背景:尽管生存有所改善,关于神经母细胞瘤幸存者的神经认知结局的数据很少.本研究解决了这一文献空白。
方法:使用CCSS神经认知问卷比较儿童癌症幸存者研究(CCSS)中幸存者的神经认知障碍与兄弟姐妹对照。情绪调节受损,组织,任务效率,记忆定义为兄弟姐妹范数的第90个百分位数。改进的泊松回归模型评估了与治疗暴露的关联,诊断时代,和慢性病。根据诊断年龄(≤1岁和>1岁)进行分层分析,以替代低危和高危疾病。
结果:幸存者(N=837;年龄中位数,25[17-58]年,诊断年龄,1[0-21]岁)与兄弟姐妹对照(N=728;年龄,32[16-43]年)。幸存者任务效率受损的风险较高(≤1年相对风险[RR],1.48;95%置信区间[CI],1.08-2.03;>1年RR,1.58;95%CI,1.22-2.06)和情绪调节(≤1年RR,1.51;95%CI,1.07-2.12;>1年RR,1.44;95%CI,1.06-1.95)。与铂暴露相关的任务效率受损(≤1年RR,1.74;95%CI,1.01-2.97),听力损失(≤1年RR,1.95;95%CI,1.26-3.00;>1年RR,1.56;95%CI,1.09-2.24),心血管(≤1年RR,1.83;95%CI,1.15-2.89;>1年RR,1.74;95%CI,1.12-2.69),神经系统(≤1年RR,2.00;95%CI,1.32-3.03;>1年RR,2.29;95%CI,1.64-3.21),和呼吸(>1年RR,2.35;95%CI,1.60-3.45)条件。幸存者≤1年;女性(RR,1.54;95%CI,1.02-2.33),心血管(RR,1.71;95%CI,1.08-2.70)和呼吸(RR,1.99;95%CI,1.14-3.49)与情绪调节受损相关的病症。幸存者不太可能全职工作(p<0.0001),研究生学院(p=.035),独立生活(p<0.0001)。
结论:神经母细胞瘤幸存者报告影响成人里程碑的神经认知障碍。确定的健康状况和治疗暴露可以有针对性地改善结果。
结论:神经母细胞瘤患者的生存率持续改善。缺乏有关神经母细胞瘤幸存者神经认知结果的信息;大多数研究检查了白血病或脑肿瘤的幸存者。在这项研究中,将837名儿童神经母细胞瘤的成年幸存者与儿童癌症幸存者研究的兄弟姐妹进行了比较。幸存者的注意力/处理速度(任务效率)和情绪反应性/挫折耐受性(情绪调节)的损害风险高50%。幸存者不太可能达到成年里程碑,例如独立生活。患有慢性健康状况的幸存者有更高的受损风险。早期识别和积极管理慢性病可能有助于减轻损伤水平。
Despite survival improvements, there is a paucity of data on
neurocognitive outcomes in neuroblastoma survivors. This
study addresses this literature gap.
Neurocognitive impairments in survivors were compared to sibling controls from the Childhood Cancer Survivor
Study (CCSS) using the CCSS
Neurocognitive Questionnaire. Impaired emotional regulation, organization, task efficiency, and memory defined as scores ≥90th percentile of sibling norms. Modified Poisson regression models evaluated associations with treatment exposures, era of diagnosis, and chronic conditions. Analyses were stratified by age at diagnosis (≤1 and >1 year) as proxy for lower versus higher risk disease.
Survivors (N = 837; median [range] age, 25 [17-58] years, age diagnosed, 1 [0-21] years) were compared to sibling controls (N = 728; age, 32 [16-43] years). Survivors had higher risk of impaired task efficiency (≤1 year relative risk [RR], 1.48; 95% confidence interval [CI], 1.08-2.03; >1 year RR, 1.58; 95% CI, 1.22-2.06) and emotional regulation (≤1 year RR, 1.51; 95% CI, 1.07-2.12; >1 year RR, 1.44; 95% CI, 1.06-1.95). Impaired task efficiency associated with platinum exposure (≤1 year RR, 1.74; 95% CI, 1.01-2.97), hearing loss (≤1 year RR, 1.95; 95% CI, 1.26-3.00; >1 year RR, 1.56; 95% CI, 1.09-2.24), cardiovascular (≤1 year RR, 1.83; 95% CI, 1.15-2.89; >1 year RR, 1.74; 95% CI, 1.12-2.69), neurologic (≤1 year RR, 2.00; 95% CI, 1.32-3.03; >1 year RR, 2.29; 95% CI, 1.64-3.21), and respiratory (>1 year RR, 2.35; 95% CI, 1.60-3.45) conditions. Survivors ≤1 year; female sex (RR, 1.54; 95% CI, 1.02-2.33), cardiovascular (RR, 1.71; 95% CI, 1.08-2.70) and respiratory (RR, 1.99; 95% CI, 1.14-3.49) conditions associated impaired emotional regulation. Survivors were less likely to be employed full-time (p < .0001), graduate college (p = .035), and live independently (p < .0001).
Neuroblastoma survivors report neurocognitive impairment impacting adult milestones. Identified health conditions and treatment exposures can be targeted to improve outcomes.
Survival rates continue to improve in patients with neuroblastoma. There is a lack of information regarding neurocognitive outcomes in neuroblastoma survivors; most studies examined survivors of leukemia or brain tumors. In this
study, 837 adult survivors of childhood neuroblastoma were compared to siblings from the Childhood Cancer Survivorship
Study. Survivors had a 50% higher risk of impairment with attention/processing speed (task efficiency) and emotional reactivity/frustration tolerance (emotional regulation). Survivors were less likely to reach adult milestones such as living independently. Survivors with chronic health conditions are at a higher risk of impairment. Early identification and aggressive management of chronic conditions may help mitigate the level of impairment.