Suppr超能文献

代谢性癫痫:应对诊断挑战的方法

Metabolic epilepsies: approaches to a diagnostic challenge.

作者信息

Stöckler-Ipsiroglu Sylvia, Plecko Barbara

机构信息

Department of Pediatrics, University of British Columbia, Vancouver, British Columbia. Canada.

出版信息

Can J Neurol Sci. 2009 Aug;36 Suppl 2:S67-72.

Abstract

Although inborn errors of metabolism (IEM) are a relatively rare cause of epilepsy in children, their diagnosis is important with respect to treatment, prognosis and genetic counselling. In addition to seizures and epilepsy, IEM may produce a complex clinical picture in which epilepsy is only one of the various neurologic manifestations including developmental delay/regression, mental retardation, movement disorders, micro-/macrocephaly, as well as cerebral grey and white matter changes. Dysmorphic features and cerebral dysgenesis may also be part of a metabolic epilepsy syndrome (e.g. disorders of peroxisomal biogenesis, glutaric aciduria type 2, pyruvate dehydrogenease complex deficiency). Metabolic epilepsies may dominate the clinical presentation (e.g. pyridoxine dependent epilepsy) or may precede further neurologic deterioration (e.g. neuronal ceroid lipofuscinosis) and additional organ involvement (e.g. liver failure in Alpers (POLG1) disease). Metabolic epilepsies often present with myoclonic seizures (e.g. Gaucher Disease type 3, mitochondrial syndromes) and, as a rule, patients presenting with predominantly myoclonic seizures should be carefully investigated for these types of metabolic epilepsies. Patients with very early onset of epilepsy are considered at high risk for an underlying IEM as well. In this review we present an overview of metabolic epilepsies based on various criteria such as treatability, age of onset, seizure type, and pathogenetic background. Exemplary disorders will be described in more detail including cerebral glucose transporter (GLUT1) deficiency, pyridoxine dependent epilepsy, neuronal ceroid lipofuscinosis, cathepsin D deficiency, Alpers syndrome (POLG deficiency), and guanidinoacetate methyltransferase (GAMT) deficiency.

摘要

尽管先天性代谢缺陷(IEM)是儿童癫痫相对少见的病因,但其诊断对于治疗、预后及遗传咨询而言至关重要。除了癫痫发作外,IEM可能会呈现出复杂的临床症状,其中癫痫只是包括发育迟缓/倒退、智力障碍、运动障碍、小头/巨头畸形以及脑灰白质改变等多种神经表现之一。畸形特征和脑发育异常也可能是代谢性癫痫综合征的一部分(例如过氧化物酶体生物发生障碍、2型戊二酸尿症、丙酮酸脱氢酶复合物缺乏症)。代谢性癫痫可能主导临床表现(例如吡哆醇依赖性癫痫),或者可能先于进一步的神经功能恶化(例如神经元蜡样脂褐质沉积症)及其他器官受累(例如阿尔珀斯病(POLG1)中的肝功能衰竭)。代谢性癫痫常表现为肌阵挛发作(例如3型戈谢病、线粒体综合征),通常,以肌阵挛发作为主的患者应仔细排查这类代谢性癫痫。癫痫发病极早的患者也被认为存在潜在IEM的高风险。在本综述中,我们基于可治疗性、发病年龄、发作类型和发病机制背景等各种标准,对代谢性癫痫进行了概述。将更详细地描述一些典型疾病,包括脑葡萄糖转运体(GLUT1)缺乏症、吡哆醇依赖性癫痫、神经元蜡样脂褐质沉积症、组织蛋白酶D缺乏症、阿尔珀斯综合征(POLG缺乏症)和胍基乙酸甲基转移酶(GAMT)缺乏症。

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验