Holinger Dorothy P, Bellugi Ursula, Mills Debra L, Korenberg Julie R, Reiss Allan L, Sherman Gordon F, Galaburda Albert M
Division of Behavioral Neurology, Beth Israel Deaconess Medical Center and Harvard Medical School, Boston, MA 02215, USA.
Brain Res. 2005 Mar 10;1037(1-2):35-42. doi: 10.1016/j.brainres.2004.11.038.
Williams Syndrome (WS) is a neurodevelopment disorder associated with a hemizygous deletion on chromosome 7. WS is characterized with mental retardation, severe visual-spatial deficits, relative language preservation, and excellent facial recognition. Distinctive auditory features include musical ability, heightened sound sensitivity, and specific patterns of auditory evoked potentials. These features have led to the hypothesis that the dorsal forebrain is more affected than the ventral. Previously, we reported primary visual area 17 abnormalities in rostral striate cortex, a region contributing to the dorsal visual pathway. Based on the dorsal-ventral hypothesis, and language and auditory findings, we predicted a more normal histometric picture in auditory area 41. We used an optical dissector method to measure neurons in layers II-VI of area 41 in right and left hemispheres of the same 3 WS and 3 control brains used in the area 17 study. There was a hemisphere by diagnosis interaction in cell packing density (CPD) in layer IV and in cell size in layer III between WS and control brains. Post hoc analysis disclosed in control brains, but not WS, a layer IV left > right asymmetry in CPD, and a layer III left < right asymmetry in cell size. WS brains showed more large neurons bilaterally in layer II and in left layer VI. Histometric alterations in area 41 were less widespread than rostral visual cortex. Also, there was less asymmetry in the WS brain. We interpret layers II and VI differences as reflecting increased limbic connectivity in primary auditory cortex of WS.
威廉姆斯综合征(WS)是一种与7号染色体半合子缺失相关的神经发育障碍。WS的特征是智力迟钝、严重的视觉空间缺陷、相对保留语言能力以及出色的面部识别能力。独特的听觉特征包括音乐能力、增强的声音敏感性以及听觉诱发电位的特定模式。这些特征导致了一种假设,即背侧前脑比腹侧受影响更大。此前,我们报告了在参与背侧视觉通路的区域——喙状纹状皮质中,初级视觉区17存在异常。基于背腹侧假设以及语言和听觉方面的发现,我们预测听觉区41的组织计量学图像会更正常。我们使用光学分割法,对17区研究中使用的相同3个WS大脑和3个对照大脑的左右半球听觉区41的II - VI层神经元进行测量。在WS大脑和对照大脑之间,IV层的细胞堆积密度(CPD)以及III层的细胞大小存在诊断与半球的交互作用。事后分析显示,在对照大脑中,而非WS大脑中,IV层的CPD存在左>右的不对称性,III层的细胞大小存在左<右的不对称性。WS大脑双侧II层和左侧VI层显示出更多的大神经元。41区的组织计量学改变不如喙状视觉皮质广泛。此外,WS大脑中的不对称性较小。我们将II层和VI层的差异解释为反映了WS初级听觉皮质中边缘系统连接性的增加。