Abstract
ObjectiveTo perform a systematic review and individual participant data meta-analysis to identify preoperative factors associated with a good seizure outcome in children with Tuberous Sclerosis Complex undergoing resective epilepsy surgery.Data SourcesElectronic databases (MEDLINE, EMBASE, CINAHL and Web of Science), archives of major epilepsy and neurosurgery meetings, and bibliographies of relevant articles, with no language or date restrictions.Study SelectionWe included case-control or cohort studies of consecutive participants undergoing resective epilepsy surgery that reported seizure outcomes. We performed title and abstract and full text screening independently and in duplicate. We resolved disagreements through discussion.Data ExtractionOne author performed data extraction which was verified by a second author using predefined data fields including study quality assessment using a risk of bias instrument we developed. We recorded all preoperative factors that may plausibly predict seizure outcomes.Data SynthesisTo identify predictors of a good seizure outcome (i.e. Engel Class I or II) we used logistic regression adjusting for length of follow-up for each preoperative variable.ResultsOf 9863 citations, 20 articles reporting on 181 participants were eligible. Good seizure outcomes were observed in 126 (69%) participants (Engel Class I: 102(56%); Engel class II: 24(13%)). In univariable analyses, absence of generalized seizure semiology (OR = 3.1, 95%CI = 1.2–8.2, p = 0.022), no or mild developmental delay (OR = 7.3, 95%CI = 2.1–24.7, p = 0.001), unifocal ictal scalp electroencephalographic (EEG) abnormality (OR = 3.2, 95%CI = 1.4–7.6, p = 0.008) and EEG/Magnetic resonance imaging concordance (OR = 4.9, 95%CI = 1.8–13.5, p = 0.002) were associated with a good postoperative seizure outcome.ConclusionsSmall retrospective cohort studies are inherently prone to bias, some of which are overcome using individual participant data. The best available evidence suggests four preoperative factors predictive of good seizure outcomes following resective epilepsy surgery. Large long-term prospective multicenter observational studies are required to further evaluate the risk factors identified in this review.
Highlights
Problem definition Tuberous sclerosis complex (TSC) is a genetic, variably expressed and multisystem disorder with a prevalence of 1 in 10,000 [1]
Of 9863 citations, 20 articles reporting on 181 participants were eligible
Absence of generalized seizure semiology (OR = 3.1, 95%confidence intervals (CI) = 1.2–8.2, p = 0.022), no or mild developmental delay (OR = 7.3, 95%CI = 2.1–24.7, p = 0.001), unifocal ictal scalp electroencephalographic (EEG) abnormality (OR = 3.2, 95%CI = 1.4–7.6, p = 0.008) and EEG/ Magnetic resonance imaging concordance (OR = 4.9, 95%CI = 1.8–13.5, p = 0.002) were associated with a good postoperative seizure outcome
Summary
Problem definition Tuberous sclerosis complex (TSC) is a genetic, variably expressed and multisystem disorder with a prevalence of 1 in 10,000 [1]. TSC is one of the leading causes of genetic epilepsy with seizures affecting almost 90% of affected individuals [2]. A third of these patients will achieve seizure freedom on antiepileptic drugs [3]. If an epileptogenic zone (EZ) associated with one or more tubers, ideally in non-eloquent cortex, can be localized, resective surgery may be offered as a cure. 57% of children achieve seizure freedom and another 18% experience a reduction (.90%) in seizure frequency at 1-year follow-up [4]. Other benefits include the possibility of decreasing or discontinuing antiepileptic drugs, ability to obtain/ retain employment, ability to drive, improved independent functioning and improved social relationships with family and friends
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