{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,11,3]],"date-time":"2026-11-03T18:53:55Z","timestamp":1793732035486,"version":"4.6.1"},"reference-count":21,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2024,5,31]],"date-time":"2024-05-31T00:00:00Z","timestamp":1717113600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100000156","name":"Fonds de Recherche du Qu\u00e9bec - Sant\u00e9","doi-asserted-by":"publisher","award":["282228"],"award-info":[{"award-number":["282228"]}],"id":[{"id":"10.13039\/501100000156","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100000156","name":"Fonds de Recherche du Qu\u00e9bec - Sant\u00e9","doi-asserted-by":"publisher","award":["295639"],"award-info":[{"award-number":["295639"]}],"id":[{"id":"10.13039\/501100000156","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100000156","name":"Fonds de Recherche du Qu\u00e9bec - Sant\u00e9","doi-asserted-by":"publisher","award":["309121"],"award-info":[{"award-number":["309121"]}],"id":[{"id":"10.13039\/501100000156","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Epileptic Disorders"],"published-print":{"date-parts":[[2024,10]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Objective<\/jats:title><jats:p>This retrospective study aimed to assess the efficacy and tolerability of sulthiame as an add\u2010on treatment in children with pharmacoresistant epilepsies.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>All patients with epilepsy who received sulthiame at Montreal Children's Hospital over an 11\u2010year period were included. Medical charts were reviewed, and extracted data included patient age and sex, seizure types, epilepsy syndrome, electroencephalography (EEG) reports, brain imaging reports, antiseizure treatments trialed, starting and final dose of sulthiame, duration of sulthiame treatment, adverse events attributed to sulthiame, and seizure frequency before and after sulthiame treatment. EEG studies were also analyzed and spike\u2013wave index (SWI) in the first 10\u2009min of sleep was calculated.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Sixteen patients were included, all of whom had pharmacoresistant epilepsies (mean of 9.9 trials of other antiseizure treatments). Six had genetic diagnoses, four had in utero\/perinatal acquired brain injury, one had a suspected focal cortical dysplasia, and five were idiopathic. Ten patients had developmental and epileptic encephalopathy with spike\u2013wave activation in sleep, three had Lennox\u2013Gastaut syndrome, and one each had sleep\u2010related hyperkinetic epilepsy, self\u2010limited epilepsy with centrotemporal spikes, and mixed generalized and multifocal epilepsy. Of the 12 patients with uncontrolled seizures at the time of sulthiame initiation, 4 had improvement in seizure frequency, including 2 who became seizure free. Eight patients had EEG data available that allowed calculation of sleep SWI; from this group, SWI decreased from 81.1% +\/\u2212 17.6% to 45.1% +\/\u2212 36.5% (<jats:italic>p<\/jats:italic>\u2009=\u2009.007). The most common adverse events reported were somnolence\/drowsiness, aggression, and increased seizure frequency. Of the patients with genetic etiologies, significant positive responses were seen in patients with pathogenic variants in <jats:italic>NDUFS1<\/jats:italic> and <jats:italic>SATB1<\/jats:italic>.<\/jats:p><\/jats:sec><jats:sec><jats:title>Significance<\/jats:title><jats:p>These data demonstrate the therapeutic potential of sulthiame, even in patients with highly pharmacoresistant epilepsy. Improvements may be seen in both seizure frequency and sleep SWI.