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结节性硬化症相关癫痫的管理

Management of epilepsy in tuberous sclerosis complex.

作者信息

Curatolo P, D'Argenzio L, Cerminara C, Bombardieri R

机构信息

Tor Vergata University, Department of Neurosciences, Pediatric Neurology Unit, Rome, Italy.

出版信息

Expert Rev Neurother. 2008 Mar;8(3):457-67. doi: 10.1586/14737175.8.3.457.

Abstract

Tuberous sclerosis complex (TSC) is an inherited disorder resulting from mutations in one of two genes, TSC1 (Hamartin) and TSC2 (Tuberin). These two proteins form a cytosolic complex that inhibits the mTOR pathway that controls cell growth and proliferation. Pathologically, abnormalities of neuronal migration, cellular differentiation and excessive cellular proliferation all contribute to the formation of the different brain lesions of TSC. Seizure is the most common presenting symptom. Seizures can be present in the first year of life and up to one third of children develop infantile spasms. Seizures usually have a focal or multifocal origin, are often resistant to antiepileptic drugs and have a negative impact on the neurocognitive development. Vigabatrin has proved to be effective against infantile spasms due to TSC. New evidence suggests that it is possible to noninvasively identify using multimodality techniques, TSC children who are likely to become seizure-free following surgical treatment. Understanding the mechanisms of epileptogenesis and the possible role of the mTOR pathway in this process might increase the availability of novel and targeted therapies.

摘要

结节性硬化症(TSC)是一种遗传性疾病,由TSC1(错构瘤蛋白)和TSC2(结节蛋白)这两个基因中的一个发生突变引起。这两种蛋白质形成一种胞质复合物,抑制控制细胞生长和增殖的mTOR通路。在病理上,神经元迁移异常、细胞分化异常和细胞过度增殖均导致TSC不同脑损伤的形成。癫痫发作是最常见的首发症状。癫痫发作可在出生后第一年出现,多达三分之一的儿童会发生婴儿痉挛。癫痫发作通常起源于局灶性或多灶性,对抗癫痫药物往往耐药,并对神经认知发育产生负面影响。已证明vigabatrin对TSC所致婴儿痉挛有效。新证据表明,使用多模态技术有可能无创地识别出经手术治疗后可能无癫痫发作的TSC患儿。了解癫痫发生机制以及mTOR通路在此过程中可能发挥的作用,可能会增加新型靶向治疗方法的可及性。

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