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(Abst. 1.218), 2019

Preventive Antiepileptic Treatment Reduces Risk of Intellectual Disability and Epilepsy Severity in Tuberous Sclerosis Complex: Long-Term, Prospective, Controlled Trial 
Authors: Sergiusz Jozwiak, Warsaw Medical University; Monika Slowinska, Medical University Warsaw; Julita Borkowska, The Children's Memorial Health Institute; Krzysztof Sadowski, The Children's Memorial Health Institute; Barbara Lojszczyk, The Children's Memorial Health Institute; Dorota Domańska-Pakieła, The Children's Memorial Health Institute; Dariusz Chmielewski, The Children's Memorial Health Institute; Magdalena Kaczorowska-Frontczak, The Children's Memorial Health Institute; Jagoda Głowacka, Transition Technologies; Kamil Sijko, Transition Technologies; Katarzyna Kotulska-Jóźwiak, The Children's Memorial Health Institute
Content: Rationale: Refractory drug-resistant epilepsy is the main risk factor for future intellectual disability in patients with tuberous sclerosis complex. Clinical epileptic seizures are often precededby electroencephalographic changes that provide an opportunity for preventive treatment. The study aimed to evaluate neuropsychological and epilepsy outcomes at school age in tuberous sclerosis complex children who received preventive antiepileptic treatment in infancy.  Methods: A pilot non-randomized clinical trial was performed for 14 infants diagnosed with tuberous sclerosis complex in whom serial electroencephalographic recordings were performed, and preventive treatment with vigabatrin initiated when active epileptic discharges were detected. An age-matched control group consisted of 31 tuberous sclerosis complex infants in whom treatment with vigabatrin was given only after onsetof clinical seizures. Vigabatrin was startedwhen epileptic discharges were detected (preventive group) or after clinical seizures (standard group). Results of clinical assessment concerning epilepsy and cognitive outcome were analysed. They included neuropsychological tests, detailed clinical follow-up on epilepsy status and use of antiepileptic drugs.  Results: All patients in the preventive group (n=14) and 25 of 31 patients in the standard treatment group were followed through minimum age 5, median 8.8 and 8.0 years in the preventive and standard groups, respectively. The median IQ was 94 for the preventive group in comparison to 46 for the standard group (p<0.03). Seven of 14 patients (50%) in the preventive group never had a clinical seizure in comparison to 1/25 patients (5%) in the standard treatment group (p=0.001). The likelihood of successful antiepileptic drug withdrawal was also higher in the preventive group (6/14, 55% vs 4/24, 17%, p<0.03). Conclusions: This study provides evidence that preventive antiepileptic treatment in infants with tuberous sclerosis complex improves long-term epilepsy control and cognitive outcome at school age. Funding: 7th Framework Programme of European Commission within the Large-scale Integrating Project EPISTOP, Polish Ministerial funds for science, the EPIMARKER grant of the Polish National Center for Research and Development.