Frassoni Carolina, Avagliano Laura, Inverardi Francesca, Spaccini Luigina, Parazzini Cecilia, Rustico Maria Angela, Bulfamante Gaetano, Righini Andrea
Clinical Epileptology and Experimental Neurophysiology Unit, Fondazione IRCCS, Istituto Neurologico "C. Besta," Milano, Italy.
Department of Health Sciences, San Paolo Hospital Medical School, University of Milan, Milano, Italy.
Neuropediatrics. 2016 Aug;47(4):253-8. doi: 10.1055/s-0036-1583185. Epub 2016 May 13.
The development of the human cerebral cortex is a complex and precisely programmed process by which alterations may lead to morphological and functional neurological abnormalities. We report familial cases of prenatally diagnosed abnormal brain, characterized by aberrant symmetrical mesial oversulcation of the parietooccipital lobes, in fetuses affected by abnormal skeletal features. Fetal brain anomalies were characterized by prenatal magnetic resonance imaging at 21 weeks of gestation and histologically evaluated at 22 weeks. Histological examination added relevant information showing some focal cortical areas of micropoligyria and heterotopic extension of the cortical plate into the marginal zone beneath the cortical surface. Genetic analysis of the fetuses excluded FGFR3 mutations known to be related to skeletal dysplasia and aberrant symmetrical oversulcation in other brain areas (temporal lobes). Hence, the present report suggests the existence of a class of rare syndromes of skeleton and brain development abnormality unrelated to FGFR3 mutations or related to other not described FGFR3 gene defects. Using magnetic resonance imaging, histopathology and molecular characterization we provide an example of a translational study of a rare and unreported brain congenital malformation.
人类大脑皮层的发育是一个复杂且精确编程的过程,在此过程中发生的改变可能会导致形态和功能上的神经学异常。我们报告了产前诊断为脑异常的家族性病例,其特征为受异常骨骼特征影响的胎儿顶枕叶出现异常对称性内侧过度脑沟化。胎儿脑异常通过妊娠21周时的产前磁共振成像进行表征,并在22周时进行组织学评估。组织学检查补充了相关信息,显示出一些局灶性皮质微小脑回以及皮质板向皮质表面下方边缘区的异位延伸。对胎儿的基因分析排除了已知与骨骼发育不良以及其他脑区(颞叶)异常对称性过度脑沟化相关的FGFR3突变。因此,本报告表明存在一类与FGFR3突变无关或与其他未描述的FGFR3基因缺陷相关的骨骼和脑发育异常的罕见综合征。通过磁共振成像、组织病理学和分子特征分析,我们提供了一个对罕见且未报道的脑先天性畸形进行转化研究的实例。