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Pilot Study of Neurodevelopmental Impact of Early Epilepsy Surgery in Tuberous Sclerosis Complex
Leslie E Grayson1, Jurriaan M Peters2, Tarrant McPherson3
1Department of Neurology, University of Alabama at Birmingham, Birmingham, Alabama.
Insights
Early epilepsy surgery in children with tuberous sclerosis complex showed some neurodevelopmental gains, particularly in language, despite ongoing seizures in some cases. Further research is needed to confirm these findings.
Area of Science:
- Pediatric Neurology
- Developmental Neuroscience
- Epileptology
Background:
- Refractory epilepsy in children with tuberous sclerosis complex (TSC) can negatively impact neurodevelopment.
- Early intervention through epilepsy surgery is being explored as a potential method to mitigate these effects.
Purpose of the Study:
- To investigate the surgical and neurodevelopmental outcomes of early epilepsy surgery in children with TSC.
- To determine if surgery before two years of age can improve developmental trajectories.
Main Methods:
- A prospective multicenter observational study included 160 children with TSC.
- Surgical outcomes were assessed using the Engel classification.
- Neurodevelopment was evaluated using Vineland-II, Mullen Scales, and Preschool Language Scales at multiple time points up to 36 months.
Main Results:
- Nineteen children underwent surgery, with 63% achieving favorable seizure control (Engel I-II).
- All children with seizures showed developmental decline or attenuated gains; the surgical group had the lowest scores.
- Favorable surgical outcomes correlated with improved language subscores on the Mullen Scales compared to medically refractory groups.
Conclusions:
- Pilot data suggest potential neurodevelopmental benefits in specific domains following early epilepsy surgery in children with TSC.
- A larger, powered study is required to validate these findings using both surgical and developmental outcome measures.
Background:
To determine if early epilepsy surgery mitigates detrimental effects of refractory epilepsy on development, we investigated surgical and neurodevelopmental outcomes in children with tuberous sclerosis complex who underwent surgery before age two years.
Methods:
Prospective multicenter observational study of 160 children with tuberous sclerosis complex. Surgical outcome was determined for the seizure type targeted by surgery. We obtained Vineland Adaptive Behavior Scales, Second Edition (Vineland-II); Mullen Scales of Early Learning; and Preschool Language Scales, Fifth Edition, at age three, six, nine, 12, 18, 24, and 36 months. Surgical cases were compared with children without seizures, with controlled seizures, and with medically refractory seizures.
Results:
Nineteen children underwent surgery (median age 17 months, range 3.7 to 21.3), and mean follow-up was 22.8 months (range 12 to 48). Surgical outcomes were favorable in 12 (63%, Engel I-II) and poor in seven (37%, Engel III-IV). Nine (47%) had new or ongoing seizures distinct from those surgically targeted. All children with seizures demonstrated longitudinal decline or attenuated gains in neurodevelopment, the surgical group scoring the lowest. Favorable surgical outcome was associated with increased Mullen Scales of Early Learning receptive and expressive language subscores compared with the medically refractory seizure group. A nonsignificant but consistent pattern of improvement with surgery was seen in all tested domains.
Conclusions:
These pilot data show neurodevelopmental gains in some domains following epilepsy surgery. A properly powered, prospective multicenter observational study of early epilepsy surgery is needed, using both surgical and developmental outcome metrics.
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