先天性脑瘫伴癫痫:临床与基因比较

N. V. Chebanenko, P. L. Sokol, A. G. Prityko
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摘要

背景。先天性脑瘫(CP)的问题是相关的,因为这类患者的适应和社会适应的复杂性有限。目前正在积极研究该病发病机制的遗传方面。CP常伴发癫痫,其特点是难以治愈。分析CP患者的临床、遗传学和神经影像学特征。材料和方法。本研究纳入136例CP患者。采用NGS和Sanger三联法对静脉血材料进行遗传研究。对基因在决定因子组中的分布进行了分析。在136例患者中,发现了91个致病变异基因。在CS(细胞骨架形成和功能的调控)、ENM(神经元膜兴奋性的调控)、CMTR(染色质修饰、转录和复制过程的调控)、NTS(神经递质代谢和突触功能的调控)等决定因子组中,它们的数量较多。基因根据运动缺陷程度的分布具有特异性:在所有组中,除管病基因(ENM)外,某些基因对应于每个运动缺陷程度。这种特异性在ENM组中不太明显。脑结构异常最多的是CMTR(控制染色质修饰、转录和复制过程)、CS(调节细胞骨架的形成和功能)和ENM(调节神经元膜的兴奋性)组。RMF组(线粒体装置功能的调节)的特点是对癫痫的抵抗力最高。在带有管病基因(ENM)的组中,癫痫过程不是最难治性的。根据对CP伴癫痫发病机制的贡献,神经组织兴奋性和传导的决定因素(ENM和NTS)的分布,神经细胞形成过程的调节(NOG和CMTR),以及导致储存病(GSD)的酶缺陷的预先确定是允许的。决定因素ENM负责运动缺陷的形成和癫痫过程的形成。同时,其对运动缺陷的影响是非特异性的,癫痫过程的难治愈程度在很大程度上决定了线粒体功能调节的决定因素。
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Congenital cerebral palsy with epilepsy: clinical and genetic comparisons
Background. The problem of congenital cerebral palsy (CP) is relevant due to the limited complexity of habilitation and social adaptation of such patients. The genetic aspects of the pathogenesis of the disease are being actively studied. CP is often accompanied by epilepsy, which is characterized by refractoriness.Aim. To analyze the clinical, genetic and neuroimaging aspects of this pathology in CP patients.Materials and methods. The study included 136 patients with CP. Genetic studies were carried out on venous blood material using NGS and Sanger trio methods. The distribution of genes into groups of determinants was carried out.Results. In 136 patients, 91 genes with pathogenic variants were found. There were more of them in the determinant groups CS (regulation of cytoskeleton formation and functioning), ENM (regulation of neuronal membrane excitability), CMTR (control of chromatin modifications, transcription and replication processes), NTS (regulation of neurotransmitter metabolism and synapse functioning). The distribution of genes according to the degree of motor deficiency was specific: in all groups, except for canalopathy genes (ENM): certain genes corresponded to each degree of motor deficiency. This specificity was less pronounced in the ENM group. The largest number of cases of abnormalities in the structure of the brain was in the CMTR (control of chromatin modifications, transcription and replication processes), CS (regulation of the formation and functioning of the cytoskeleton) and ENM (regulation of the excitability of the neuronal membrane) groups. The RMF group (regulation of the functions of the mitochondrial apparatus) was characterized by the highest resistance to epilepsy. In cases from the group with the canalopathy genes (ENM), the epileptic process was not the most refractory.Conclusions. According to the contribution to the pathogenesis of CP with epilepsy, the distribution of determinants for the provision of excitability and conduction of the nervous tissue (ENM and NTS), the regulation of neuroontogenesis processes (NOG and CMTR), and the predetermination of enzymatic defects leading to storage diseases (GSD) are permissible. The determinant ENM is responsible for both the formation of motor deficits and the formation of the epileptic process. At the same time, its influence on motor deficit is nonspecific, and the degree of refractoriness of the epileptic process largely determines the determinant of mitochondrial function regulation.
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