<\/jats:p><\/jats:sec>","DOI":"10.1002\/epd2.20250","type":"journal-article","created":{"date-parts":[[2024,5,31]],"date-time":"2024-05-31T09:25:18Z","timestamp":1717147518000},"page":"591-599","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":3,"title":["Sulthiame use in children with pharmacoresistant epilepsies: A retrospective study"],"prefix":"10.1002","volume":"26","author":[{"given":"Alexandra","family":"Lalibert\u00e9","sequence":"first","affiliation":[{"name":"Faculty of Medicine and Health Sciences McGill University  Montreal Quebec Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Saoussen","family":"Berrahmoune","sequence":"additional","affiliation":[{"name":"Child Health and Human Development Program Research Institute of the McGill University Health Centre  Montreal Quebec Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kenneth A.","family":"Myers","sequence":"additional","affiliation":[{"name":"Child Health and Human Development Program Research Institute of the McGill University Health Centre  Montreal Quebec Canada"},{"name":"Division of Neurology, Department of Pediatrics, Montreal Children's Hospital McGill University Health Centre  Montreal Quebec Canada"},{"name":"Department of Neurology and Neurosurgery McGill University Health Centre  Montreal Quebec Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2024,5,31]]},"reference":[{"key":"e_1_2_8_2_1","first-page":"2199","article-title":"On the role of the butanesultam derivative Ospolot in the treatment of cerebral epilepsy","volume":"85","author":"Fluegel F","year":"1960","journal-title":"Dtsch Med Wochenschr"},{"key":"e_1_2_8_3_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0006-8993(01)02262-4"},{"key":"e_1_2_8_4_1","doi-asserted-by":"publisher","DOI":"10.1055\/s-0028-1112717"},{"key":"e_1_2_8_5_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1528-1157.2003.51001.x"},{"key":"e_1_2_8_6_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1528-1157.2000.tb04606.x"},{"key":"e_1_2_8_7_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1469-8749.1988.tb04809.x"},{"key":"e_1_2_8_8_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.seizure.2018.09.019"},{"key":"e_1_2_8_9_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.yebeh.2022.108702"},{"key":"e_1_2_8_10_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1528-1167.2012.03458.x"},{"key":"e_1_2_8_11_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.yebeh.2021.107793"},{"key":"e_1_2_8_12_1","doi-asserted-by":"publisher","DOI":"10.1111\/epi.13709"},{"key":"e_1_2_8_13_1","doi-asserted-by":"publisher","DOI":"10.1111\/epi.13670"},{"key":"e_1_2_8_14_1","doi-asserted-by":"publisher","DOI":"10.1111\/epi.17241"},{"key":"e_1_2_8_15_1","doi-asserted-by":"publisher","DOI":"10.1542\/peds.2019-0599"},{"key":"e_1_2_8_16_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00415-023-12174-3"},{"key":"e_1_2_8_17_1","unstructured":"McNamaraN ArndtD BaumerF BernardoD BhatiaS ChuC et\u00a0al.Spike wave index calculation in electrical status epilepticus of sleep: first step towards standardized practice in American Epilepsy Society annual meeting.2023. 2023: Orlando Florida.https:\/\/aesnet.org\/abstractslisting\/spike\u2010wave\u2010index\u2010calculation\u2010in\u2010electrical\u2010status\u2010epilepticus\u2010of\u2010sleep\u2010first\u2010step\u2010towards\u2010standardized\u2010practice"},{"key":"e_1_2_8_18_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.pediatrneurol.2012.06.016"},{"key":"e_1_2_8_19_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.mito.2015.01.003"},{"key":"e_1_2_8_20_1","doi-asserted-by":"publisher","DOI":"10.1186\/s13023-021-01690-y"},{"key":"e_1_2_8_21_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajhg.2021.01.007"},{"key":"e_1_2_8_22_1","doi-asserted-by":"publisher","DOI":"10.3389\/fped.2022.931667"}],"container-title":["Epileptic Disorders"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/epd2.20250","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,10,12]],"date-time":"2024-10-12T05:38:55Z","timestamp":1728711535000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/epd2.20250"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,5,31]]},"references-count":21,"journal-issue":{"issue":"5","published-print":{"date-parts":[[2024,10]]}},"alternative-id":["10.1002\/epd2.20250"],"URL":"https:\/\/doi.org\/10.1002\/epd2.20250","archive":["Portico"],"relation":{},"ISSN":["1294-9361","1950-6945"],"issn-type":[{"value":"1294-9361","type":"print"},{"value":"1950-6945","type":"electronic"}],"subject":[],"published":{"date-parts":[[2024,5,31]]},"assertion":[{"value":"2024-04-16","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2024-05-21","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2024-05-31","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}