{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,28]],"date-time":"2026-08-28T18:49:59Z","timestamp":1787942999662,"version":"build-2784847793"},"reference-count":26,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2021,3,29]],"date-time":"2021-03-29T00:00:00Z","timestamp":1616976000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/501100004963","name":"Seventh Framework Programme","doi-asserted-by":"publisher","award":["602391"],"award-info":[{"award-number":["602391"]}],"id":[{"id":"10.13039\/501100004963","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Epilepsia"],"published-print":{"date-parts":[[2021,5]]},"abstract":"<jats:title>Summary<\/jats:title><jats:sec><jats:title>Objective<\/jats:title><jats:p>To study the association between timing and characteristics of the first electroencephalography (EEG) with epileptiform discharges (ED\u2010EEG) and epilepsy and neurodevelopment at 24\u00a0months in infants with tuberous sclerosis complex (TSC).<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Patients enrolled in the prospective Epileptogenesis in a genetic model of epilepsy \u2013 Tuberous sclerosis complex (EPISTOP) trial, had serial EEG monitoring until the age of 24\u00a0months. The timing and characteristics of the first ED\u2010EEG were studied in relation to clinical outcome. Epilepsy\u2010related outcomes were analyzed separately in a conventionally followed group (initiation of vigabatrin after seizure onset) and a preventive group (initiation of vigabatrin before seizures, but after appearance of interictal epileptiform discharges [IEDs]).<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Eighty\u2010three infants with TSC were enrolled at a median age of 28\u00a0days (interquartile range [IQR] 14\u201354). Seventy\u2010nine of 83 patients (95%) developed epileptiform discharges at a median age of 77\u00a0days (IQR 23\u2013111). Patients with a pathogenic <jats:italic>TSC2<\/jats:italic> variant were significantly younger (<jats:italic>P<\/jats:italic>\u2010value .009) at first ED\u2010EEG and more frequently had multifocal IED (<jats:italic>P<\/jats:italic>\u2010value .042) than patients with a pathogenic <jats:italic>TSC1<\/jats:italic> variant. A younger age at first ED\u2010EEG was significantly associated with lower cognitive (<jats:italic>P<\/jats:italic>\u2010value .010), language (<jats:italic>P<\/jats:italic>\u2010value .001), and motor (<jats:italic>P<\/jats:italic>\u2010value .013) developmental quotients at 24\u00a0months.<\/jats:p><jats:p>In the conventional group, 48 of 60 developed seizures. In this group, the presence of focal slowing on the first ED\u2010EEG was predictive of earlier seizure onset (<jats:italic>P<\/jats:italic>\u2010value .030). Earlier recording of epileptiform discharges (<jats:italic>P<\/jats:italic>\u2010value .019), especially when multifocal (<jats:italic>P<\/jats:italic>\u2010value .026) was associated with higher risk of drug\u2010resistant epilepsy.<\/jats:p><jats:p>In the preventive group, timing, distribution of IED, or focal slowing, was not associated with the epilepsy outcomes. However, when multifocal IEDs were present on the first ED\u2010EEG, preventive treatment delayed the onset of seizures significantly (<jats:italic>P<\/jats:italic>\u2010value &lt;.001).<\/jats:p><\/jats:sec><jats:sec><jats:title>Significance<\/jats:title><jats:p>Early EEG findings help to identify TSC infants at risk of severe epilepsy and neurodevelopmental delay and those who may benefit from preventive treatment with vigabatrin.<\/jats:p><\/jats:sec>","DOI":"10.1111\/epi.16892","type":"journal-article","created":{"date-parts":[[2021,3,29]],"date-time":"2021-03-29T07:12:30Z","timestamp":1617001950000},"page":"1208-1219","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":45,"title":["Early epileptiform EEG activity in infants with tuberous sclerosis complex predicts epilepsy and neurodevelopmental outcomes"],"prefix":"10.1111","volume":"62","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-8555-7822","authenticated-orcid":false,"given":"Jessie","family":"De Ridder","sequence":"first","affiliation":[{"name":"Department of Development and Regeneration Section Pediatric Neurology Catholic University of Leuven (KU Leuven) Leuven Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Birgit","family":"Verhelle","sequence":"additional","affiliation":[{"name":"Department of Development and Regeneration Section Pediatric Neurology Catholic University of Leuven (KU Leuven) Leuven Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jan","family":"Vervisch","sequence":"additional","affiliation":[{"name":"Department