From the Departments of Neurology (A.K., W.O.T., S.S.S.-B., A.M.F.) and Radiology (E.H.M.), Mayo Clinic, Jacksonville, FL; Department of Pediatric Neurology (P.J.), Norton Children's Hospital, Louisville, KY; and Department of Neurosurgery (S.S.S.-B.), Mayo Clinic, Jacksonville, FL.
Neurology. 2023 May 23;100(21):1020-1024. doi: 10.1212/WNL.0000000000206900. Epub 2023 Jan 25.
Focal cortical dysplasia (FCD) is a congenital developmental malformation and is one of the leading causes of drug-resistant focal epilepsy (DRFE). Although focal epilepsies traditionally have been regarded as acquired disorders, increasing evidence suggests a substantial genetic contribution to the pathogenesis of focal structural epilepsies, including FCDs. Variations in the Dishevelled, Egl-10, and domain-containing protein 5 (DEPDC5) have recently emerged as a causative gene mutation in familial focal epilepsies associated with FCD type 2a, including bottom-of-sulcus dysplasia (BOSD). We present the case of a 20-year-old man with DRFE, positive for DEPDC5 c.1555C>T (p.GIn519*) heterozygous pathogenic variant. Initial 3T brain MRI was unrevealing, but subsequent 7T MRI including 7T edge-enhancing gradient echo revealed a left superior frontal sulcus BOSD concordant with the electroclinical data. The patient underwent treatment with MR-guided laser interstitial thermal ablation of the left frontal BOSD without intracranial EEG monitoring (skipped candidate), resulting in a seizure-free outcome of 9 months since the last follow-up. Our case highlights the real-world application of summative information obtained through advancements in epilepsy genetic testing, minimally invasive surgeries, and ultra-high field MRI, allowing us to provide a safe and effective treatment for a patient with a genetic DRFE.
局灶性皮质发育不良(FCD)是一种先天性发育畸形,是耐药性局灶性癫痫(DRFE)的主要原因之一。尽管局灶性癫痫传统上被认为是后天获得性疾病,但越来越多的证据表明,局灶性结构性癫痫(包括 FCD)的发病机制存在很大的遗传因素。Dishevelled、Egl-10 和结构域蛋白 5(DEPDC5)的变异最近被认为是与 FCD 2a 相关的家族性局灶性癫痫的致病基因突变,包括底沟发育不良(BOSD)。我们报告了一例 20 岁男性 DRFE 患者,DEPDC5 c.1555C>T(p.Gln519*)杂合致病性变异阳性。初始 3T 脑部 MRI 无明显异常,但随后的 7T MRI 包括 7T 边缘增强梯度回波显示左侧额上沟 BOSD 与电临床数据一致。患者接受了左额 BOSD 的磁共振引导激光间质热消融治疗,没有颅内脑电图监测(跳过候选者),自最后一次随访以来,无癫痫发作,结果为 9 个月。我们的病例强调了通过癫痫遗传测试、微创手术和超高场 MRI 获得的综合信息的实际应用,使我们能够为患有遗传性 DRFE 的患者提供安全有效的治疗。