加拿大安大略省儿童室管膜瘤幸存者的医疗与功能负担：一项基于人群的研究
The medical and functional burden of surviving childhood ependymoma: A population-based study in Ontario, Canada
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影响因子:2.3
分区:医学3区 / 血液学3区 儿科3区 肿瘤学4区
发表日期:2024 Nov
作者:
Hallie Coltin, Priscila Pequeno, Ning Liu, Derek S Tsang, Sumit Gupta, Michael D Taylor, Eric Bouffet, Vijay Ramaswamy, Paul C Nathan
DOI:
10.1002/pbc.31275
摘要
目前关于儿童室管膜瘤幸存者的晚期效应负担的研究较少。为弥补这一空白，我们利用真实世界的医疗服务数据，建立了一个基于人群的室管膜瘤幸存者队列，评估相关的后遗症。研究对象为1987年至2015年在加拿大安大略省诊断的未满18岁的室管膜瘤患者，且从其最新的儿童癌症事件起存活至少5年（索引日期），并以1：5比例匹配人口对照。通过与省级医疗服务数据的连接，比较幸存者与对照组在多种医疗和功能性结局的累积发生率。结果显示，在96名幸存者中，77.1%接受了放疗，9.4%接受了顺铂治疗。10年后，幸存者的全因死亡风险显著高于对照（7.1%比0.3%；p=0.0002），非孕产期住院率为45.1%对10.6%；p<0.0001)，中风发生率为6.5%对0%；p<0.0001)，严重听力损失需辅助设备者为7.5%对0%；p<0.0001)，家庭护理服务为27.6%对7.7%；p<0.0001)，领取残疾支持申诉的比例为24.0%对5.4%；p<0.0001)。儿童室管膜瘤幸存者极易出现严重的晚期后遗症，包括死亡、中风、严重听力丧失和残疾。亟需改进风险分层策略，减少毒性治疗的暴露，制定干预措施以预防或减少晚期后遗症的发生，从而优化幸存者的长期健康。
Abstract
Few studies have characterized the burden of late effects among childhood ependymoma survivors. To address this gap, we examined these sequelae using real-world health services data in a population-based ependymoma survivor cohort.All individuals younger than 18 years diagnosed with an ependymoma in Ontario, Canada between 1987 and 2015 who had survived at least 5 years from their latest pediatric cancer event (index date) were matched 1:5 with population controls. Following linkage with provincial health services data, the cumulative incidences of multiple medical and functional outcomes between survivors and controls were compared.Among 96 survivors, 77.1% had been irradiated and 9.4% had received cisplatin. At 10 years post-index, survivors were at significantly higher risk of all-cause mortality (7.1%, 95% confidence interval [CI]: 1.0-13.3 vs. 0.3%, 95% CI: 0.0-1.0; p = .0002), non-obstetric hospitalization (45.1%, 95% CI: 32.6-56.7 vs. 10.6%, 95% CI: 7.6-14.1; p < .0001), stroke (6.5%, 95% CI: 2.3-13.7 vs. 0%; p < .0001), severe hearing loss requiring an amplification device (7.5%, 95% CI: 2.7-15.7 vs. 0%; p < .0001), receiving homecare service (27.6%, 95% CI: 18.5-37.5 vs. 7.7%, 95% CI: 5.3-10.7; p < .0001), and submitting a disability support prescription claim (24.0%, 95% CI: 14.8-34.3 vs. 5.4%, 95% CI: 3.5-7.8; p < .0001) compared to controls.Pediatric ependymoma survivors are highly vulnerable to severe late sequelae, including death, stroke, severe hearing loss, and disability. Urgent efforts are needed to improve risk-stratification approaches that mitigate exposure to toxic therapies for children with lower risk disease. Interventions to prevent or decrease the risk of developing late sequelae are critical to optimizing survivor long-term health.