Mandarano Romina, Danieli Alberto, Petacchi Elisa, Di Pede Chiara, Mondani Massimo, Armellin Maria Teresa, Facchin Dina, Martinuzzi Andrea
Unit for Severe Disabilities in Developmental Age, Scientific Institute IRCCS E. Medea, Conegliano, Treviso, Italy.
Unit of Epilepsy and Clinical Neurophysiology, Scientific Institute IRCCS E. Medea, Conegliano, Treviso, Italy.
Eur J Paediatr Neurol. 2022 Mar;37:62-67. doi: 10.1016/j.ejpn.2022.01.014. Epub 2022 Feb 1.
Pallidal Deep Brain Stimulation (DBS) is an established treatment option for isolated, inherited or idiopathic dystonia, however data on its safety and efficacy in other forms of dystonia are more limited.
Retrospective analysis of motor and non-motor outcomes in pediatric onset refractory dystonia due to static or progressive brain disorders in a cohort of patients with a DBS treatment duration ≥12 months.
Multidisciplinary assessments including standardised scales/tests of motor function, pain, quality of life, cognition and language were carried out before implantation and longitudinally afterwards.
9 patients were included, 7 had cerebral palsy. Mean age at implantation was 209 months ± 156, mean treatment duration 84 ± 37 months. DBS was well tolerated and positively affected both motor and non-motor functions. In particular, statistically significant improvements were documented in Burke-Fahn-Marsden Scale scores (- 19.9% p 0.01031) at 12 months and in long-term quality of life (+28.6%, p 0.0292).
DBS may be a useful treatment option in generalized dystonia associated with brain pathology. Even when the motor benefits are limited, improvements in quality of life and non-motor functions, or the possible prevention of serious dystonia-related complications, may have a significant impact on overall clinical status.
苍白球深部脑刺激术(DBS)是孤立性、遗传性或特发性肌张力障碍的既定治疗选择,然而其在其他形式肌张力障碍中的安全性和有效性数据更为有限。
对一组接受DBS治疗≥12个月的儿童期起病的难治性肌张力障碍患者进行回顾性分析,这些患者因静态或进行性脑部疾病导致肌张力障碍。
在植入前及之后进行纵向多学科评估,包括运动功能、疼痛、生活质量、认知和语言的标准化量表/测试。
纳入9例患者,7例患有脑瘫。植入时的平均年龄为209个月±156个月,平均治疗时间为84±37个月。DBS耐受性良好,对运动和非运动功能均有积极影响。特别是,在12个月时伯克-法恩-马斯登量表评分有统计学意义的改善(-19.9%,p=0.01031),长期生活质量也有改善(+28.6%,p=0.0292)。
DBS可能是与脑部病变相关的全身性肌张力障碍的一种有效治疗选择。即使运动益处有限,生活质量和非运动功能的改善,或可能预防严重的肌张力障碍相关并发症,可能对整体临床状况产生重大影响。