Department of Neurology, University Medical Center Groningen, University of Groningen, the Netherlands.
Department of Radiology, University Medical Center Groningen, University of Groningen, the Netherlands.
Eur J Paediatr Neurol. 2020 Sep;28:70-76. doi: 10.1016/j.ejpn.2020.07.013. Epub 2020 Aug 5.
Neonatal therapeutic hypothermia (TH) can ameliorate or prevent the development of dyskinetic cerebral palsy (CP) after hypoxic-ischemic encephalopathy (HIE). The Dyskinesia Impairment Scale (DIS) was recently launched to quantify dyskinetic (dystonic and choreatic) motor features in patients with CP. In TH treated children, who are at risk of developing dyskinetic CP, we aimed to determine DIS-scores at pre-school age.
In 21 Dutch pre-school children (3-6 years of age) who had received TH according to the Dutch-Flemish treatment protocol, we determined DIS-scores. We associated DIS-scores with 1. age-matched control values (Kuiper et al., 2018) [1], and 2. previously reported DIS-score range in dyskinetic CP (Monbaliu E et al., 2015).
The motor phenotype was determined as: normal (n = 18/21), mildly impaired (reduced coordination (n = 2/21)) and abnormal (dyskinetic CP; n = 1/21). In absence of CP (n = 20/21), DIS-scores were lower (more favorable) than in dyskinetic CP, without any overlapping group scores (mean difference: 71 points; p < .05). However, the obtained DIS-scores were still higher than previously reported in healthy age-matched controls (mean difference: 14 points; p < .05). There was an association between DIS-scores and retrospective neonatal MRI (basal ganglia and thalamus injury on diffusion weighted imaging (DWI)) and (a)EEG parameters (p < .05).
In the vast majority (95%) of Dutch TH-HIE treated pre-school children, the phenotypic motor outcome was favorable. However, DIS-scores were moderately increased compared with healthy age-matched controls. Future studies may elucidate the significance of moderately increased DIS-scores should to further extent.
新生儿治疗性低温(therapeutic hypothermia,TH)可改善或预防缺氧缺血性脑病(hypoxic-ischemic encephalopathy,HIE)后不随意运动性脑瘫(dyskinetic cerebral palsy,CP)的发生。不随意运动障碍量表(Dyskinesia Impairment Scale,DIS)最近被提出,用于量化 CP 患者的不随意运动(包括肌张力障碍和舞蹈徐动症)特征。在接受 TH 治疗的 HIE 患儿中,存在发生不随意运动 CP 的风险,我们旨在学龄前阶段评估其 DIS 评分。
在 21 名荷兰学龄前儿童(3-6 岁)中,我们根据荷兰-佛兰芒治疗方案评估了 TH 治疗后儿童的 DIS 评分。我们将 DIS 评分与 1. 年龄匹配的对照组数据(Kuiper 等人,2018 年)[1]和 2. 先前报道的不随意运动 CP 中 DIS 评分范围(Monbaliu 等人,2015 年)[2]进行了比较。
运动表型被确定为:正常(n=21/21)、轻度受损(协调能力下降[n=2/21])和异常(不随意运动 CP;n=1/21)。在无 CP 组(n=20/21),DIS 评分较低(更有利),且无重叠的组间评分(平均差异:71 分;p<.05)。然而,所获得的 DIS 评分仍高于先前报道的健康年龄匹配对照组(平均差异:14 分;p<.05)。DIS 评分与回顾性新生儿 MRI(弥散加权成像上的基底节和丘脑损伤)和(a)脑电图参数之间存在关联(p<.05)。
在绝大多数(95%)接受荷兰 TH-HIE 治疗的学龄前儿童中,表型运动结局良好。然而,与健康年龄匹配对照组相比,DIS 评分仍有中度升高。未来的研究可能会进一步阐明中度升高的 DIS 评分的意义。