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儿童中枢神经系统肿瘤幸存者中迟发性神经系统疾病的纵向评估:儿童癌症幸存者研究报告。

Longitudinal assessment of late-onset neurologic conditions in survivors of childhood central nervous system tumors: a Childhood Cancer Survivor Study report.

机构信息

Center for Neuroscience and Behavioral Medicine, Brain Tumor Institute.

Division of Neurology, Children's National Health System, Washington, DC.

出版信息

Neuro Oncol. 2018 Jan 10;20(1):132-142. doi: 10.1093/neuonc/nox148.

Abstract

BACKGROUND

Survivors of childhood central nervous system (CNS) tumors experience high rates of treatment-related neurologic sequelae. Whether survivors continue to be at increased risk for new events as they age is unknown.

METHODS

Adverse neurologic health conditions in 5-year survivors of CNS tumors from the Childhood Cancer Survivor Study (n = 1876) were evaluated longitudinally at a median 23.0 years from diagnosis (range, 5.1-38.9), median age at last evaluation 30.3 years (range, 6.1-56.4). Multivariable regression estimated hazard ratios (HRs) and 95% CIs.

RESULTS

From 5 to 30 years post diagnosis, cumulative incidence increased for seizures from 27% to 41%, motor impairment 21% to 35%, and hearing loss 9% to 23%. Risks were elevated compared with siblings (eg, seizures HR: 12.7; 95% CI: 9.6-16.7; motor impairment HR: 7.6; 95% CI: 5.8-9.9; hearing loss HR: 18.4; 95% CI: 13.1-25.9). Regional brain doses of radiation therapy were associated with development of new deficits (eg, frontal ≥50 Gy and motor impairment HR: 2.0; 95% CI: 1.2-3.4). Increased risk for motor impairment was also associated with tumor recurrence (HR: 2.6; 95% CI: 1.8-3.8), development of a meningioma (HR: 2.3; 95% CI: 0.9-5.4), and stroke (HR: 14.9; 95% CI: 10.4-21.4). Seizure risk was doubled by recurrence (HR: 2.3; 95% CI: 1.6-3.2), meningioma (HR: 2.6; 95% CI: 1.1-6.5), and stroke (HR: 2.0; 95% CI: 1.1-3.4).

CONCLUSIONS

CNS tumor survivors remain at risk for new-onset adverse neurologic events across their lifespans at a rate greater than siblings. Cranial radiation, stroke, tumor recurrence, and development of meningioma were independently associated with late-onset adverse neurologic sequelae.

摘要

背景

儿童中枢神经系统 (CNS) 肿瘤幸存者经历高比例的治疗相关神经后遗症。随着年龄的增长,幸存者是否继续面临新事件的风险增加尚不清楚。

方法

对来自儿童癌症幸存者研究(n=1876)的 CNS 肿瘤 5 年幸存者的不良神经健康状况进行纵向评估,从诊断中位时间 23.0 年(范围,5.1-38.9),最后评估的中位年龄为 30.3 岁(范围,6.1-56.4)。多变量回归估计了危险比 (HR) 和 95%置信区间。

结果

从诊断后 5 年到 30 年,癫痫的累积发病率从 27%增加到 41%,运动障碍从 21%增加到 35%,听力损失从 9%增加到 23%。与兄弟姐妹相比,风险升高(例如,癫痫 HR:12.7;95%CI:9.6-16.7;运动障碍 HR:7.6;95%CI:5.8-9.9;听力损失 HR:18.4;95%CI:13.1-25.9)。放射治疗的局部脑剂量与新缺陷的发展有关(例如,额叶≥50 Gy 与运动障碍 HR:2.0;95%CI:1.2-3.4)。运动障碍风险增加也与肿瘤复发(HR:2.6;95%CI:1.8-3.8)、脑膜瘤(HR:2.3;95%CI:0.9-5.4)和中风(HR:14.9;95%CI:10.4-21.4)相关。复发(HR:2.3;95%CI:1.6-3.2)、脑膜瘤(HR:2.6;95%CI:1.1-6.5)和中风(HR:2.0;95%CI:1.1-3.4)使癫痫风险增加一倍。

结论

CNS 肿瘤幸存者在其整个生命周期内仍面临新的不良神经事件的风险,其发生率高于兄弟姐妹。颅放射、中风、肿瘤复发和脑膜瘤的发生与迟发性不良神经后遗症独立相关。

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