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反应性神经刺激治疗耐药性癫痫患儿:病例系列及文献复习。

Responsive neurostimulation for pediatric patients with drug-resistant epilepsy: a case series and review of the literature.

机构信息

1Department of Neurosurgery, University of Pittsburgh School of Medicine, Pittsburgh.

2Division of Child Neurology, Department of Pediatrics, University of Pittsburgh School of Medicine, Pittsburgh; and.

出版信息

Neurosurg Focus. 2022 Oct;53(4):E10. doi: 10.3171/2022.7.FOCUS22331.

Abstract

OBJECTIVE

Responsive neurostimulation (RNS) is a promising treatment for pediatric patients with drug-resistant epilepsy for whom resective surgery is not an option. The relative indications and risk for pediatric patients undergoing RNS therapy require further investigation. Here, the authors report their experience with RNS implantation and therapy in pediatric patients.

METHODS

The authors performed a retrospective chart review to identify patients implanted with RNS depth or strip electrodes for the treatment of drug-resistant epilepsy at their institution between 2020 and 2022. Patient demographics, surgical variables, and patient seizure outcomes (Engel class and International League Against Epilepsy [ILAE] reporting) were evaluated.

RESULTS

The authors identified 20 pediatric patients ranging in age from 8 to 21 years (mean 15 [SD 4] years), who underwent RNS implantation, including depth electrodes (n = 15), strip electrodes (n = 2), or both (n = 3). Patient seizure semiology, onset, and implantation strategy were heterogeneous, including bilateral centromedian nucleus (n = 5), mesial temporal lobe (n = 4), motor cortex or supplementary motor area (n = 7), or within an extratemporal epileptogenic zone (n = 4). There were no acute complications of RNS implantation (hemorrhage or stroke) or device malfunctions. One patient required rehospitalization for postoperative infection. At the longest follow-up (mean 10 [SD 7] months), 13% patients had Engel class IIB, 38% had Engel class IIIA, 6% had Engel class IIIB, 19% had Engel class IVA, 19% had Engel class IVB, and 6% had Engel class IVC outcomes. Using ILAE metrics, 6% were ILAE class 3, 25% were ILAE class 4, and 69% were ILAE class 5.

CONCLUSIONS

This case series supports current literature suggesting that RNS is a safe and potentially effective surgical intervention for pediatric patients with drug-resistant epilepsy. The authors report comparable rates of serious adverse events to current RNS literature in pediatric and adult populations. Seizure outcomes may continue to improve with follow-up as stimulation strategy is refined and the chronic neuromodulatory effect evolves, as previously described in patients with RNS. Further large-scale, multicenter case series of RNS in pediatric patients with drug-resistant epilepsy are required to determine long-term pediatric safety and effectiveness.

摘要

目的

反应性神经刺激(RNS)是一种有前途的治疗方法,适用于药物难治性癫痫且不能进行切除术的儿科患者。需要进一步研究儿科患者接受 RNS 治疗的相对适应证和风险。在此,作者报告了他们在该机构对接受 RNS 深度或带状电极植入以治疗药物难治性癫痫的儿科患者的经验。

方法

作者进行了回顾性图表审查,以确定 2020 年至 2022 年期间在他们的机构中接受 RNS 深度或带状电极植入以治疗药物难治性癫痫的患者。评估了患者的人口统计学、手术变量和患者的癫痫发作结果(Engel 分级和国际抗癫痫联盟[ILAE]报告)。

结果

作者确定了 20 名年龄在 8 至 21 岁之间(平均 15 [SD 4] 岁)的儿科患者,他们接受了 RNS 植入,包括深度电极(n = 15)、带状电极(n = 2)或两者(n = 3)。患者的癫痫发作症状学、发作起始和植入策略各不相同,包括中央核团(n = 5)、内侧颞叶(n = 4)、运动皮层或辅助运动区(n = 7)或在颞外致痫区(n = 4)。RNS 植入没有急性并发症(出血或中风)或设备故障。1 名患者因术后感染需要再次住院治疗。在最长的随访时间(平均 10 [SD 7] 个月),13%的患者为 Engel 分级 IIB,38%的患者为 Engel 分级 IIIA,6%的患者为 Engel 分级 IIIB,19%的患者为 Engel 分级 IVA,19%的患者为 Engel 分级 IVB,6%的患者为 Engel 分级 IVC。根据 ILAE 指标,6%的患者为 ILAE 分级 3,25%的患者为 ILAE 分级 4,69%的患者为 ILAE 分级 5。

结论

本病例系列支持现有文献表明,RNS 是一种安全且潜在有效的手术干预方法,适用于药物难治性癫痫的儿科患者。作者报告的严重不良事件发生率与儿科和成人 RNS 文献中的发生率相当。随着刺激策略的不断完善和慢性神经调节作用的发展,癫痫发作结果可能会随着随访时间的延长而继续改善,正如先前在接受 RNS 治疗的患者中所描述的那样。需要进一步开展大规模的多中心 RNS 治疗儿科药物难治性癫痫的病例系列研究,以确定儿科患者的长期安全性和有效性。

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