Seizure outcomes in children with Rasmussen’s encephalitis undergoing resective or hemispheric epilepsy surgery: an individual participant data meta-analysis

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  • 1 John A. Burns School of Medicine, University of Hawai’i, Honolulu, Hawai’i;
  • 2 Department of Neurosurgery, University of California, Los Angeles, California;
  • 3 Brown University, Providence, Rhode Island;
  • 4 Division of Neurosurgery, Ste. Justine Hospital, University of Montreal, Quebec, Canada; and
  • 5 Division of Neurosurgery, The Hospital for Sick Children, University of Toronto, Ontario, Canada
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OBJECTIVE

The objective of this study was to perform an individual participant data meta-analysis to identify preoperative factors associated with a good seizure outcome in children with Rasmussen’s encephalitis (RE) undergoing resective or hemispheric epilepsy surgery.

METHODS

Electronic databases (PubMed, Web of Science, CINAHL) were searched with no language or date restrictions to identify cohort studies of consecutive participants undergoing resective surgery that reported seizure outcomes. The authors recorded all preoperative factors that could plausibly be associated with seizure outcomes and used Cox regression analysis to identify which of these variables were associated with seizure freedom (i.e., Engel class I).

RESULTS

Of 720 citations, 19 articles reporting on 187 participants were eligible. Seizure freedom (Engel class I) was observed in 113 participants (60.4%). On univariate analyses, younger age at disease onset (hazard ratio [HR] 0.906, p = 0.001), younger age at surgery (HR 0.928, p < 0.001), shorter time to surgery (HR 0.921, p = 0.001), and hemispherectomy (HR 0.283, p < 0.001) were all associated with longer time to postoperative seizure recurrence. Additionally, multivariable analysis including the aforementioned variables showed that younger age at surgery (HR 0.946, p = 0.043) and hemispherectomy (HR 0.297, p < 0.001) were independently and significantly associated with a greater time to seizure recurrence and longer duration of seizure freedom.

CONCLUSIONS

The majority of pediatric patients undergoing resective or hemispheric surgery for RE achieve good seizure outcome. Although small retrospective cohort studies are inherently prone to bias, the best available evidence utilizing individual participant data suggests hemispheric surgery and younger age at surgery are associated with good seizure outcomes following epilepsy surgery. Large, multicenter observational studies with long-term follow-up are required to evaluate the risk factors identified in this review.

ABBREVIATIONS AED = antiepileptic drug; CI = confidence interval; HR = hazard ratio; IPD = individual participant data; IQR = interquartile range; RE = Rasmussen’s encephalitis.

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Contributor Notes

Correspondence Aria Fallah: University of California, Los Angeles, CA. afallah@mednet.ucla.edu.

INCLUDE WHEN CITING Published online December 6, 2019; DOI: 10.3171/2019.9.PEDS19380.

Disclosures The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this paper.

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