Deep brain stimulation in pediatric dystonia: a systematic review.
Deep brain stimulation in pediatric dystonia: a systematic review.
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小儿肌张力障碍中的大脑刺激：系统评价。
DOI：
10.1007/s10143-018-1047-9
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发表时间：
2020-06
影响因子：
2.8
通讯作者：
Englot DJ
中科院分区：
文献类型：
--
作者：
Hale AT;Monsour MA;Rolston JD;Naftel RP;Englot DJ
虽然脑深部电刺激（DBS）治疗在儿童中相对罕见，但它可能在肌张力障碍中发挥作用，以减少运动症状和残疾。儿科DBS研究较少，且受样本量小的限制，因此，对结局了解甚少。因此，我们对文献进行了系统回顾，包括DBS治疗儿童（年龄< 21岁）肌张力障碍的研究。在基线和末次术后随访时记录患者人口统计学资料、疾病原因和特征、运动评分和残疾评分。我们确定了19项研究，报告了76例肌张力障碍儿童的DBS结局。手术年龄为13.8 ± 3.9（平均值±标准差）岁，58%的患者为男性。术后随访时间为2.8 ± 2.8年。68%的患者患有原发性肌张力障碍（PD），其中56%的患者存在DYT 1病理性突变（DYT 1+）。在所有患者中，无论肌张力障碍类型如何，DBS后Burke-Fahn-Marsden肌张力障碍评定量表（BFMDRS）运动（-M）评分改善43.8 ± 36%，而BFMDRS残疾（-D）评分改善43.7 ± 31%。与肌张力障碍的次要原因患者（21%，p = 0.004）相比，PD患者的BFMDRS-M评分更有可能改善≥ 50%（56%）。DYT 1+患者的BFMDRS-D改善率（65%）高于DTY 1 −患者（29%，p = 0.02），尽管DYT 1+（66%）或DYT 1 −（43%）儿童的BFMDRS-M改善率无差异（p = 0.11）。虽然DBS在儿科患者中不太常见，但患有严重肌张力障碍的患者可能会从神经调节治疗中获得值得的益处。
While deep brain stimulation (DBS) treatment is relatively rare in children, it may have a role in dystonia to reduce motor symptoms and disability. Pediatric DBS studies are sparse and limited by small sample size, and thus, outcomes are poorly understood. Thus, we performed a systematic review of the literature including studies of DBS for pediatric (age < 21) dystonia. Patient demographics, disease causes and characteristics, motor scores, and disability scores were recorded at baseline and at last post-operative follow-up. We identified 19 studies reporting DBS outcomes in 76 children with dystonia. Age at surgery was13.8 ± 3.9 (mean ± SD) years, and 58% of individuals were male. Post-operative follow-up duration was 2.8 ± 2.8 years. Sixty-eight percent of patients had primary dystonia (PD), of whom 56% had a pathological mutation in DYT1 (DYT1+). Across all patients, regardless of dystonia type, 43.8 ± 36% improvement was seen in Burke-Fahn-Marsden Dystonia Rating Scale (BFMDRS) motor (-M) scores after DBS, while 43.7 ± 31% improvement was observed in BFMDRS disability (-D) scores. Patients with PD were more likely to experience ≥ 50% improvement (56%) in BFMDRS-M scores compared to patients with secondary causes of dystonia (21%, p = 0.004). DYT1+ patients were more likely to achieve ≥ 50% improvement (65%) in BFMDRS-D than DTY1− individuals (29%, p = 0.02), although there was no difference in BFMDRS-M ≥ 50% improvement rates between DYT1+ (66%) or DYT1− (43%) children (p = 0.11). While DBS is less common in pediatric patients, individuals with severe dystonia may receive worthwhile benefit with neuromodulation treatment.
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