Successful Combination of Pallidal and Thalamic Stimulation for Intractable Involuntary Movements in Patients with Neuroacanthocytosis
Successful Combination of Pallidal and Thalamic Stimulation for Intractable Involuntary Movements in Patients with Neuroacanthocytosis
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苍白球和丘脑刺激的成功结合治疗神经棘红细胞增多症患者的顽固性不自主运动
DOI：
10.1016/j.wneu.2015.06.052
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发表时间：
2015
期刊：
影响因子：
2
通讯作者：
kato A（4名省略）
中科院分区：
文献类型：
--
作者：
Nakano N; kato A（4名省略）
神经棘红细胞增多症（NA）是一种罕见的神经退行性疾病，涉及严重的不自主运动，包括舞蹈病，肌张力障碍和躯干痉挛。目前的治疗方法对这些不自主运动无效。虽然有一些关于使用脑深部电刺激治疗NA患者的报告，但最佳刺激靶点尚未确定。一些作者报道了刺激内侧苍白球可成功改善NA症状，另一些作者报道了刺激丘脑腹侧口侧复合体可减少躯干痉挛。我们调查是否最佳的目标是很好地定义为NA.MethodsWe描述的效果相结合的苍白球内丘脑和腹口复合体的刺激在2例NA谁提出了严重的顽固性不自主movements.ResultsGpi刺激单独是一个不够的效果躯干痉挛和/或舞蹈症。在没有Gpi刺激的情况下给予Vo复合体刺激导致2周后躯干痉挛改善，并且还可能对不自主运动（包括舞蹈病）产生不完全影响。Gpi和Vo复合刺激的组合减少了躯干痉挛和舞蹈病。这种改善在术后3个月保持不变。手术后1年的统一亨廷顿氏病评定量表评分低于术前。结论Gpi刺激似乎不足以控制暴力不自主运动，因此，GPi和Vo复合刺激相结合提供了一些温和的优势，Gpi刺激单独。
ObjectiveNeuroacanthocytosis (NA) is a rare neurodegenerative disease that involves severe involuntary movements including chorea, dystonia, and trunk spasms. Current treatments are not effective for these involuntary movements. Although there are a few reports on the use of deep brain stimulation to treat patients with NA, the optimal stimulation target is not yet definitive. Some authors have reported successful improvement of NA symptoms with stimulation of the globus pallidum interna, and others have reported a reduction in trunk spasm with stimulation of the ventralis oralis complex of the thalamus. We investigated whether the optimal target is well defined for NA.MethodsWe describe the effect of combination stimulation of the globus pallidum interna and the ventralis oralis complex of the thalamus in 2 patients with NA who presented with severe intractable involuntary movements.ResultsGpi stimulation alone was an insufficient effect for trunk spasm and/or chorea. Vo complex stimulation given without Gpi stimulation resulted in improvement of trunk spasm after 2 weeks and might also have had an incomplete effect on involuntary movement including a chorea. The combination of Gpi and Vo complex stimulation reduced the trunk spasms and chorea. This improvement was maintained at 3 months after surgery. The Unified Huntington's Disease Rating Scale score at 1 year after surgery was lower than that before surgery.ConclusionsGpi stimulation appears to be insufficient to control violent involuntary movements; therefore, combined GPi and Vo complex stimulation provided some moderate advantage over Gpi stimulation alone.