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中枢神经系统肿瘤患儿的重度听力损失:加拿大青少年癌症人群(CYP-C)报告

Severe Hearing Loss in Children With Central Nervous System Tumors: A Population-Based Cancer in Young People in Canada (CYP-C) Report.

作者信息

Coltin Hallie, Trudeau-Ferrin Ofélie, Perreault Sébastien, Lafay-Cousin Lucie, Tsang Derek S, Renzi Samuele, Larouche Valérie, Erker Craig, Hukin Juliette, Barber Randy, Nathan Paul C, Huang Annie, Bouffet Eric, Ramaswamy Vijay

机构信息

Division of Hematology-Oncology, CHU Sainte-Justine, Montreal, Quebec, Canada.

CHU Sainte-Justine Research Center, Montreal, Quebec, Canada.

出版信息

Pediatr Blood Cancer. 2025 Nov;72(11):e32024. doi: 10.1002/pbc.32024. Epub 2025 Aug 29.

DOI:10.1002/pbc.32024
PMID:40879078
Abstract

PURPOSE

Children with central nervous system (CNS) tumors are prone to treatment-related hearing loss (HL) and subsequent functional impairment. This study reports a dedicated population-based analysis of CNS tumor-specific rates and predictors of early severe HL.

METHODS

A cohort study of children ≤15 years diagnosed with CNS tumors between 2001 and 2019 through the Cancer in Young People in Canada (CYP-C) program. The primary outcome was Grade 3 and 4 severe HL within 5 years following diagnosis.

RESULTS

Among 3201 children with CNS tumors, 5.1% experienced early severe HL. Children with medulloblastoma (N = 570) and ATRT/other embryonal tumors (N = 269) had higher rates of early HL (16.1%, 15.2%, respectively). Cisplatin was administered to 80.1% of children with embryonal tumors, and 67.3% received radiotherapy. In children with medulloblastoma, age less than 6 years at diagnosis (OR 2.4, 1.5-3.8; vs. ≥6 years), radiation (OR 3.5, 1.6-7.6), and cisplatin (OR 20.4, 1.3-329.7) predicted early severe HL. Younger age at diagnosis doubled the probability of early severe HL (10.6% in <6 years vs. 4.8% in ≥6 years), while radiation exposure tripled the probability across age groups (29.8% and 10.6% if <6 years; 15.3% and 4.8% in ≥6 years). In children with ATRT/other embryonal tumors, cisplatin (OR 31.6, 1.9-521.9) was the sole predictor of early severe HL.

CONCLUSIONS

High rates of early HL were observed in children with embryonal tumors. Younger children who received radiotherapy had higher probabilities of early HL, suggesting an additive interaction between age and radiation. Standardized otoprotection and research on cisplatin avoidance and therapy de-escalation in young children with embryonal tumors are urgently needed.

摘要

目的

中枢神经系统(CNS)肿瘤患儿易发生与治疗相关的听力损失(HL)及随后的功能损害。本研究报告了一项基于人群的专门分析,涉及CNS肿瘤特异性HL发生率及早期严重HL的预测因素。

方法

一项队列研究,研究对象为2001年至2019年间通过加拿大青少年癌症(CYP-C)项目确诊为CNS肿瘤的15岁及以下儿童。主要结局为诊断后5年内发生的3级和4级严重HL。

结果

在3201例CNS肿瘤患儿中,5.1%发生了早期严重HL。髓母细胞瘤患儿(n = 570)和非典型畸胎瘤/横纹肌样瘤(ATRT)/其他胚胎性肿瘤患儿(n = 269)的早期HL发生率更高(分别为16.1%和15.2%)。80.1%的胚胎性肿瘤患儿接受了顺铂治疗,67.3%接受了放射治疗。在髓母细胞瘤患儿中,诊断时年龄小于6岁(比值比[OR] 2.4,1.5 - 3.8;与≥6岁相比)、放射治疗(OR 3.5,1.6 - 7.6)和顺铂治疗(OR 20.4,1.3 - 329.7)可预测早期严重HL。诊断时年龄较小使早期严重HL的发生概率增加一倍(<6岁者为10.6%,≥6岁者为4.8%),而放射暴露使各年龄组的发生概率增加两倍(<6岁者为29.8%和10.6%;≥6岁者为15.3%和4.8%)。在ATRT/其他胚胎性肿瘤患儿中,顺铂(OR 31.6,1.9 - 521.9)是早期严重HL的唯一预测因素。

结论

胚胎性肿瘤患儿的早期HL发生率较高。接受放射治疗的年幼患儿早期HL发生概率更高,提示年龄与放射治疗之间存在相加性相互作用。迫切需要对胚胎性肿瘤患儿进行标准化的耳保护以及关于避免顺铂治疗和降低治疗强度的研究。

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