{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,14]],"date-time":"2026-10-14T22:20:34Z","timestamp":1792016434963,"version":"4.4.0"},"reference-count":24,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2023,9,25]],"date-time":"2023-09-25T00:00:00Z","timestamp":1695600000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"funder":[{"DOI":"10.13039\/100000065","name":"National Institute of Neurological Disorders and Stroke","doi-asserted-by":"publisher","award":["NCT028494571"],"award-info":[{"award-number":["NCT028494571"]}],"id":[{"id":"10.13039\/100000065","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100001771","name":"Tuberous Sclerosis Alliance","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100001771","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100010621","name":"Pediatric Epilepsy Research Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100010621","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Annals of Neurology"],"published-print":{"date-parts":[[2024,1]]},"abstract":"<jats:sec>\n                    <jats:title>Objective<\/jats:title>\n                    <jats:p>This study was undertaken to test the hypothesis that early vigabatrin treatment in tuberous sclerosis complex (TSC) infants improves neurocognitive outcome at 24\u2009months of age.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>A phase IIb multicenter randomized double\u2010blind placebo\u2010controlled trial was conducted of vigabatrin at first epileptiform electroencephalogram (EEG) versus vigabatrin at seizure onset in infants with TSC. Primary outcome was Bayley Scales of Infant and Toddler Development, Third Edition (Bayley\u2010III) cognitive assessment score at 24\u2009months. Secondary outcomes were prevalence of drug\u2010resistant epilepsy, additional developmental outcomes, and safety of vigabatrin.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Of 84 infants enrolled, 12 were screen failures, 4 went straight to open label vigabatrin, and 12 were not randomized (normal EEG throughout). Fifty\u2010six were randomized to early vigabatrin (n\u2009=\u200929) or placebo (n\u2009=\u200927). Nineteen of 27 in the placebo arm transitioned to open label vigabatrin, with a median delay of 44\u2009days after randomization. Bayley\u2010III cognitive composite scores at 24\u2009months were similar for participants randomized to vigabatrin or placebo. Additionally, no significant differences were found between groups in overall epilepsy incidence and drug\u2010resistant epilepsy at 24\u2009months, time to first seizure after randomization, and secondary developmental outcomes. Incidence of infantile spasms was lower and time to spasms after randomization was later in the vigabatrin group. Adverse events were similar across groups.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Interpretation<\/jats:title>\n                    <jats:p>Preventative treatment with vigabatrin based on EEG epileptiform activity prior to seizure onset does not improve neurocognitive outcome at 24\u2009months in TSC children, nor does it delay onset or lower the incidence of focal seizures and drug\u2010resistant epilepsy at 24\u2009months. Preventative vigabatrin was associated with later time to onset and lower incidence of infantile spasms. ANN NEUROL 2024;95:15\u201326<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/ana.26778","type":"journal-article","created":{"date-parts":[[2023,8,28]],"date-time":"2023-08-28T06:42:20Z","timestamp":1693204940000},"page":"15-26","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":68,"title":["Early Treatment with Vigabatrin Does Not Decrease Focal Seizures or Improve Cognition in Tuberous Sclerosis Complex: The\n                    <scp>PREVeNT<\/scp>\n                    Trial"],"prefix":"10.1002","volume":"95","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-1264-3428","authenticated-orcid":false,"given":"Elizabeth Martina","family":"Bebin","sequence":"first","affiliation":[{"name":"Department of Neurology University of Alabama at Birmingham  Birmingham AL USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6725-2814","authenticated-orcid":false,"given":"Jurriaan M.","family":"Peters","sequence":"additional","affiliation":[{"name":"Department of Neurology, Boston Children's Hospital Harvard Medical School  Boston MA USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Brenda E.","family":"Porter","sequence":"additional","affiliation":[{"name":"Department of Neurology Stanford University  Stanford CA USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tarrant O.","family":"McPherson","sequence":"additional","affiliation":[{"name":"Department of Biostatistics and Bioinformatics Emory University  Atlanta GA USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sarah","family":"O'Kelley","sequence":"additional","affiliation":[{"name":"Department of Psychology University of Alabama at Birmingham  Birmingham AL USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7044-2953","authenticated-orcid":false,"given":"Mustafa","family":"Sahin","sequence":"additional","affiliation":[{"name":"Department of Neurology, Boston Children's Hospital Harvard Medical School  Boston MA USA"},{"name":"Rosamund Stone Zander Translational Neuroscience Center, Boston Children's Hospital, Harvard Medical School Harvard University  Boston MA USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Katherine S.","family":"Taub","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Children's Hospital of Philadelphia  Philadelphia PA USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rajsekar","family":"Rajaraman","sequence":"additional","affiliation":[{"name":"Department of Pediatrics and Psychiatry and Biobehavioral Sciences University of California, Los Angeles  Los Angeles CA USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Stephanie C.","family":"Randle","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Division Pediatric Neurology and Epilepsy Seattle Children's Hospital  Seattle WA USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"William M.","family":"McClintock","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Division of Neurology Children's National Medical Center  Washington DC USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mary Kay","family":"Koenig","sequence":"additional","affiliation":[{"name":"Department of Pediatrics McGovern Medical School at University of Texas Health Science Center at Houston and Children's Memorial Hermann Hospital  Houston TX USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mike D.","family":"Frost","sequence":"additional","affiliation":[{"name":"Minnesota Epilepsy Group, P.A. 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