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Sleep-related hypermotor seizures of insulo-opercular origin: A review of 27 cases
Clinical Neurophysiology ( IF 4.7 ) Pub Date : 2021-08-19 , DOI: 10.1016/j.clinph.2021.03.032
Pauline M. Lobbezoo 1, 2 , Lino Nobili 3 , Giorgio Lo Russo 4 , Steve A. Gibbs 1
Affiliation  

Introduction. Insular epilepsy is known to be a “great mimicker” of various types of focal epilepsy, including sleep-related hypermotor epilepsy (SHE) in about 15% of cases. Because SHE tends to have a frontal origin, misdiagnosis can result in a poor surgical outcome when epilepsy surgery is considered. In this study, we reviewed the existing literature to identify electro-clinical features suggestive of an insulo-opercular (IO) origin in SHE.

Methodology. We found 11 case series of SHE or insular epilepsy with sufficiently detailed patient data. We identified 27 patients with IO SHE from 6 studies. When available, the semiology patterns (SP), early nonmotor manifestations, alteration of consciousness (AOC), EEG and MRI features and histopathology were collected.

Results. SP analysis of the 27 identified patients showed a wide variety of indiscriminate SP, including hyperkinetic and/or dystonic features. Early nonmotor manifestations were observed in 80%, most commonly somatosensory auras (44%). Laryngeal constriction, pain, oral and widespread paresthesia were observed in IO SHE but not in frontal SHE. AOC was uncommon (20%). EEG was rarely suggestive of an IO origin (17%) and MRI was diagnostic in 48%. Focal cortical dysplasia was the most common diagnosis (78%). Following surgery, 83% were seizure-free (follow-up: 12–60 months).

Conclusions. Few electro-clinical features, except early non-motor manifestations, are suggestive of an IO onset in SHE. Therefore, in MRI-negative patients, a heightened vigilance for subtle subjective or behavioral signs is warranted to avoid misdiagnosis and poor surgical outcome.



中文翻译:

岛盖起源的与睡眠相关的过度运动性癫痫发作:27 例回顾

介绍。众所周知,岛叶癫痫是各种类型局灶性癫痫的“极好模仿者”,包括约 15% 的睡眠相关过度运动性癫痫 (SHE)。由于 SHE 往往起源于额叶,因此在考虑进行癫痫手术时,误诊可能会导致手术结果不佳。在这项研究中,我们回顾了现有文献,以确定 SHE 中提示胰岛鳃盖 (IO) 起源的电临床特征。

方法论。我们发现了 11 个具有足够详细患者数据的 SHE 或岛叶癫痫病例系列。我们从 6 项研究中确定了 27 名 IO SHE 患者。当可用时,收集符号学模式 (SP)、早期非运动表现、意识改变 (AOC)、脑电图和 MRI 特征以及组织病理学。

结果。对 27 名已识别患者的 SP 分析显示出各种不加区分的 SP,包括运动过度和/或肌张力障碍特征。在 80% 中观察到早期非运动表现,最常见的是体感先兆 (44%)。在 IO SHE 中观察到喉部收缩、疼痛、口腔和广泛的感觉异常,但在额叶 SHE 中未观察到。AOC 并不常见(20%)。脑电图很少提示 IO 起源 (17%),48% 的 MRI 可诊断。局灶性皮质发育不良是最常见的诊断(78%)。手术后,83% 的患者无癫痫发作(随访:12-60 个月)。

结论。除了早期的非运动表现外,很少有电临床特征提示 SHE 的 IO 发作。因此,对于 MRI 阴性患者,有必要提高对细微主观或行为体征的警惕性,以避免误诊和手术结果不佳。

更新日期:2021-08-20
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