病变性肌张力障碍患者的临床和结构发现：已发表病例的描述性和定量分析。
Clinical and Structural Findings in Patients With Lesion-Induced Dystonia: Descriptive and Quantitative Analysis of Published Cases.
机构信息
From the Cognitive Neuroscience Unit (D.T.C., J.M.-H., G.M., E.Y.), School of Psychology, Deakin University, Geelong, Australia; Center for Brain Circuit Therapeutics (D.T.C., M.D.F., J.J.), Brigham and Women's Hospital, Boston, MA; Deakin University (C.G.), Centre for Social and Early Emotional Development, School of Psychology, Faculty of Health, Geelong, Australia; Murdoch Children's Research Institute (C.G.), Centre for Adolescent Health, Melbourne, Australia; Turku Brain and Mind Center (J.P., J.J.), Clinical Neurosciences, University of Turku, Finland; Departments of Neurology and Human Genetics (H.J.), Emory University, School of Medicine, Atlanta, GA; Department of Neurology (M.D.F.), Harvard Medical School, Boston, MA; and Turku PET Centre (J.J.), Neurocenter, Turku University Hospital, Finland.
出版信息
Neurology. 2022 Nov 1;99(18):e1957-e1967. doi: 10.1212/WNL.0000000000201042. Epub 2022 Aug 17.
BACKGROUND AND OBJECTIVES
Brain lesions are a well-recognized etiology of dystonia. These cases are especially valuable because they offer causal insight into the neuroanatomical substrates of dystonia. To date, knowledge of lesion-induced dystonia comes mainly from isolated case reports or small case series, restricting broader description and analysis.
METHODS
Cases of lesion-induced dystonia were first identified from a systematic review of published literature. Latent class analysis then investigated whether patients could be classified into subgroups based on lesion location and body regions affected by dystonia. Regression analyses subsequently investigated whether subgroup membership predicted clinical characteristics of dystonia.
RESULTS
Three hundred fifty-nine published cases were included. Lesions causing dystonia occurred in heterogeneous locations, most commonly in the basal ganglia (46.2%), followed by the thalamus (28.1%), brainstem (22.6%), and white matter (21.2%). The most common form of lesion-induced dystonia was focal dystonia (53.2%), with the hand (49.9%) and arm (44.3%) most commonly affected. Of all cases, 86.6% reported co-occurring neurologic manifestations and 26.1% reported other movement disorders. Latent class analysis identified 3 distinct subgroups of patients: those with predominantly limb dystonias, which were associated with basal ganglia lesions; those with hand dystonia, associated with thalamic lesions; and those with predominantly cervical dystonia, associated with brainstem and cerebellar lesions. Regression demonstrated significant differences between these subgroups on a range of dystonia symptoms, including dystonic tremor, symptom latency, other movement disorders, and dystonia variability.
DISCUSSION
Although dystonia can be induced by lesions to numerous brain regions, there are distinct relationships between lesion locations and dystonic body parts. This suggests that the affected brain networks are different between types of dystonia.
背景与目的
脑损伤是导致肌张力障碍的一个公认病因。这些病例特别有价值，因为它们为肌张力障碍的神经解剖学基础提供了因果关系的见解。迄今为止，关于损伤引起的肌张力障碍的知识主要来自于孤立的病例报告或小的病例系列，限制了更广泛的描述和分析。
方法
首先从已发表文献的系统综述中确定了由损伤引起的肌张力障碍的病例。然后，潜在类别分析调查了患者是否可以根据病灶位置和受肌张力障碍影响的身体区域分为亚组。回归分析随后调查了亚组成员是否可以预测肌张力障碍的临床特征。
结果
共纳入 359 例已发表的病例。导致肌张力障碍的病变发生在不同的位置，最常见于基底节（46.2%），其次是丘脑（28.1%）、脑干（22.6%）和白质（21.2%）。最常见的损伤引起的肌张力障碍形式是局灶性肌张力障碍（53.2%），手部（49.9%）和手臂（44.3%）最常见受累。所有病例中，86.6%报告伴有同时发生的神经系统表现，26.1%报告伴有其他运动障碍。潜在类别分析确定了 3 组不同的患者亚组：主要表现为肢体肌张力障碍的患者，与基底节病变相关；手部肌张力障碍的患者，与丘脑病变相关；主要表现为颈肌张力障碍的患者，与脑干和小脑病变相关。回归分析显示，这些亚组之间在多种肌张力障碍症状上存在显著差异，包括张力障碍性震颤、症状潜伏期、其他运动障碍和肌张力障碍的可变性。
讨论
尽管许多脑区的损伤都可以导致肌张力障碍，但病灶位置与痉挛部位之间存在明显的关系。这表明不同类型的肌张力障碍之间受影响的大脑网络不同。
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