Husain A M, Foley C M, Legido A, Chandler D A, Miles D K, Grover W D
Department of Medicine, Division of Neurology, Duke University and Neurodiagnostic Center, Veterans Affairs Medical Center, Durham, North Carolina 27710, USA.
Pediatr Neurol. 2000 Sep;23(3):233-5. doi: 10.1016/s0887-8994(00)00186-7.
West syndrome occurs commonly in children with tuberous sclerosis complex and is associated with a grave prognosis for cognitive and seizure outcomes. We sought to determine the epilepsy outcome of children with tuberous sclerosis complex and West syndrome and whether EEG, MRI, or steroid therapy duration were different in those whose epilepsy improved compared with those with intractable seizures. Seventeen patients with tuberous sclerosis complex and West syndrome were identified. For each patient, two sets of clinical evaluations, EEG and MRI data, and treatment information separated by at least 12 months were obtained. The patients were divided into two seizure outcome groups. EEG, MRI, and treatment data were compared between the groups. The intellectual deficiency was either severe (76%) or moderate (24%). Seizure control improved in 10 and worsened in seven, without mortality (follow-up range = 12-216 months). No significant differences in EEG background, MRI findings, or steroid treatment duration were evident between the groups. The difference in EEG-sleep approached statistical significance (P = 0.06). Our findings did not confirm reports of high mortality and poor epilepsy outcome in intellectually deficient children with West syndrome and tuberous sclerosis complex. EEG sleep was the best indicator of seizure control and approached statistical significance. The duration of steroid therapy had no influence on seizure control.
韦斯特综合征常见于患有结节性硬化症的儿童,且与认知和癫痫发作预后不良相关。我们试图确定患有结节性硬化症和韦斯特综合征的儿童的癫痫预后,以及癫痫改善的儿童与难治性癫痫儿童相比,脑电图(EEG)、磁共振成像(MRI)或类固醇治疗持续时间是否存在差异。我们确定了17例患有结节性硬化症和韦斯特综合征的患者。为每位患者获取了两组临床评估、EEG和MRI数据以及至少间隔12个月的治疗信息。将患者分为两个癫痫发作结果组。对两组之间的EEG、MRI和治疗数据进行了比较。智力缺陷严重的占76%,中度的占24%。10例患者的癫痫发作得到控制,7例恶化,无死亡病例(随访范围为12 - 216个月)。两组之间在EEG背景、MRI表现或类固醇治疗持续时间方面无明显差异。EEG睡眠方面的差异接近统计学意义(P = 0.06)。我们的研究结果未证实关于智力缺陷的韦斯特综合征和结节性硬化症儿童高死亡率和癫痫预后不良的报道。EEG睡眠是癫痫发作控制的最佳指标,且接近统计学意义。类固醇治疗持续时间对癫痫发作控制没有影响。