of Development and Regeneration Section Pediatric Neurology Catholic University of Leuven (KU Leuven) Leuven Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Katrien","family":"Lemmens","sequence":"additional","affiliation":[{"name":"Department of Development and Regeneration Section Pediatric Neurology Catholic University of Leuven (KU Leuven) Leuven Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Katarzyna","family":"Kotulska","sequence":"additional","affiliation":[{"name":"Department of Neurology and Epileptology The Children's Memorial Health Institute Warsaw Poland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Romina","family":"Moavero","sequence":"additional","affiliation":[{"name":"Child Neurology and Psychiatry Unit Systems Medicine Department Tor Vergata University Rome Italy"},{"name":"Child Neurology Unit, Neuroscience and Neurorehabilitation Department \u201cBambino Ges\u00f9\u201d Children\u2019s Hospital Rome Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Paolo","family":"Curatolo","sequence":"additional","affiliation":[{"name":"Child Neurology and Psychiatry Unit Systems Medicine Department Tor Vergata University Rome Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bernhard","family":"Weschke","sequence":"additional","affiliation":[{"name":"Department of Child Neurology Charit\u00e9 University Medicine Berlin Berlin Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kate","family":"Riney","sequence":"additional","affiliation":[{"name":"Neuroscience Unit Queensland Children\u2019s Hospital Brisbane Australia"},{"name":"University of Queensland School of Clinical Medicine Brisbane Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7691-8158","authenticated-orcid":false,"given":"Martha","family":"Feucht","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Medical University Vienna Vienna Austria"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pavel","family":"Krsek","sequence":"additional","affiliation":[{"name":"Department of Paediatric Neurology Second Faculty of Medicine Motol University Hospital Charles University Prague Czech Republic"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rima","family":"Nabbout","sequence":"additional","affiliation":[{"name":"Department of Pediatric Neurology Reference Centre for Rare Epilepsies Necker\u2010 Enfants Malades Hospital Imagine Institute, INSERM U1163 University Paris Descartes Paris France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anna C.","family":"Jansen","sequence":"additional","affiliation":[{"name":"Pediatric Neurology Unit University Hospital Brussel Brussels Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Konrad","family":"Wojdan","sequence":"additional","affiliation":[{"name":"Transition Technologies Warsaw Poland"},{"name":"Institute of Heat Engineering Warsaw University and Technology Warsaw Poland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dorota","family":"Domanska\u2010Pakie\u0142a","sequence":"additional","affiliation":[{"name":"Department of Neurology and Epileptology The Children's Memorial Health Institute Warsaw Poland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Magdalena","family":"Kaczorowska\u2010Frontczak","sequence":"additional","affiliation":[{"name":"Department of Neurology and Epileptology The Children's Memorial Health Institute Warsaw Poland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christoph","family":"Hertzberg","sequence":"additional","affiliation":[{"name":"Diagnose und Behandlungszentrum f\u00fcr Kinder und Jugendliche Vivantes Klinikum Neuk\u00f6ln Berlin Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Cyrille H.","family":"Ferrier","sequence":"additional","affiliation":[{"name":"Department of Child Neurology, Brain Centre University Medical Centre Utrecht Utrecht The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sharon","family":"Samueli","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Medical University Vienna Vienna Austria"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8448-2833","authenticated-orcid":false,"given":"Barbora","family":"Benova","sequence":"additional","affiliation":[{"name":"Department of Paediatric Neurology Second Faculty of Medicine Motol University Hospital Charles University Prague Czech Republic"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Eleonora","family":"Aronica","sequence":"additional","affiliation":[{"name":"Department of (Neuro)Pathology Amsterdam University Medical Centres (UMC) University of Amsterdam Amsterdam The 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