.1016\/j.stemcr.2020.08.006","name":"articlelink","label":"CrossRef DOI link to publisher maintained version"},{"value":"article","name":"content_type","label":"Content Type"},{"value":"\u00a9 2020 The Author(s).","name":"copyright","label":"Copyright"}]}}