Network Localization of Pediatric Lesion-Induced Dystonia

IF 8.1 1区医学 Q1 CLINICAL NEUROLOGY

Annals of Neurology Pub Date : 2025-03-10 DOI:10.1002/ana.27224

Rose Gelineau-Morel MD, Nomazulu Dlamini MD, PhD, Joel Bruss BA, Alexander L. Cohen MD, PhD, Amanda Robertson MSc, Dimitrios Alexopoulos MS,, Christopher D. Smyser MD, Aaron D. Boes MD, PhD

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Abstract

Objective

Dystonia is a movement disorder defined by involuntary muscle contractions leading to abnormal postures or twisting and repetitive movements. Classically dystonia has been thought of as a disorder of the basal ganglia, but newer results in idiopathic dystonia and lesion-induced dystonia in adults point to broader motor network dysfunction spanning the basal ganglia, cerebellum, premotor cortex, sensorimotor, and frontoparietal regions. It is unclear whether a similar network is shared between different etiologies of pediatric lesion-induced dystonia.

Methods

Three cohorts of pediatric patients with lesion-induced dystonia were identified. The lesion etiologies included hypoxia, kernicterus, and stroke versus comparison subjects with acquired lesions not associated with dystonia. Multivariate lesion-symptom mapping and lesion network mapping were used to evaluate the anatomy and networks associated with dystonia.

Results

Multivariate lesion-symptom mapping showed that lesions of the putamen and globus pallidus were associated with dystonia (r = 0.41, p < 0.001). Lesion network mapping using normative connectome data from healthy children demonstrated that these regional findings occurred within a common brain-wide network that involves the basal ganglia, anterior and medial cerebellum, and cortical regions that overlap the cingulo-opercular action-mode and somato-cognitive-action networks.

Interpretation

We interpret these findings as novel evidence for a unified dystonia brain network that involves the somato-cognitive-action network, which is implicated in the coordination of movement. Elucidation of this network gives insight into the functional origins of dystonia and provides novel targets to investigate for therapeutic intervention. ANN NEUROL 2025;98:152–162

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小儿病变性肌张力障碍的网络定位。

目的：肌张力障碍是一种运动障碍，由不随意肌收缩导致异常姿势或扭曲和重复运动。传统上，肌张力障碍被认为是基底神经节的一种疾病，但在成人中特发性肌张力障碍和病变性肌张力障碍的最新研究结果表明，更广泛的运动网络功能障碍跨越基底神经节、小脑、运动前皮层、感觉运动和额顶叶区。目前尚不清楚在儿童病变性肌张力障碍的不同病因之间是否存在相似的网络。方法：确定了三组患有病变性肌张力障碍的儿童患者。病变病因包括缺氧、核黄疸和中风，而与之相比，获得性病变与肌张力障碍无关。多变量病变-症状映射和病变网络映射被用来评估与肌张力障碍相关的解剖学和网络。结果：多变量病变-症状图谱显示，壳核和苍白球的病变与肌张力障碍有关（r = 0.41, p）。解释：我们将这些发现解释为肌张力障碍脑网络统一的新证据，该网络涉及躯体-认知-行动网络，该网络涉及运动协调。这一网络的阐明有助于深入了解肌张力障碍的功能起源，并为研究治疗干预提供了新的靶点。Ann neurol 2025。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

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来源期刊

Annals of Neurology 医学-临床神经学

CiteScore

18.00

自引率

1.80%

发文量

270

审稿时长

3-8 weeks

期刊介绍： Annals of Neurology publishes original articles with potential for high impact in understanding the pathogenesis, clinical and laboratory features, diagnosis, treatment, outcomes and science underlying diseases of the human nervous system. Articles should ideally be of broad interest to the academic neurological community rather than solely to subspecialists in a particular field. Studies involving experimental model system, including those in cell and organ cultures and animals, of direct translational relevance to the understanding of neurological disease are also encouraged.