Acharya Jayant N, Acharya Vinita J
Department of Neurology, Penn State University Hershey Medical Center, Hershey, Pennsylvania, U.S.A.
J Clin Neurophysiol. 2023 Feb 1;40(2):100-108. doi: 10.1097/WNP.0000000000000913. Epub 2022 Jun 30.
Myoclonus can be epileptic or nonepileptic. Epileptic myoclonus has been defined in clinical, neurophysiological, and neuroanatomical terms. Juvenile myoclonic epilepsy (JME) is typically considered to be an adolescent-onset idiopathic generalized epilepsy with a combination of myoclonic, generalized tonic-clonic, and absence seizures and normal cognitive status that responds well to anti-seizure medications but requires lifelong treatment. EEG shows generalized epileptiform discharges and photosensitivity. Recent observations indicate that the clinical picture of JME is heterogeneous and a number of neuropsychological and imaging studies have shown structural and functional abnormalities in the frontal lobes and thalamus. Advances in neurophysiology and imaging suggest that JME may not be a truly generalized epilepsy, in that restricted cortical and subcortical networks appear to be involved rather than the entire brain. Some patients with JME may be refractory to anti-seizure medications and attempts have been made to identify neurophysiological biomarkers predicting resistance. Progressive myoclonic epilepsy is a syndrome with multiple specific causes. It is distinct from JME because of the occurrence of progressive neurologic dysfunction in addition to myoclonus and generalized tonic-clonic seizures but may sometimes be difficult to distinguish from JME or misdiagnosed as drug-resistant JME. This article provides an overview of progressive myoclonic epilepsy and focuses on the clinical and neurophysiological findings in the two most commonly recognized forms of progressive myoclonic epilepsy-Unverricht-Lundborg disease (EPM1) and Lafora disease (EPM2). A variety of neurophysiological tests can be used to distinguish between JME and progressive myoclonic epilepsy and between EPM1 and EPM2.
肌阵挛可分为癫痫性和非癫痫性。癫痫性肌阵挛已从临床、神经生理学和神经解剖学角度进行了定义。青少年肌阵挛癫痫(JME)通常被认为是一种青少年起病的特发性全身性癫痫,伴有肌阵挛、全身性强直阵挛发作和失神发作,认知状态正常,对抗癫痫药物反应良好,但需要终身治疗。脑电图显示全身性癫痫样放电和光敏感性。最近的观察表明,JME的临床表现具有异质性,多项神经心理学和影像学研究显示额叶和丘脑存在结构和功能异常。神经生理学和影像学的进展表明,JME可能并非真正的全身性癫痫,因为受累的似乎是局限的皮质和皮质下网络,而非整个大脑。一些JME患者可能对抗癫痫药物难治,人们已尝试识别预测耐药性的神经生理学生物标志物。进行性肌阵挛癫痫是一种有多种特定病因的综合征。它与JME不同,因为除了肌阵挛和全身性强直阵挛发作外,还会出现进行性神经功能障碍,但有时可能难以与JME区分,或被误诊为耐药性JME。本文概述了进行性肌阵挛癫痫,并重点介绍了两种最常见的进行性肌阵挛癫痫——翁韦里希特-伦德堡病(EPM1)和拉福拉病(EPM2)的临床和神经生理学发现。多种神经生理学检查可用于区分JME与进行性肌阵挛癫痫,以及EPM1与EPM2。