Charles Shor Epilepsy Center, Neurological Institute, Cleveland, Ohio, USA.
Genomic Medicine Institute, Cleveland Clinic, Cleveland, Ohio, USA.
Epilepsia. 2022 Aug;63(8):1899-1919. doi: 10.1111/epi.17301. Epub 2022 Jun 15.
Ongoing challenges in diagnosing focal cortical dysplasia (FCD) mandate continuous research and consensus agreement to improve disease definition and classification. An International League Against Epilepsy (ILAE) Task Force (TF) reviewed the FCD classification of 2011 to identify existing gaps and provide a timely update. The following methodology was applied to achieve this goal: a survey of published literature indexed with ((Focal Cortical Dysplasia) AND (epilepsy)) between 01/01/2012 and 06/30/2021 (n = 1349) in PubMed identified the knowledge gained since 2012 and new developments in the field. An online survey consulted the ILAE community about the current use of the FCD classification scheme with 367 people answering. The TF performed an iterative clinico-pathological and genetic agreement study to objectively measure the diagnostic gap in blood/brain samples from 22 patients suspicious for FCD and submitted to epilepsy surgery. The literature confirmed new molecular-genetic characterizations involving the mechanistic Target Of Rapamycin (mTOR) pathway in FCD type II (FCDII), and SLC35A2 in mild malformations of cortical development (mMCDs) with oligodendroglial hyperplasia (MOGHE). The electro-clinical-imaging phenotypes and surgical outcomes were better defined and validated for FCDII. Little new information was acquired on clinical, histopathological, or genetic characteristics of FCD type I (FCDI) and FCD type III (FCDIII). The survey identified mMCDs, FCDI, and genetic characterization as fields for improvement in an updated classification. Our iterative clinico-pathological and genetic agreement study confirmed the importance of immunohistochemical staining, neuroimaging, and genetic tests to improve the diagnostic yield. The TF proposes to include mMCDs, MOGHE, and "no definite FCD on histopathology" as new categories in the updated FCD classification. The histopathological classification can be further augmented by advanced neuroimaging and genetic studies to comprehensively diagnose FCD subtypes; these different levels should then be integrated into a multi-layered diagnostic scheme. This update may help to foster multidisciplinary efforts toward a better understanding of FCD and the development of novel targeted treatment options.
目前在诊断局灶性皮质发育不良(FCD)方面仍存在诸多挑战,这需要不断进行研究并达成共识,以改善疾病定义和分类。国际抗癫痫联盟(ILAE)工作组(TF)回顾了 2011 年的 FCD 分类,以确定现有的差距并提供及时的更新。为了实现这一目标,采用了以下方法:对 2012 年 1 月 1 日至 2021 年 6 月 30 日期间在 PubMed 上索引的((局灶性皮质发育不良)和(癫痫))文献进行调查,以确定自 2012 年以来的知识进展和该领域的新发展。在线调查咨询了 ILAE 社区,了解目前对 FCD 分类方案的使用情况,有 367 人回答。TF 进行了迭代的临床-病理和遗传一致性研究,以客观地衡量 22 名疑似 FCD 并接受癫痫手术的患者的血液/脑组织样本中的诊断差距。文献证实了新的分子遗传学特征,涉及机制靶向雷帕霉素(mTOR)途径在 FCDII 型和 SLC35A2 型轻度皮质发育不良(mMCDs)伴少突胶质细胞增生(MOGHE)中。FCDII 的电临床成像表型和手术结果得到了更好的定义和验证。在 FCDI 型和 FCDIII 型的临床、组织病理学或遗传特征方面几乎没有获得新的信息。调查确定了 mMCDs、FCDI 和遗传特征是更新分类中需要改进的领域。我们的迭代临床病理和遗传一致性研究证实了免疫组织化学染色、神经影像学和基因测试对提高诊断产量的重要性。TF 提议在更新的 FCD 分类中增加新类别,包括 mMCDs、MOGHE 和“组织病理学无明确 FCD”。通过先进的神经影像学和遗传研究可以进一步增强组织病理学分类,以全面诊断 FCD 亚型;然后将这些不同层次的信息整合到一个多层次的诊断方案中。此次更新可能有助于促进多学科努力,以更好地了解 FCD 并开发新的靶向治疗选择。