Localization of the epileptogenic foci in tuberous sclerosis complex: a pediatric case report.
Localization of the epileptogenic foci in tuberous sclerosis complex: a pediatric case report.
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DOI：
10.3389/fnhum.2014.00175
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发表时间：
2014
影响因子：
2.9
通讯作者：
Papadelis C
中科院分区：
文献类型：
--
作者：
Hunold A;Haueisen J;Ahtam B;Doshi C;Harini C;Camposano S;Warfield SK;Grant PE;Okada Y;Papadelis C
多发性硬化症是一种罕见的组织生长和分化障碍，其特征是脑和其他器官中的良性错构瘤。高达90%的TSC患者会发展为癫痫，50%的患者会变得难以治疗，需要切除手术。TSC患者的手术结果取决于对由结节和周围致痫组织组成的致痫区的准确识别。致痫区究竟是在结节本身还是在异常发育的周围皮质，存在相互矛盾的证据。在这里，我们报告的定位癫痫样活动之间的许多皮质结节在一个4岁的患者与TSC相关的难治性癫痫进行脑磁图（MEG），脑电图（EEG），和扩散张量成像（DTI）。对于脑磁图，我们使用了一个原型系统，提供更高的空间分辨率和灵敏度相比，传统的成人系统。根据现行临床标准，采用等效电流偶极子（ECD）和最小范数估计（MNE）方法，使用EEG和MEG定位发作间期活动的发生器。对于DTI，我们计算了通过四个ROI的纤维的四个扩散标量参数：（i）在右象限识别的大皮质结节处，（ii）在结节对侧的正常外观组织处，（iii）在由发作间期尖峰峰值处拟合的ECD形成的簇处，以及（iv）在簇对侧的正常外观组织处。ECD一致地聚集在大的钙化皮质结节附近。MNE和ECD显示相同区域的癫痫样活动。DTI分析表明，通过结节的径迹与ECD簇的径迹的标量值之间存在差异。在这个说明性的情况下，我们提供的证据，从不同的神经影像学方式，支持认为癫痫样活动可能来自周围的结节，而不是结节本身的异常发展的组织。
Tuberous sclerosis complex (TSC) is a rare disorder of tissue growth and differentiation, characterized by benign hamartomas in the brain and other organs. Up to 90% of TSC patients develop epilepsy and 50% become medically intractable requiring resective surgery. The surgical outcome of TSC patients depends on the accurate identification of the epileptogenic zone consisting of tubers and the surrounding epileptogenic tissue. There is conflicting evidence whether the epileptogenic zone is in the tuber itself or in abnormally developed surrounding cortex. Here, we report the localization of the epileptiform activity among the many cortical tubers in a 4-year-old patient with TSC-related refractory epilepsy undergoing magnetoencephalography (MEG), electroencephalography (EEG), and diffusion tensor imaging (DTI). For MEG, we used a prototype system that offers higher spatial resolution and sensitivity compared to the conventional adult systems. The generators of interictal activity were localized using both EEG and MEG with equivalent current dipole (ECD) and minimum norm estimation (MNE) methods according to the current clinical standards. For DTI, we calculated four diffusion scalar parameters for the fibers passing through four ROIs defined: (i) at a large cortical tuber identified at the right quadrant, (ii) at the normal appearing tissue contralateral to the tuber, (iii) at the cluster formed by ECDs fitted at the peak of interictal spikes, and (iv) at the normal appearing tissue contralateral to the cluster. ECDs were consistently clustered at the vicinity of the large calcified cortical tuber. MNE and ECDs indicated epileptiform activity in the same areas. DTI analysis showed differences between the scalar values of the tracks passing through the tuber and the ECD cluster. In this illustrative case, we provide evidence from different neuroimaging modalities, which support the view that epileptiform activity may derive from abnormally developed tissue surrounding the tuber rather than the tuber itself.
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