Division of Neurology, Department of Pediatrics, Western University, London, Ontario, Canada.
Division of Neurology, Department of Paediatrics, The Hospital for Sick Children, Toronto, Ontario, Canada.
Pediatr Neurol. 2022 Jan;126:11-19. doi: 10.1016/j.pediatrneurol.2021.09.017. Epub 2021 Sep 30.
Tuberous sclerosis complex (TSC) is a rare autosomal dominant condition that affects multiple body systems. Disruption of the mammalian target of rapamycin (mTOR) pathway results in abnormal cell growth, proliferation, protein synthesis, and cell differentiation and migration in TSC. In the central nervous system, mTOR disruption is also believed to influence neuronal excitability and promote epileptogenesis. Epilepsy is the most common neurological manifestation of TSC and affects 80% to 90% of individuals with high rates of treatment resistance (up to 75%). The onset of epilepsy in the majority of individuals with TSC occurs before the age of two years, which is a critical time in neurodevelopment. Both medically refractory epilepsy and early-onset epilepsy are associated with intellectual disability in TSC, while seizure control and remission are associated with lower rates of cognitive impairment. Our current knowledge of the treatment of epilepsy in TSC has expanded immensely over the last decade. Several new therapies such as preemptive vigabatrin therapy in infants, cannabidiol, and mTOR inhibitors have emerged in recent years for the treatment of epilepsy in TSC. This review will provide clinicians with a comprehensive overview of the pharmacological and nonpharmacological therapies available for the treatment of epilepsy related to TSC.
结节性硬化症复合征(TSC)是一种罕见的常染色体显性遗传疾病,影响多个身体系统。哺乳动物雷帕霉素靶蛋白(mTOR)通路的破坏导致 TSC 中异常的细胞生长、增殖、蛋白质合成以及细胞分化和迁移。在中枢神经系统中,mTOR 破坏也被认为会影响神经元兴奋性并促进癫痫发生。癫痫是 TSC 最常见的神经学表现,影响 80%至 90%的个体,且治疗抵抗率高(高达 75%)。大多数 TSC 患者的癫痫发作发生在两岁之前,这是神经发育的关键时期。TSC 中的药物难治性癫痫和早发性癫痫都与智力残疾有关,而癫痫发作的控制和缓解与认知障碍的发生率较低有关。在过去十年中,我们对 TSC 中癫痫治疗的认识已经大大扩展。近年来出现了几种新的治疗方法,如婴儿期的预防性氨己烯酸治疗、大麻二酚和 mTOR 抑制剂,用于治疗 TSC 相关癫痫。这篇综述将为临床医生提供有关治疗与 TSC 相关癫痫的药理学和非药理学治疗方法的全面概述。