关键词: drug-refractory epilepsy epilepsy surgery language development neurocognitive outcomes surgical timing tuberous sclerosis complex verbal ability

来  源:   DOI:10.3171/2024.4.PEDS2481

Abstract:
OBJECTIVE: The authors evaluated the impact of the timing of epilepsy surgery on postoperative neurocognitive outcomes in a cohort of children followed in the multiinstitutional Tuberous Sclerosis Complex (TSC) Autism Center of Excellence Research Network (TACERN) study.
METHODS: Twenty-seven of 159 patients in the TACERN cohort had drug-refractory epilepsy and underwent surgery. Ages at surgery ranged from 15.86 to 154.14 weeks (median 91.93 weeks). Changes in patients\' first preoperative (10-58 weeks) to last postoperative (155-188 weeks) scores on three neuropsychological tests-the Mullen Scales of Early Learning (MSEL), the Vineland Adaptive Behavior Scales, 2nd edition (VABS-2), and the Preschool Language Scales, 5th edition (PLS-5)-were calculated. Pearson correlation and multivariate linear regression models were used to correlate test outcomes separately with age at surgery and duration of epilepsy prior to surgery. Analyses were separately conducted for patients whose seizure burdens decreased postoperatively (n = 21) and those whose seizure burdens did not (n = 6). Regression analysis was specifically focused on the 21 patients who achieved successful seizure control.
RESULTS: Age at surgery was significantly negatively correlated with the change in the combined verbal subtests of the MSEL (R = -0.45, p = 0.039) and predicted this score in a multivariate linear regression model (β = -0.09, p = 0.035). Similar trends were seen in the total language score of the PLS-5 (R = -0.4, p = 0.089; β = -0.12, p = 0.014) and in analyses examining the duration of epilepsy prior to surgery as the independent variable of interest. Associations between age at surgery and duration of epilepsy prior to surgery with changes in the verbal subscores of VABS-2 were more variable (R = -0.15, p = 0.52; β = -0.05, p = 0.482).
CONCLUSIONS: Earlier surgery and shorter epilepsy duration prior to surgery were associated with greater improvement in postoperative language in patients with TSC. Prospective or comparative effectiveness clinical trials are needed to further elucidate surgical timing impacts on neurocognitive outcomes.
摘要:
目的:作者在多机构结节性硬化症(TSC)自闭症卓越研究网络(TACERN)研究中评估了癫痫手术时机对术后神经认知结果的影响。
方法:TACERN队列中159例患者中有27例患有药物难治性癫痫并接受了手术。手术年龄范围为15.86至154.14周(中位数为91.93周)。在三个神经心理学测试中,患者术前第一次(10-58周)到术后最后一次(155-188周)得分的变化-早期学习的Mullen量表(MSEL),Vineland适应性行为量表,第2版(VABS-2),和学龄前语言量表,计算第5版(PLS-5)。使用Pearson相关性和多元线性回归模型分别将测试结果与手术年龄和手术前癫痫持续时间相关联。分别对癫痫发作负担减轻的患者(n=21)和癫痫发作负担没有减轻的患者(n=6)进行分析。回归分析特别针对21例成功控制癫痫发作的患者。
结果:手术时的年龄与MSEL的组合言语子测验的变化呈显着负相关(R=-0.45,p=0.039),并在多元线性回归模型中预测了该得分(β=-0.09,p=0.035)。在PLS-5的总语言得分(R=-0.4,p=0.089;β=-0.12,p=0.014)以及在手术前检查癫痫持续时间的分析中也看到了类似的趋势。手术年龄和手术前癫痫持续时间与VABS-2语言评分变化之间的关联差异更大(R=-0.15,p=0.52;β=-0.05,p=0.482)。
结论:早期手术和手术前癫痫持续时间较短与TSC患者术后语言改善有关。需要前瞻性或比较有效性临床试验来进一步阐明手术时机对神经认知结果的影响。
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