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.
Highlights
Tuberous sclerosis (TS) is the leading cause of genetic epilepsy worldwide
In the last 15 years, a greater proportion of younger children aslo underwent resective surgery compared to older cohorts
The search strategy was limited to humans and incorporated the following terms: “tuberous sclerosis,” “epilepsy surgery,” “seizure outcomes,” “Engel classification,” and “predictors.” The inclusion criteria for studies were (1) case-controlled or cohort studies; (2) consecutive participants; (3) at least 90% of participants be under the age of 19 at time of surgery; (4) at least 90% of subjects have TS; (5) at least 90% of subjects have undergone resective epilepsy surgery; and (6) seizure outcomes are reported
Summary
We evaluate changes in seizure outcomes following resective epilepsy surgery in children with TS over time. Multivariate logistic regression was used to determine significant associations between seizure outcomes and time of surgery. 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). A trend towards improved seizure outcomes following resective surgery for TS was observed from 1966 to present on multivariate analysis. The condition is characterized by inactivating mutations in TSC1, encoding hamartin, or TSC2, encoding tuberin [1] These lead to downstream hyperactivation of the mammalian target of rapamycin (mTOR) pathway and associated kinase signalling cascades, resulting in abnormal cortical development and epilepsy in the majority of affected children. Seizure frequency and epilepsy severity have been previously related to the extent of cognitive impairment in children with TS [5]
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