Highlights
- •Individual participant data meta-analysis of pediatric Sturge-Weber syndrome.
- •Resective and hemispheric epilepsy surgery achieve comparable seizure outcomes.
- •Resective surgery may be an appropriate alternative in some patients.
Abstract
Background
A subpopulation of patients with Sturge-Weber syndrome (SWS) develop medically intractable
epilepsy. There is a paucity of literature on preoperative factors that predict postoperative
seizure outcomes in these patients. An individual participant data meta-analysis (IPDMA)
was performed to discern preoperative variables associated with favorable seizure
outcomes in pediatric SWS patients undergoing epilepsy surgery.
Methods
PubMed, Cochrane, Web of Science, and Scopus were independently queried following
PRISMA guidelines. Studies that reported seizure outcomes in individual pediatric
SWS patients were selected. Preoperative demographic variables and disease characteristics
were recorded and evaluated in a time-to-event fashion via Cox regression and Kaplan-Meier
analysis with log-rank test.
Results
A total of 18 studies with 108 patients were included for meta-analysis. Median age
at seizure onset was 4.5 months, and 85 patients (78.7%) were seizure-free at last
follow-up (median: 72 months). On multivariable Cox regression, no variables were
independent predictors of post-operative seizure freedom duration, including the extent
of hemispheric resection. There were also no differences in time-to-seizure recurrence
on Kaplan-Meier analysis when comparing those treated with hemispheric surgery and
those with less than hemispheric surgery (p = 0.52).
Conclusion
This IPDMA showed that both resective and hemispheric epilepsy surgery achieve favorable
and comparable seizure outcomes in pediatric SWS patients. The best available evidence
using IPD suggests that resective surgery may be an appropriate alternative to hemispheric
epilepsy surgery in well-selected patients. Prospective multi-institutional studies
with greater follow-up are warranted to further investigate predictors of seizure
outcome in pediatric SWS patients.
Keywords
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Article info
Publication history
Published online: March 15, 2023
Accepted:
March 13,
2023
Received in revised form:
March 6,
2023
Received:
December 24,
2022
Footnotes
Disclosures: Aria Fallah is supported by the Dr. Alfonsina Q. Davies Endowed Chair for Epilepsy Research. This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors. The authors report no conflict of interests concerning the methods or findings of this paper.
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© 2023 British Epilepsy Association. Published by Elsevier Ltd. All rights reserved.
