Abstracts

Rationale: Preventive treatment with vigabatrin (VGB) improves epilepsy outcome and neurodevelopment in patients with tuberous sclerosis complex (TSC). Studies have shown the positive effect of pharmacotherapy during the treatment. However, there is insufficient knowledge on the longstanding effects after successful discontinuation of the treatment. This study evaluates the long-term outcomes of preventive treatment with vigabatrin after pharmacotherapy cessation. _x000D_
Methods: We performed a retrospective review of children diagnosed with TSC and treated preventively in the first 2 years of age with vigabatrin. The data were collected from two clinical centers: the Department of Neurology and Epileptology, The Children’s Memorial Health Institute, and the Department of Child Neurology, the Medical University of Warsaw in Warsaw, Poland. The inclusion criteria were: (1) patients with TSC who obtained preventive initial treatment with vigabatrin within 24 months of age, (2) discontinuation of the treatment with all antiepileptic drugs (AEDs), (3) follow-up after treatment removal at least 12 months. The exclusion criteria included: 1(1) all patients who required any antiepileptic treatment, had active epilepsy after preventive treatment, and (2) whose follow-up after VGB discontinuation was less then 12 months. _x000D_
Results: Seventeen patients were included in the study, 8 females and 9 males. All participants were treated preventively with vigabatrin due to paroxysmal epileptiform activity on EEG. Vigabatrin was introduced at the median age of 175 days (8-766 days). The median treatment duration was 2.9 years (1.7-6.7 years) in all patients. During the preventive treatment period with VGB 15 out of 17 (88.2%) children remained seizure-free. In 2 infants, despite of the treatment, appeared seizures but dissolved after drug modification. In those who remained seizure free till 24 months of age or had controlled seizures at least 2 years, the decision of drug withdrawal has been taken (17 children). After discontinuation of antiepileptic treatment patients underwent control visits. Follow-up after drug withdrawal was 2.5 years (median) (1.2-12.3 years). During this follow-up epilepsy was reported in 1 out of 17 (5.9%) children. Sixteen out of 17 (94.1%) remained seizure-free and treatment-free._x000D_
Conclusions: In the vast majority of patients with TSC seizure control continues after discontinuation of the preventive treatment with vigabatrin during median follow-up. However, the median time of observation in the study was 2.5 years and results require further studies. _x000D_
Funding: None