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首页> 外文期刊>Journal of neurology >White matter and cerebellar involvement in alternating hemiplegia of childhood
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White matter and cerebellar involvement in alternating hemiplegia of childhood

机译：白质和小脑参与交替童年的偏瘫

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Objective To determine whether brain volumetric and white matter microstructural changes are present and correlate with neurological impairment in subjects with alternating hemiplegia of childhood (AHC). Methods In this prospective single-center study, 12 AHC subjects (mean age 22.9 years) and 24 controls were studied with 3DT1-weighted MR imaging and high angular resolution diffusion imaging at 3T. Data obtained with voxel-based morphometry and tract-based spatial statistics were correlated with motor impairment using the International Cooperative Ataxia Rating Scale (ICARS) and Movement and Disability sub-scales of Burke-Fahn-Marsden Dystonia Rating Scale (BFMMS and BFMDS). Results Compared to healthy controls, AHC subjects showed lower total brain volume (P < 0.001) and white matter volume (P = 0.002), with reduced clusters of white matter in frontal and parietal regions (P < 0.001). No significant regional differences were found in cortical or subcortical grey matter volumes. Lower cerebellar subvolumes correlated with worse ataxic symptoms and global motor impairment in AHC group (P < 0.001). Increased mean and radial diffusivity values were found in the corpus callosum, corticospinal tracts, superior and inferior longitudinal fasciculi, subcortical frontotemporal white matter, internal and external capsules, and optic radiations (P < 0.001). These diffusion scalar changes correlated with higher ICARS and BFMDS scores (P < 0.001). Interpretation AHC subjects showed prevalent white matter involvement, with reduced volume in several cerebral and cerebellar regions associated with widespread microstructural changes reflecting secondary myelin injury rather than axonal loss. Conversely, no specific pattern of grey matter atrophy emerged. Lower cerebellar volumes, correlating with severity of neurological manifestations, seems related to disrupted developmental rather than neurodegenerative processes.
机译：目的判断是否存在脑体积和白质微观结构的变化并与具有儿童（AHC）交替偏瘫的受试者的神经损伤相关。该前瞻性单中心研究中的方法，使用3DT1加权MR成像和3T的高角度分辨率扩散成像研究了12 AHC受试者（平均22.9岁）和24个对照。使用基于体素的形态学和基于传道的空间统计而获得的数据与Motor-Fahn-Marsden Dystonia评级规模（BFMMS和BFMDS）的国际合作共济失调评级规模（ICAR）和运动和残疾子标度相关联。结果与健康对照相比，AHC受试者表现出较低的总脑体积（P <0.001）和白质量（P = 0.002），额外的白质簇减少（P <0.001）。皮质或皮质灰质卷中没有发现显着的区域差异。下部小脑亚伏与AHC组中的差异差异和全球电机损伤相关联（P <0.001）。在胼callosum，皮质脊髓，高级和较差的纵向筋膜，皮质辐射仪，内部和外胶囊和视光辐射中发现了增加的平均值和径向扩散值增加率和径向扩散值。这些扩散标量改变与更高的ICAR和BFMDS分数相关（P <0.001）。解释AHC受试者表现出普遍的白质涉及，其几种脑和小脑区域中的体积减少，其与反映第二髓鞘损伤而非轴突损失的普遍微观结构变化相关。相反，出现了灰质萎缩的特定模式。较低的小脑体积，与神经表现的严重程度相关，似乎与破坏的发育而非神经变性过程有关。

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