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三十多年来诊断的儿童髓母细胞瘤成年幸存者的神经认知结果和功能独立性。

Neurocognitive outcomes and functional independence in adult survivors of childhood medulloblastoma diagnosed over three decades.

发表日期:2024 Jul 04
作者: Chiara Papini, Sedigheh Mirzaei S, Mengqi Xing, Ingrid Tonning Olsson, Ralph Salloum, Peter M K de Blank, Katharine R Lange, Tricia Z King, Deokumar Srivastava, Wendy M Leisenring, Rebecca M Howell, Kevin C Oeffinger, Leslie L Robison, Gregory T Armstrong, Kevin R Krull, Tara M Brinkman
来源: NEURO-ONCOLOGY

摘要:

儿童髓母细胞瘤的治疗已经发展到减少神经毒性,同时提高生存率。然而,不断发展的疗法对晚期神经认知结果和成人功能独立性的影响仍然未知。 儿童髓母细胞瘤的成年幸存者(n = 505;中位[最小-最大]年龄,29[18-46]岁)和兄弟姐妹对照(n = 727;32[18-58]岁)来自儿童癌症幸存者研究完成了评估神经认知问题和慢性健康状况(CHC)的调查。治疗暴露分为历史治疗(颅脊髓照射 [CSI]≥30 Gy,无化疗)、标准风险(CSI>0 至 <30 Gy 化疗)和高风险(CSI≥30 Gy 化疗)治疗。潜在类别分析通过就业、独立生活、日常/个人护理需求协助、驾驶执照、婚姻/伴侣状况来确定功能独立模式。多变量模型根据治疗暴露组估计了幸存者与兄弟姐妹的神经认知障碍风险,以及神经认知障碍、CHC 和功能独立性之间的关联。每个治疗暴露组的幸存者记忆力和任务效率受损的风险增加了 4 至 5 倍与兄弟姐妹相比。与历史疗法相比,当代基于风险的疗法并未降低风险。与 1970 年代接受治疗的幸存者相比,20 世纪 90 年代接受治疗的幸存者出现记忆障碍的风险更高(相对风险 [RR] 2.24,95% 置信区间 [CI] 1.39-3.60)。感觉运动、听力问题和癫痫发作分别与任务效率和记忆障碍风险升高 33%-34%、25-26% 和 21%-42% 相关。治疗相关的 CHC 和神经认知障碍与非独立性相关。尽管治疗发生了变化,儿童髓母细胞瘤的长期幸存者仍然面临神经认知障碍的风险,这与 CHC 相关。现代治疗方案后的神经认知监测势在必行。© 作者 2024。由牛津大学出版社代表神经肿瘤学会出版。版权所有。如需商业重复使用,请联系 reprints@oup.com 获取转载和转载的翻译权。所有其他权限都可以通过我们网站文章页面上的权限链接通过我们的 RightsLink 服务获得 - 如需更多信息,请联系journals.permissions@oup.com。
Treatment of childhood medulloblastoma has evolved to reduce neurotoxicity while improving survival. However, the impact of evolving therapies on late neurocognitive outcomes and adult functional independence remains unknown.Adult survivors of childhood medulloblastoma (n=505; median[minimum-maximum] age, 29[18-46] years) and sibling controls (n=727; 32[18-58] years) from the Childhood Cancer Survivor Study completed surveys assessing neurocognitive problems and chronic health conditions (CHCs). Treatment exposures were categorized as historical (craniospinal irradiation [CSI]≥30 Gy, no chemotherapy), standard-risk (CSI>0 to <30 Gy +chemotherapy) and high-risk (CSI≥30 Gy +chemotherapy) therapy. Latent class analysis identified patterns of functional independence using employment, independent living, assistance with routine/personal care needs, driver's license, marital/partner status. Multivariable models estimated risk of neurocognitive impairment in survivors versus siblings and by treatment exposure group, and associations between neurocognitive impairment, CHCs, and functional independence.Survivors in each treatment exposure group had 4- to 5-fold elevated risk of impaired memory and task efficiency compared to siblings. Contemporary risk-based therapies did not confer lower risk compared to historical therapy. Survivors treated in the 1990s had higher risk of memory impairment (relative risk [RR] 2.24, 95% confidence interval [CI] 1.39-3.60) compared to survivors treated in the 1970s. Sensorimotor, hearing problems and seizures were associated with 33%-34%, 25-26% and 21%-42% elevated risk of task efficiency and memory impairment, respectively. Treatment-related CHCs and neurocognitive impairment were associated with non-independence.Despite treatment changes, long-term survivors of childhood medulloblastoma remain at risk for neurocognitive impairment, which was associated with CHCs. Neurocognitive surveillance after contemporary regimens is imperative.© The Author(s) 2024. Published by Oxford University Press on behalf of the Society for Neuro-Oncology. All rights reserved. For commercial re-use, please contact reprints@oup.com for reprints and translation rights for reprints. All other permissions can be obtained through our RightsLink service via the Permissions link on the article page on our site—for further information please contact journals.permissions@oup.com.