Changing global trends in seizure outcomes following resective surgery for tuberous sclerosis in children with medically intractable epilepsy

Abstract

Introduction. Tuberous sclerosis (TS) is the leading cause of genetic epilepsy worldwide. Here, we evaluate changes in seizure outcomes following resective epilepsy surgery in children with TS over time. Methods. A systematic review of the literature was performed to identify studies reporting seizure outcomes following resective epilepsy surgery in children with TS. Using an individual participant meta-analysis approach, seizure outcomes and associated covariates were combined. Multivariate logistic regression was used to determine significant associations between seizure outcomes and time of surgery. Results. Twenty studies from 1966 to present, yielding 186 participants, met the inclusion criteria for the study. On univariate analysis, there was a significant improvement in seizure outcomes in children who underwent resective epilepsy surgery within the last 15 years compared to older cohorts (chi-square 4.1; P = 0.043). On multivariate analysis, adjusting for length of followup, this trend was not significant (OR 0.52; 95% CI 0.23-1.17; P = 0.11). In the last 15 years, a greater proportion of younger children also underwent resective surgery compared to older cohorts (OR 0.93; 95% CI 0.89-0.97; P < 0.01). Conclusions. A trend towards improved seizure outcomes following resective surgery for TS was observed from 1966 to present on multivariate analysis.

Figure 1

Proportion of children with tuberous sclerosis achieving favourable seizure outcome (Engel I-II) following resection surgery in recent and older cohorts. Recent cohorts demonstrate significantly improved seizure outcomes on univariate analysis.