Department of Pediatrics, Children's Hospital of Michigan, Detroit Medical Center, Wayne State University School of Medicine, Detroit, MI, USA.
Neurology. 2013 Aug 13;81(7):674-80. doi: 10.1212/WNL.0b013e3182a08f3f. Epub 2013 Jul 12.
This was an observational study done on a large cohort of patients with tuberous sclerosis complex (TSC) to determine whether i) the presence of α-[(11)C]-methyl-l-tryptophan (AMT) hotspots is related to the duration of seizure intractability, ii) the presence of AMT hotspots is related to specific TSC gene mutations, and iii) there is concordance between areas with an AMT hotspot and seizure lateralization/localization on scalp EEG.
One hundred ninety-one patients (mean age: 6.7 years; median: 5 years; range: 3 months to 37 years) with TSC and intractable epilepsy were included. All patients underwent AMT-PET scan. AMT uptake in each tuber and normal-appearing cortex was measured and correlated with clinical, scalp EEG, and, if available, electrocorticographic data.
The longer the duration of seizure intractability, the greater the number of AMT hotspots (r = 0.2; p = 0.03). AMT hotspots were seen in both TSC1 and TSC2. There was excellent agreement in seizure focus lateralization between ictal scalp EEG and AMT-PET (Cohen κ 0.94) in 68 of 95 patients in whom both ictal video-EEG and AMT-PET showed lateralizing findings; in 28 of 68 patients (41%), AMT was more localizing. Furthermore, AMT-PET was localizing in 10 of 17 patients (58%) with nonlateralized ictal EEG.
AMT-PET, when used together with video-EEG, provides additional lateralization/localization data, regardless of TSC mutation. The duration of seizure intractability may predict the multiplicity of areas with AMT hotspots.
本研究对一大群结节性硬化症(TSC)患者进行了观察性研究,以确定以下内容:i)α-[(11)C]-甲基-l-色氨酸(AMT)热点的存在是否与癫痫发作的难治性持续时间有关,ii)AMT 热点的存在是否与特定的 TSC 基因突变有关,以及 iii)在 AMT 热点区域与头皮 EEG 上的癫痫发作偏侧化/定位之间是否存在一致性。
纳入了 191 名患有 TSC 和耐药性癫痫的患者(平均年龄:6.7 岁;中位数:5 岁;范围:3 个月至 37 岁)。所有患者均接受了 AMT-PET 扫描。测量了每个结节和正常皮层中的 AMT 摄取量,并将其与临床、头皮 EEG 以及如果有的话,皮质电图数据相关联。
癫痫发作难治性持续时间越长,AMT 热点的数量就越多(r = 0.2;p = 0.03)。AMT 热点在 TSC1 和 TSC2 中均可见。在 95 名同时具有发作期视频-EEG 和 AMT-PET 的患者中,68 名患者发作期头皮 EEG 和 AMT-PET 的癫痫发作焦点偏侧化具有极好的一致性(Cohen κ 0.94);在 68 名患者中的 28 名(41%),AMT 具有更好的定位作用。此外,在 17 名非偏侧性发作 EEG 患者中的 10 名(58%),AMT-PET 具有定位作用。
AMT-PET 与视频-EEG 一起使用时,无论 TSC 突变如何,均可提供额外的偏侧化/定位数据。癫痫发作的难治性持续时间可能预测 AMT 热点的多发性区域。