Institute of Cell Biophysics of the Russian Academy of Sciences, Federal Research Center "Pushchino Scientific Center for Biological Research of the Russian Academy of Sciences", 142290 Pushchino, Russia.
Institute of Neuroscience, Lobachevsky State University of Nizhny Novgorod, 23 Gagarin Ave., 603022 Nizhny Novgorod, Russia.
Cells. 2024 Oct 22;13(21):1747. doi: 10.3390/cells13211747.
The mechanisms of epileptogenesis after brain injury, ischemic stroke, or brain tumors have been extensively studied. As a result, many effective antiseizure drugs have been developed. However, there are still many patients who are resistant to therapy. The molecular and genetic bases regarding such drug-resistant seizures have been poorly elucidated. In many cases, heavy seizures are instigated by brain development malformations and often caused by gene mutations. Such malformations can be demonstrated in mouse models by generating mutant strains. One of the most potent mutagens is ENU (N-ethyl-N-nitrosourea). In the present study, we describe three novel mutant strains generated by ENU-directed mutagenesis. Two of these strains present a very strong epileptic phenotype triggered by audiogenic stimuli (G9-1 and S5-1 strains). The third mouse strain is characterized by behavioral disorders and hyperexcitation of neuronal networks. We identified changes in the expression of those genes encoding neurotransmission proteins in the cerebral cortexes of these mice. It turned out that the G9-1 strain demonstrated the strongest disruptions in the expression of those genes encoding plasma membrane channels, excitatory glutamate receptors, and protein kinases. On the other hand, the number of GABAergic neurons was also affected by the mutation. All three lines are characterized by increased anxiety, excitability, and suppressed motor and orientational-exploratory activities. On the other hand, the strains with an epileptic phenotype-G9-1 and S5-1ave reduced learning ability, and the A9-2 mice line retains high learning ability.
脑损伤、缺血性中风或脑肿瘤后癫痫发生的机制已经得到了广泛的研究。因此,已经开发出许多有效的抗癫痫药物。然而,仍有许多患者对治疗有抗药性。关于这种耐药性癫痫的分子和遗传基础还没有得到很好的阐明。在许多情况下,剧烈的癫痫发作是由大脑发育畸形引发的,通常是由基因突变引起的。这种畸形可以通过生成突变株在小鼠模型中得到证明。最有效的诱变剂之一是 ENU(N-乙基-N-亚硝脲)。在本研究中,我们描述了通过 ENU 定向诱变产生的三个新的突变株。其中两个菌株表现出很强的听觉刺激触发的癫痫表型(G9-1 和 S5-1 株)。第三个小鼠品系的特征是行为障碍和神经元网络的过度兴奋。我们确定了这些小鼠大脑皮层中编码神经递质蛋白的基因表达的变化。结果表明,G9-1 株在编码质膜通道、兴奋性谷氨酸受体和蛋白激酶的基因表达中表现出最强的干扰。另一方面,突变也影响了 GABA 能神经元的数量。所有三种品系的特征是焦虑增加、兴奋性增强以及运动和定向探索活动受到抑制。另一方面,具有癫痫表型的菌株 - G9-1 和 S5-1 表现出学习能力下降,而 A9-2 小鼠系保持较高的学习能力。