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儿童斯特奇-韦伯综合征伴癫痫手术的癫痫发作结局:一项个体参与者数据荟萃分析。

Seizure outcomes in children with Sturge-Weber syndrome undergoing epilepsy surgery: An individual participant data meta-analysis.

机构信息

School of Medicine and Health Sciences, The George Washington University, Washington, DC, United States.

School of Medicine, Virginia Commonwealth University, Richmond, United States.

出版信息

Seizure. 2023 Apr;107:43-51. doi: 10.1016/j.seizure.2023.03.008. Epub 2023 Mar 15.

DOI:10.1016/j.seizure.2023.03.008
PMID:36958063
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.

摘要

背景

斯特奇-韦伯综合征(SWS)的一个亚组患者会出现药物难治性癫痫。关于预测这些患者术后癫痫发作结果的术前因素的文献很少。进行了一项个体参与者数据荟萃分析(IPDMA),以辨别接受癫痫手术的儿科 SWS 患者中与术后良好癫痫发作结果相关的术前变量。

方法

按照 PRISMA 指南,分别在 PubMed、Cochrane、Web of Science 和 Scopus 上进行独立查询。选择报告了个别儿科 SWS 患者癫痫发作结果的研究。记录了术前人口统计学变量和疾病特征,并通过 Cox 回归和 Kaplan-Meier 分析(对数秩检验)以时间事件的方式进行评估。

结果

共有 18 项研究纳入了 108 名患者进行荟萃分析。癫痫发作的中位年龄为 4.5 个月,85 名患者(78.7%)在最后一次随访时无癫痫发作(中位时间:72 个月)。多变量 Cox 回归显示,包括半球切除术的范围在内,没有任何变量是术后癫痫无发作持续时间的独立预测因素。在 Kaplan-Meier 分析中,比较半球手术和半球以下手术的患者,在癫痫复发时间上也没有差异(p=0.52)。

结论

这项 IPDMA 表明,在儿科 SWS 患者中,切除性手术和半球性癫痫手术都能获得良好且可比的癫痫发作结果。使用 IPD 的最佳现有证据表明,在选择合适的患者时,切除性手术可能是半球性癫痫手术的一种合适替代方法。需要进行前瞻性多机构研究并进行更长时间的随访,以进一步研究儿科 SWS 患者癫痫发作结果的预测因素。

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引用本文的文献

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J Clin Med. 2025 Mar 22;14(7):2182. doi: 10.3390/jcm14072182.
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Epileptic seizures as an initial symptom for Sturge‑Weber syndrome type III: A report of two cases.癫痫发作作为III型斯特奇-韦伯综合征的首发症状:两例报告。
Exp Ther Med. 2024 May 24;28(1):299. doi: 10.3892/etm.2024.12588. eCollection 2024 Jul.
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Sturge-Weber Syndrome: A Report of a Rare Case.斯特奇-韦伯综合征:一例罕见病例报告。
Cureus. 2023 Dec 26;15(12):e51110. doi: 10.7759/cureus.51110. eCollection 2023 Dec.