青少年肌阵挛性癫痫预后和治疗效果的差异:青少年肌阵挛性癫痫生物学联合会关于实用定义和分层医学分类的建议。

Brain Communications Pub Date : 2023-06-09 eCollection Date: 2023-01-01 DOI:10.1093/braincomms/fcad182
Guido Rubboli, Christoph P Beier, Kaja K Selmer, Marte Syvertsen, Amy Shakeshaft, Amber Collingwood, Anna Hall, Danielle M Andrade, Choong Yi Fong, Joanna Gesche, David A Greenberg, Khalid Hamandi, Kheng Seang Lim, Ching Ching Ng, Alessandro Orsini, Pasquale Striano, Rhys H Thomas, Jana Zarubova, Mark P Richardson, Lisa J Strug, Deb K Pal
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引用次数: 0

摘要

可靠的定义、分类和预后模型是分层医学的基石,但目前的癫痫分类系统均未涉及预后或结果问题。虽然癫痫综合征的异质性已得到广泛承认,但尚未探讨电临床特征、合并症和治疗反应的差异对诊断和预后的意义。在本文中,我们旨在为幼年肌阵挛性癫痫提供一个基于证据的定义,表明通过一组预先定义的、有限的强制性特征,可以利用幼年肌阵挛性癫痫表型的变异达到预后目的。我们的研究以青少年肌阵挛性癫痫生物学联合会收集的临床数据为基础,并辅以文献数据。我们回顾了有关死亡率和癫痫缓解的预后研究、抗癫痫药物耐药性的预测因素以及丙戊酸钠、左乙拉西坦和拉莫三嗪的部分不良药物事件。根据我们的分析,一套简化的幼年肌阵挛性癫痫诊断标准包括以下几点:(i)肌阵挛抽搐是必须的发作类型;(ii)肌阵挛的昼夜节律时间不是诊断青少年肌阵挛性癫痫的必备条件;(iii)发病年龄在6至40岁之间;(iv)全身脑电图异常;(v)智力符合人群分布。我们发现了足够的证据来提出抗癫痫药物耐受性的预测模型,该模型强调:(i) 失神发作是男女患者抗癫痫药物耐受性或癫痫发作自由度的最强分层因素;(ii) 性别是一个主要分层因素,显示抗癫痫药物耐受性的几率升高与自我报告的诱因和压力相关因素(包括睡眠不足)有关。在女性中,与脑电图测量或自我报告的光敏性相关的抗癫痫药物耐受几率降低。总之,通过应用一套简化的标准来定义幼年肌阵挛性癫痫的表型变异,我们的论文提出了基于证据的幼年肌阵挛性癫痫定义和预后分层。对现有患者个体数据集的进一步研究将有助于复制我们的研究结果,而对起始队列的前瞻性研究将有助于在幼年肌阵挛性癫痫的实际管理实践中验证这些研究结果。
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Variation in prognosis and treatment outcome in juvenile myoclonic epilepsy: a Biology of Juvenile Myoclonic Epilepsy Consortium proposal for a practical definition and stratified medicine classifications.

Reliable definitions, classifications and prognostic models are the cornerstones of stratified medicine, but none of the current classifications systems in epilepsy address prognostic or outcome issues. Although heterogeneity is widely acknowledged within epilepsy syndromes, the significance of variation in electroclinical features, comorbidities and treatment response, as they relate to diagnostic and prognostic purposes, has not been explored. In this paper, we aim to provide an evidence-based definition of juvenile myoclonic epilepsy showing that with a predefined and limited set of mandatory features, variation in juvenile myoclonic epilepsy phenotype can be exploited for prognostic purposes. Our study is based on clinical data collected by the Biology of Juvenile Myoclonic Epilepsy Consortium augmented by literature data. We review prognosis research on mortality and seizure remission, predictors of antiseizure medication resistance and selected adverse drug events to valproate, levetiracetam and lamotrigine. Based on our analysis, a simplified set of diagnostic criteria for juvenile myoclonic epilepsy includes the following: (i) myoclonic jerks as mandatory seizure type; (ii) a circadian timing for myoclonia not mandatory for the diagnosis of juvenile myoclonic epilepsy; (iii) age of onset ranging from 6 to 40 years; (iv) generalized EEG abnormalities; and (v) intelligence conforming to population distribution. We find sufficient evidence to propose a predictive model of antiseizure medication resistance that emphasises (i) absence seizures as the strongest stratifying factor with regard to antiseizure medication resistance or seizure freedom for both sexes and (ii) sex as a major stratifying factor, revealing elevated odds of antiseizure medication resistance that correlates to self-report of catamenial and stress-related factors including sleep deprivation. In women, there are reduced odds of antiseizure medication resistance associated with EEG-measured or self-reported photosensitivity. In conclusion, by applying a simplified set of criteria to define phenotypic variations of juvenile myoclonic epilepsy, our paper proposes an evidence-based definition and prognostic stratification of juvenile myoclonic epilepsy. Further studies in existing data sets of individual patient data would be helpful to replicate our findings, and prospective studies in inception cohorts will contribute to validate them in real-world practice for juvenile myoclonic epilepsy management.

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