Brain structural differences associated with the behavioural phenotype in children with Williams syndrome
Brain structural differences associated with the behavioural phenotype in children with Williams syndrome
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DOI：
10.1016/j.brainres.2008.11.101
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发表时间：
2009-03-03
期刊：
影响因子：
2.9
通讯作者：
Murphy, Kieran C.
中科院分区：
文献类型：
--
作者：
Campbell, Linda E.;Daly, Eileen;Murphy, Kieran C.
背景资料：我们研究了患有威廉姆斯（WS）综合征的智力残疾儿童的脑结构形态及其与行为表型的关系。研究方法：我们比较了15名患有WS的儿童（平均年龄：13 +/- 2）和15名年龄/性别匹配的健康儿童的神经解剖学，使用手动感兴趣区域分析来测量大块（白色+灰色）组织体积，并使用无偏的全自动基于体素的形态测量来评估整个大脑中灰色/白色物质的差异。异常行为的等级与大脑结构相关。结果：与对照组相比，WS儿童的右侧顶枕区和基底神经节体积减少。我们确定了顶枕区、左侧壳核/苍白球和丘脑的灰质减少;以及基底节和右侧后扣带回的白色物质减少。相比之下，额叶、前扣带回、左颞叶的灰质和双侧前扣带回的白色物质显著增加。WS的注意力不集中与额叶、尾状核和小脑的体积差异相关，多动与左颞叶、顶叶和小脑的差异相关。最后，同伴问题的等级与颞叶、右侧基底神经节和额叶的差异有关。结论：在第一个研究中的智力残疾儿童与WS使用基于体素的形态学的大脑结构，我们的研究结果表明，这组有特定的差异，在灰色/白色物质形态。此外，还发现结构差异与WS儿童的注意力不集中、多动和同伴问题评分相关。
Background: We investigated structural brain morphology of intellectually disabled children with Williams (WS) syndrome and its relationship to the behavioural phenotype. Methods: We compared the neuroanatomy of 15 children (mean age:13 +/- 2) with WS and 15 age/gender-matched healthy children using a manual region-of-interest analysis to measure bulk (white+grey) tissue volumes and unbiased fully-automated voxel-based morphometry to assess differences in grey/white matter throughout the brain. Ratings of abnormal behaviours were correlated with brain structure. Results: Compared to controls, the brains of children with WS had a decreased volume of the right parieto-occipital regions and basal ganglia. We identified reductions of grey matter of the parieto-occipital regions, left putamen/globus pallidus and thalamus; and in white matter of the basal ganglia and right posterior cingulate gyrus. In contrast, significant increases of grey matter were identified in the frontal lobes, anterior cingulate gyrus, left temporal lobe, and of white matter bilaterally in the anterior cingulate. Inattention in WS was correlated with volumetric differences in the frontal lobes, caudate nucleus and cerebellum, and hyperactivity was related to differences in the left temporal and parietal lobes and cerebellum. Finally, ratings of peer problems were related to differences in the temporal lobes, right basal ganglia and frontal lobe. Conclusions: In one of the first studies of brain structure in intellectually disabled children with WS using voxel-based morphometry, our findings suggest that this group has specific differences in grey/white matter morphology. in addition, it was found that structural differences were correlated to ratings of inattention, hyperactivity and peer problems in children with WS. (C) 2008 Elsevier B.V. All rights reserved.