Mapping efficacious deep brain stimulation for pediatric dystonia
Mapping efficacious deep brain stimulation for pediatric dystonia
复制标题
DOI：
10.3171/2020.7.peds20322
复制
发表时间：
2021-03-01
影响因子：
1.9
通讯作者：
Fasano, Alfonso
中科院分区：
文献类型：
--
作者：
Coblentz, Ailish;Elias, Gavin J. B.;Fasano, Alfonso
目的报道采用脑深部电刺激(DBS)治疗儿童内侧苍白球(GPI)肌张力障碍的经验，并用最先进的神经影像技术分析其临床转归。方法对2009年6月至2017年9月间接受GPI-DBS植入术的11例难治性肌张力障碍患儿进行回顾性分析。使用术前和术后MRI，激活的组织体积被建模，并根据临床结果进行加权，以确定与临床结果相关的脑区。结果11例患者共植入21个电极电极，分析结果表明，植入电极的功能和结构网络与临床益处相关。平均随访时间为19+/-20个月(中位数5个月)。使用7分临床评定量表，10名患者对治疗有反应，根据评分和lt；3。Burke-Fahn-Marsden Dystung评定量表运动评分的平均改善为40%+/-23%。疗效概率图显示，与临床改善最相关的体素簇位于GPI的后侧，相对后方，高于经典GPI靶点的坐标。概率图与中央前回、中央后回、顶枕叶皮质和脑等区域之间有很强的功能和结构联系。结论本研究报道了一系列肌张力障碍的儿童患者，GPI-DBS在这些患者中产生了不同的临床益处，并描述了该队列的临床有利刺激部位，以及它的结构和功能联系。这些信息可能对改进手术计划、简化编程以及进一步告知疾病病理生理学有价值。
OBJECTIVE The objective of this study was to report the authors' experience with deep brain stimulation (DBS) of the internal globus pallidus (GPi) as a treatment for pediatric dystonia, and to elucidate substrates underlying clinical outcome using state-of-the-art neuroimaging techniques.METHODS A retrospective analysis was conducted in 11 pediatric patients (6 girls and 5 boys, mean age 12 +/- 4 years) with medically refractory dystonia who underwent GPi-DBS implantation between June 2009 and September 2017. Using pre- and postoperative MRI, volumes of tissue activated were modeled and weighted by clinical outcome to identify brain regions associated with clinical outcome. Functional and structural networks associated with clinical benefits were also determined using large-scale normative data sets.RESULTS A total of 21 implanted leads were analyzed in 11 patients. The average follow-up duration was 19 +/- 20 months (median 5 months). Using a 7-point clinical rating scale, 10 patients showed response to treatment, as defined by scores < 3. The mean improvement in the Burke-Fahn-Marsden Dystonia Rating Scale motor score was 40% +/- 23%. The probabilistic map of efficacy showed that the voxel cluster most associated with clinical improvement was located at the posterior aspect of the GPi, comparatively posterior and superior to the coordinates of the classic GPi target. Strong functional and structural connectivity was evident between the probabilistic map and areas such as the precentral and postcentral gyri, parietooccipital cortex, and brainstem.CONCLUSIONS This study reported on a series of pediatric patients with dystonia in whom GPi-DBS resulted in variable clinical benefit and described a clinically favorable stimulation site for this cohort, as well as its structural and functional connectivity. This information could be valuable for improving surgical planning, simplifying programming, and further informing disease pathophysiology.