Long-Term Risk of Subsequent Neoplasms in 5-Year Survivors of Childhood Neuroblastoma: A Dutch Childhood Cancer Survivor Study-LATER 3 Study.

IF 42.1 1区医学 Q1 ONCOLOGY

Journal of Clinical Oncology Pub Date : 2025-01-10 Epub Date: 2024-10-02 DOI:10.1200/JCO.23.01430

Aimée S R Westerveld, Godelieve A M Tytgat, Hanneke M van Santen, Max M van Noesel, Jacqueline Loonen, Andrica C H de Vries, Marloes Louwerens, Maria M W Koopman, Margriet van der Heiden-van der Loo, Geert O Janssens, Ronald R de Krijger, Cecile M Ronckers, Helena J H van der Pal, Leontien C M Kremer, Jop C Teepen

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引用次数: 0

Abstract

Purpose: Neuroblastoma survivors have an increased risk of developing subsequent malignant neoplasms (SMNs), but the risk of subsequent nonmalignant neoplasms (SNMNs) and risk factors are largely unknown. We analyzed the long-term risks and associated risk factors for developing SMNs and SNMNs in a well-characterized cohort of 5-year neuroblastoma survivors.

Methods: We included 563 5-year neuroblastoma survivors from the Dutch Childhood Cancer Survivor Study (DCCSS)-LATER cohort, diagnosed during 1963-2014. Subsequent neoplasms were ascertained by linkages with the Netherlands Cancer Registry and the Dutch Nationwide Pathology Databank (Palga) and medical chart review. We calculated standardized incidence ratios (SIRs), absolute excess risk (AER), and cumulative incidences. Multivariable competing risk regression analysis was used to evaluate risk factors.

Results: In total, 23 survivors developed an SMN and 60 an SNMN. After a median follow-up of 23.7 (range, 5.0-56.3) years, the risk of SMN was elevated compared with the general population (SIR, 4.0; 95% CI, 2.5 to 5.9; AER per 10,000 person-years, 15.1). The 30-year cumulative incidence was 3.4% (95% CI, 1.9 to 6.0) for SMNs and 10.4% (95% CI, 7.3 to 14.8) for SNMNs. Six survivors developed an SMN after iodine-metaiodobenzylguanidine (^131IMIBG) treatment. Survivors treated with ^131IMIBG had a higher risk of developing SMNs (subdistribution hazard ratio [SHR], 5.7; 95% CI, 1.8 to 17.8) and SNMNs (SHR, 2.6; 95% CI, 1.2 to 5.6) compared with survivors treated without ^131IMIBG; results for SMNs were attenuated in high-risk patients only (SMNs SHR, 3.6; 95% CI, 0.9 to 15.3; SNMNs SHR, 1.5; 95% CI, 0.7 to 3.6).

Conclusion: Our results demonstrate that neuroblastoma survivors have an elevated risk of developing SMNs and a high risk of SNMNs. ^131IMIBG may be a treatment-related risk factor for the development of SMN and SNMN, which needs further validation. Our results emphasize the need for awareness of subsequent neoplasms and the importance of follow-up care.

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儿童神经母细胞瘤 5 年存活者罹患后续肿瘤的长期风险：荷兰儿童癌症存活者研究--LATER 3 研究。

目的：神经母细胞瘤幸存者罹患后续恶性肿瘤（SMNs）的风险会增加，但罹患后续非恶性肿瘤（SNMNs）的风险和风险因素在很大程度上是未知的。我们分析了神经母细胞瘤5年存活者队列中罹患SMNs和SNMNs的长期风险和相关风险因素：我们纳入了荷兰儿童癌症幸存者研究（DCCSS）-LATER队列中的563名5年期神经母细胞瘤幸存者，他们在1963-2014年期间确诊。通过与荷兰癌症登记处和荷兰全国病理数据库（Palga）的联系以及病历审查，确定了其后的肿瘤情况。我们计算了标准化发病率（SIR）、绝对超额风险（AER）和累积发病率。多变量竞争风险回归分析用于评估风险因素：共有 23 名幸存者患上了 SMN，60 名幸存者患上了 SNMN。中位随访 23.7 年（5.0-56.3 年）后，SMN 的风险高于普通人群（SIR，4.0；95% CI，2.5-5.9；每 10,000 人年的 AER，15.1）。SMN的30年累计发病率为3.4%（95% CI，1.9至6.0），SNMN的30年累计发病率为10.4%（95% CI，7.3至14.8）。6名幸存者在接受碘-甲碘苄基胍（131IMIBG）治疗后出现了SMN。与接受 131IMIBG 治疗的幸存者相比，接受 131IMIBG 治疗的幸存者罹患 SMN（亚分布危险比 [SHR]，5.7；95% CI，1.8 至 17.8）和 SNMN（SHR，2.6；95% CI，1.2 至 5.6）的风险更高。与未接受131IMIBG治疗的幸存者相比，SMNs的结果有所降低（SMNs SHR，3.6；95% CI，0.9至15.3；SNMNs SHR，1.5；95% CI，0.7至3.6）：我们的研究结果表明，神经母细胞瘤幸存者罹患SMNs的风险较高，而罹患SNMNs的风险较高。131IMIBG可能是发生SMN和SNMN的治疗相关风险因素，这需要进一步验证。我们的研究结果强调了对后续肿瘤的认识以及后续护理的重要性。

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来源期刊

Journal of Clinical Oncology 医学-肿瘤学

CiteScore

41.20

自引率

2.20%

发文量

8215

审稿时长

2 months

期刊介绍： The Journal of Clinical Oncology serves its readers as the single most credible, authoritative resource for disseminating significant clinical oncology research. In print and in electronic format, JCO strives to publish the highest quality articles dedicated to clinical research. Original Reports remain the focus of JCO, but this scientific communication is enhanced by appropriately selected Editorials, Commentaries, Reviews, and other work that relate to the care of patients with cancer.