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Association of earlier surgery with improved postoperative language development in children with tuberous sclerosis
Sina Sadeghzadeh1, Thomas M Johnstone1, Jurriaan M Peters2
1Departments of1Neurosurgery and.
Insights
Earlier epilepsy surgery in children with Tuberous Sclerosis Complex (TSC) is linked to better language outcomes. Shorter epilepsy duration before surgery also positively impacts neurocognitive development, suggesting timely intervention is key.
Area of Science:
- Neuroscience
- Pediatric Neurology
- Developmental Psychology
Background:
- Tuberous Sclerosis Complex (TSC) is a genetic disorder that can cause epilepsy and impact neurodevelopment.
- Drug-refractory epilepsy in children with TSC often necessitates surgical intervention.
- The optimal timing for epilepsy surgery in this population remains an area of investigation.
Purpose of the Study:
- To assess the relationship between the timing of epilepsy surgery and postoperative neurocognitive outcomes in children with TSC.
- To investigate how age at surgery and duration of epilepsy influence language and adaptive behavior development.
- To analyze these effects in the context of seizure control post-surgery.
Main Methods:
- Retrospective analysis of 27 patients with drug-refractory epilepsy from the Tuberous Sclerosis Complex (TSC) Autism Center of Excellence Research Network (TACERN) study.
- Neuropsychological testing (Mullen Scales of Early Learning, Vineland Adaptive Behavior Scales, Preschool Language Scales) pre- and post-surgery.
- Statistical analysis using Pearson correlation and multivariate linear regression to correlate surgical timing with neurocognitive changes.
Main Results:
- Earlier age at surgery was significantly associated with greater improvement in verbal abilities (MSEL) and language scores (PLS-5).
- Shorter epilepsy duration prior to surgery showed similar positive trends for language development.
- Associations with adaptive behavior scores (VABS-2) were less consistent.
Conclusions:
- Earlier epilepsy surgery and reduced epilepsy duration are associated with improved postoperative language outcomes in children with TSC.
- Further prospective studies are required to confirm the impact of surgical timing on neurocognitive development.
- These findings highlight the importance of considering surgical timing for optimizing neurocognitive outcomes in pediatric epilepsy associated with TSC.
Objective:
The authors evaluated the impact of the timing of epilepsy surgery on postoperative neurocognitive outcomes in a cohort of children followed in the multiinstitutional Tuberous Sclerosis Complex (TSC) Autism Center of Excellence Research Network (TACERN) study.
Methods:
Twenty-seven of 159 patients in the TACERN cohort had drug-refractory epilepsy and underwent surgery. Ages at surgery ranged from 15.86 to 154.14 weeks (median 91.93 weeks). Changes in patients' first preoperative (10-58 weeks) to last postoperative (155-188 weeks) scores on three neuropsychological tests-the Mullen Scales of Early Learning (MSEL), the Vineland Adaptive Behavior Scales, 2nd edition (VABS-2), and the Preschool Language Scales, 5th edition (PLS-5)-were calculated. Pearson correlation and multivariate linear regression models were used to correlate test outcomes separately with age at surgery and duration of epilepsy prior to surgery. Analyses were separately conducted for patients whose seizure burdens decreased postoperatively (n = 21) and those whose seizure burdens did not (n = 6). Regression analysis was specifically focused on the 21 patients who achieved successful seizure control.
Results:
Age at surgery was significantly negatively correlated with the change in the combined verbal subtests of the MSEL (R = -0.45, p = 0.039) and predicted this score in a multivariate linear regression model (β = -0.09, p = 0.035). Similar trends were seen in the total language score of the PLS-5 (R = -0.4, p = 0.089; β = -0.12, p = 0.014) and in analyses examining the duration of epilepsy prior to surgery as the independent variable of interest. Associations between age at surgery and duration of epilepsy prior to surgery with changes in the verbal subscores of VABS-2 were more variable (R = -0.15, p = 0.52; β = -0.05, p = 0.482).
Conclusions:
Earlier surgery and shorter epilepsy duration prior to surgery were associated with greater improvement in postoperative language in patients with TSC. Prospective or comparative effectiveness clinical trials are needed to further elucidate surgical timing impacts on neurocognitive outcomes.
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