{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,21]],"date-time":"2026-07-21T02:35:47Z","timestamp":1784601347868,"version":"3.55.0"},"reference-count":20,"publisher":"Wiley","issue":"3","license":[{"start":{"date-parts":[[2026,4,16]],"date-time":"2026-04-16T00:00:00Z","timestamp":1776297600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2026,4,16]],"date-time":"2026-04-16T00:00:00Z","timestamp":1776297600000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Epilepsia Open"],"published-print":{"date-parts":[[2026,6]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:label\/>\n                    <jats:p>This multicenter retrospective study evaluated the effectiveness and safety of highly purified cannabidiol (CBD) in 22 patients with 15q11.2\u2010q13.1 duplication or deletion syndromes (15q\u2010DDS), including 12 with 15q duplication syndrome (dup15q) and 10 with Angelman syndrome (AS). Median (interquartile range [IQR]) age at CBD initiation was 14.5 (10\u201322.5) years, with a median (IQR) follow\u2010up of 21 (14\u201333) months. All dup15q and two AS patients presented with a Lennox\u2013Gastaut phenotype. At last observation, mean seizure reduction was 55.7% (95% confidence interval 38.7\u201372.7), with 63.6% patients achieving \u226550% reduction, 40.9% achieving \u226575% reduction, and 18.2% achieving seizure freedom. Tonic seizures in dup15q and myoclonic seizures in AS showed the most notable reductions. EEG improvement was observed in 7\/16 patients, with marked improvement observed in two dup15q patients. Clinical improvement on the Clinical Global Impression\u2010Improvement scale was reported in 72.7%, alongside nonseizure benefits such as improved sleep, behavior, and attention in a subset of patients. CBD was well tolerated; no patient discontinued CBD due to side effects alone, and retention at last visit was 81.8%. These findings suggest that CBD may provide clinically meaningful benefit in patients with 15q\u2010DDS, including seizure reduction and improvements in sleep, behavior, and attention in selected cases.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Plain Language Summary<\/jats:title>\n                    <jats:p>Epilepsy secondary to 15q11.2\u2010q13.1 duplication or deletion syndromes (15q\u2010DDS) is often severe, making daily life difficult for patients and their families. In this study, treatment with highly purified cannabidiol (CBD) reduced seizures in many patients with 15q\u2010DDS. CBD was generally well tolerated, and caregivers also reported improvements in sleep, behavior, and attention in a number of cases. Overall, these findings suggest that CBD may be a helpful treatment option for people with 15q\u2010DDS.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/epi4.70241","type":"journal-article","created":{"date-parts":[[2026,4,17]],"date-time":"2026-04-17T04:23:00Z","timestamp":1776399780000},"page":"1046-1053","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":2,"title":["Real\u2010world effectiveness of highly purified cannabidiol in epilepsy associated with 15q11.2\u2010q13.1 duplication and deletion syndromes: A multicenter study"],"prefix":"10.1002","volume":"11","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-3906-555X","authenticated-orcid":false,"given":"Emanuele","family":"Cerulli Irelli","sequence":"first","affiliation":[{"name":"Department of Human Neurosciences Sapienza University  Rome 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Switzerland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Domenica","family":"Battaglia","sequence":"additional","affiliation":[{"name":"Pediatric Neurology and Psychiatric Unit Fondazione Policlinico Universitario Agostino Gemelli IRCCS  Rome Italy"},{"name":"Department of Health Science and Public Health Universit\u00e0 Cattolica del Sacro Cuore  Rome Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Irene","family":"Bagnasco","sequence":"additional","affiliation":[{"name":"Child Neuropsychiatry Unit, Epilepsy Center for Children Martini Hospital  Turin Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Erica","family":"Cognolato","sequence":"additional","affiliation":[{"name":"Child Neuropsychiatry Unit, Epilepsy Center for Children Martini Hospital  Turin 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 Conegliano Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Paolo","family":"Bonanni","sequence":"additional","affiliation":[{"name":"Scientific Institute IRCCS E.Medea, Epilepsy and Clinical Neurophysiology Unit  Conegliano Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2444-8761","authenticated-orcid":false,"given":"Francesca F.","family":"Operto","sequence":"additional","affiliation":[{"name":"Department of Science of Health, School of Medicine University of Catanzaro  Catanzaro Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1890-5409","authenticated-orcid":false,"given":"Carlo","family":"Di Bonaventura","sequence":"additional","affiliation":[{"name":"Department of Human Neurosciences Sapienza University  Rome 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