Keen Joseph R, Przekop Allison, Olaya Joffre E, Zouros Alexander, Hsu Frank P K
Departments of 1 Neurosurgery and.
J Neurosurg Pediatr. 2014 Dec;14(6):585-93. doi: 10.3171/2014.8.PEDS141. Epub 2014 Oct 17.
Deep brain stimulation (DBS) for dystonic cerebral palsy (CP) has rarely been reported, and its efficacy, though modest when compared with that for primary dystonia, remains unclear, especially in the pediatric population. The authors present a small series of children with dystonic CP who underwent bilateral pallidal DBS, to evaluate the treatment's efficacy and safety in the pediatric dystonic CP population.
The authors conducted a retrospective review of patients (under the age of 18 years) with dystonic CP who had undergone DBS of the bilateral globus pallidus internus between 2010 and 2012. Two of the authors independently assessed outcomes using the Barry-Albright Dystonia Scale (BADS) and the Burke-Fahn-Marsden Dystonia Rating Scale-movement (BFMDRS-M).
Five children were diagnosed with dystonic CP due to insults occurring before the age of 1 year. Mean age at surgery was 11 years (range 8-17 years), and the mean follow-up was 26.6 months (range 2-42 months). The mean target position was 20.6 mm lateral to the midcommissural point. The mean preoperative and postoperative BADS scores were 23.8 ± 4.9 (range 18.5-29.0) and 20.0 ± 5.5 (range 14.5-28.0), respectively, with a mean overall percent improvement of 16.0% (p = 0.14). The mean preoperative and postoperative BFMDRS-M scores were 73.3 ± 26.6 (range 38.5-102.0) and 52.4 ± 21.5 (range 34.0-80.0), respectively, with a mean overall percent improvement of 28.5% (p = 0.10). Those stimulated at least 23 months (4 patients) improved 18.3% (p = 0.14) on the BADS and 30.5% (p = 0.07) on the BFMDRS-M. The percentage improvement per body region yielded conflicting results between rating scales; however, BFMDRS-M scores for speech showed some of the greatest improvements. Two patients required hardware removal (1 complete system, 1 unilateral electrode) within 4 months after implantation because of infections that resolved with antibiotics.
All postoperative dystonia rating scale scores improved with pallidal stimulation, and the greatest improvements occurred in those stimulated the longest. The results were modest but comparable to findings in other similar series. Deep brain stimulation remains a viable treatment option for childhood dystonic CP, although young children may have an increased risk of infection. Of particular note, improvements in the BFMDRS-M subscores for speech were comparable to those for other muscle groups, a finding not previously reported.
脑深部电刺激术(DBS)治疗肌张力障碍型脑瘫(CP)鲜有报道,其疗效与原发性肌张力障碍相比虽不显著,但仍不明确,尤其是在儿科患者中。作者报告了一小系列接受双侧苍白球DBS治疗的肌张力障碍型CP患儿,以评估该治疗方法在儿科肌张力障碍型CP患者中的疗效和安全性。
作者对2010年至2012年间接受双侧内侧苍白球DBS治疗的18岁以下肌张力障碍型CP患者进行了回顾性研究。两名作者使用巴里 - 奥尔布赖特肌张力障碍量表(BADS)和伯克 - 法恩 - 马斯登肌张力障碍评定量表 - 运动部分(BFMDRS - M)独立评估结果。
5名儿童因1岁前的损伤被诊断为肌张力障碍型CP。手术时的平均年龄为11岁(范围8 - 17岁),平均随访时间为26.6个月(范围2 - 42个月)。平均靶点位置在连合中点外侧20.6毫米处。术前和术后BADS评分的平均值分别为23.8±4.9(范围18.5 - 29.0)和20.0±5.5(范围14.5 - 28.0),总体平均改善率为16.0%(p = 0.14)。术前和术后BFMDRS - M评分的平均值分别为73.3±26.6(范围38.5 - 102.0)和52.4±21.5(范围34.0 - 80.0),总体平均改善率为28.5%(p = 0.10)。接受刺激至少23个月的4名患者,BADS评分改善了18.3%(p = 0.14),BFMDRS - M评分改善了30.5%(p = 0.07)。每个身体部位的改善百分比在两个评定量表之间产生了相互矛盾的结果;然而,BFMDRS - M的言语评分显示出一些最大的改善。2名患者在植入后4个月内需要取出硬件(一套完整系统,1根单侧电极),因为感染经抗生素治疗后得到解决。
苍白球刺激术后所有肌张力障碍评定量表评分均有改善,刺激时间最长的患者改善最大。结果虽不显著,但与其他类似系列的研究结果相当。脑深部电刺激术仍然是儿童肌张力障碍型CP的一种可行治疗选择,尽管幼儿感染风险可能增加。特别值得注意的是,BFMDRS - M言语亚评分的改善与其他肌肉群相当,这一发现此前未见报道。