Sustained Relief of Generalized Dystonia Despite Prolonged Interruption of Deep Brain Stimulation

Sustained Relief of Generalized Dystonia Despite Prolonged Interruption of Deep Brain Stimulation
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DOI:
10.1002/mds.25353
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发表时间:
2013-09-01
期刊:
影响因子:
8.6
通讯作者:
Tagliati, Michele
Tagliati, Michele
中科院分区:
医学1区
文献类型:
--
作者:
Cheung, Tyler;Zhang, Cen;Tagliati, Michele

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背景苍白深部脑刺激(DBS)是一种治疗致残、药物难治性全身性肌张力障碍的有效方法。当停止刺激时,患者通常会恢复到术前基线。方法报告2例肌张力障碍患者的病例。结果2例原发性全身性肌张力障碍患者(1例DYT1突变)分别接受了18个月和5年的双侧苍白性DBS治疗,在无意中中断刺激后仍能保持几个月的运动益处。单侧刺激中断3个月和7个月,双侧中断2天和2个月。肌张力障碍症状在治疗中断期间仅部分恢复,恢复刺激后迅速完全消失。结论:我们报告了2例肌张力障碍患者,尽管暂时停止pallidal DBS,但运动益处的持续保留出乎意料。区分这些个体的因素似乎是年龄小、病程短以及刺激能量相对较低的慢性DBS治疗。(c) 2013年国际帕金森和运动障碍学会
BackgroundPallidal deep brain stimulation (DBS) is an established treatment for disabling, medication-refractory generalized dystonia. Patients typically regress to their preoperative baseline when stimulation is discontinued.MethodsPresented are case reports of 2 dystonia patients.ResultsTwo patients with primary generalized dystonia (1 with the DYT1 mutation) who were treated successfully with bilateral pallidal DBS for periods of 18 months and 5 years retained motor benefit for several months after inadvertent interruption of stimulation. Stimulation was interrupted unilaterally for 3 and 7 months and bilaterally for 2 days and 2 months, respectively. Symptoms of dystonia returned only partially during the period of therapy interruption and rapidly and completely resolved after resuming stimulation.ConclusionsWe report unexpected and prolonged retention of motor benefits despite transient cessation of pallidal DBS in 2 dystonia patients. Factors that appear to differentiate these individuals are young age, short duration of disease, and chronic DBS therapy with relatively low energy of stimulation. (c) 2013 International Parkinson and Movement Disorder Society