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儿童髓母细胞瘤/原始神经外胚层肿瘤成年幸存者的长期神经健康和心理社会功能:来自儿童癌症幸存者研究的报告

Long-term neurologic health and psychosocial function of adult survivors of childhood medulloblastoma/PNET: a report from the Childhood Cancer Survivor Study.

作者信息

King Allison A, Seidel Kristy, Di Chongzhi, Leisenring Wendy M, Perkins Stephanie Mabry, Krull Kevin R, Sklar Charles A, Green Daniel M, Armstrong Gregory T, Zeltzer Lonnie K, Wells Elizabeth, Stovall Marilyn, Ullrich Nicole J, Oeffinger Kevin C, Robison Leslie L, Packer Roger J

机构信息

Washington University School of Medicine, Siteman Cancer Center, St. Louis Children's Hospital, and Barnes Jewish Hospital, St. Louis, Missouri, USA.

Fred Hutchinson Cancer Research Center, Seattle, Washington, USA.

出版信息

Neuro Oncol. 2017 May 1;19(5):689-698. doi: 10.1093/neuonc/now242.

DOI:10.1093/neuonc/now242
PMID:28039368
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC5464442/
Abstract

BACKGROUND

Medulloblastoma is the most common malignant childhood brain tumor, although long-term risks for chronic neurologic health and psychosocial functioning in aging adult survivors are incompletely characterized.

METHODS

The Childhood Cancer Survivor Study (CCSS) includes 380 five-year survivors of medulloblastoma/primitive neuroectodermal tumor (PNET; median age at follow-up: 30 y, interquartile range 24-36) and sibling comparison (n = 4031). Cumulative incidence of neurologic health conditions was reported. Cox regression models provided hazard ratios (HRs) and 95% CIs. Cross-sectional outcomes were assessed using generalized linear models.

RESULTS

Compared with siblings, survivors were at increased risk of late-onset hearing loss (HR: 36.0, 95% CI: 23.6-54.9), stroke (HR: 33.9, 95% CI: 17.8-64.7), seizure (HR: 12.8, 95% CI: 9.0-18.1), poor balance (HR: 10.4, 95% CI: 6.7-15.9), tinnitus (HR: 4.8, 95% CI: 3.5-6.8), and cataracts (HR: 31.8, 95% CI: 16.7-60.5). Temporal/frontal lobe radiotherapy of 50 Gy or more increased risk for hearing loss (HR: 1.9, 95% CI: 1.1-1.3), seizure (HR: 2.1, 95% CI: 1.1-3.9), stroke (HR: 3.5, 95% CI: 1.3-9.1), and tinnitus (HR: 2.0, 95% CI: 1.0-3.9). Survivors were less likely than siblings to earn a college degree (relative risk [RR]: 0.49, 95% CI: 0.39-0.60), marry (RR: 0.35, 95% CI: 0.29-0.42), and live independently (RR: 0.58, 95% CI: 0.52-0.66).

CONCLUSIONS

Adult survivors of childhood medulloblastoma/PNET demonstrate pronounced risk for hearing impairment, stroke, lower educational attainment, and social independence. Interventions to support survivors should be a high priority.

摘要

背景

髓母细胞瘤是儿童最常见的恶性脑肿瘤,然而成年幸存者慢性神经健康和心理社会功能的长期风险尚未完全明确。

方法

儿童癌症幸存者研究(CCSS)纳入了380例髓母细胞瘤/原始神经外胚层肿瘤(PNET)的五年幸存者(随访时的中位年龄:30岁,四分位间距24 - 36岁),并与同胞进行比较(n = 4031)。报告了神经健康状况的累积发病率。Cox回归模型提供风险比(HRs)和95%置信区间(CIs)。使用广义线性模型评估横断面结果。

结果

与同胞相比,幸存者发生迟发性听力损失(HR:36.0,95% CI:23.6 - 54.9)、中风(HR:33.9,95% CI:17.8 - 64.7)、癫痫发作(HR:12.8,95% CI:9.0 - 18.1)、平衡能力差(HR:10.4,95% CI:6.7 - 15.9)、耳鸣(HR:4.8,95% CI:3.5 - 6.8)和白内障(HR:31.8,95% CI:16.7 - 60.5)的风险增加。50 Gy或更高剂量的颞叶/额叶放疗会增加听力损失(HR:1.9,95% CI:1.1 - 1.3)、癫痫发作(HR:2.1,95% CI:1.1 - 3.9)、中风(HR:3.5,95% CI:1.3 - 9.1)和耳鸣(HR:2.0,95% CI:1.0 - 3.9)的风险。与同胞相比,幸存者获得大学学位的可能性较小(相对风险[RR]:0.49,95% CI:0.39 - 0.60)、结婚的可能性较小(RR:0.35,95% CI:0.29 - 0.42)以及独立生活的可能性较小(RR:0.58,95% CI:0.52 - 0.66)。

结论

儿童髓母细胞瘤/PNET的成年幸存者表现出明显的听力障碍、中风、较低的教育程度和社会独立性风险。支持幸存者的干预措施应成为高度优先事项。

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