Subsequent Malignant Neoplasms in Pediatric Patients Initially Diagnosed With Neuroblastoma
Subsequent Malignant Neoplasms in Pediatric Patients Initially Diagnosed With Neuroblastoma
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DOI：
10.1097/mph.0000000000000148
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发表时间：
2015-01-01
影响因子：
1.2
通讯作者：
McGregor, Lisa M.
中科院分区：
文献类型：
--
作者：
Federico, Sara M.;Allewelt, Heather B.;McGregor, Lisa M.
背景：大多数先前评估神经母细胞瘤患者后续恶性肿瘤（SMN）的研究仅限于长期幸存者和/或他们的治疗暴露。本研究调查了我们机构诊断为神经母细胞瘤的患者的 SMN。方法：回顾了 1961 年至 2005 年间在圣犹达儿童研究医院接受神经母细胞瘤治疗的 646 名患者的记录。分析 SMN 患者的数据，计算 20 年和 30 年 SMN 累积发生率以及标准化发生率。结果：21 名患者患有 SMN。 SMN 20 年和 30 年累积发生率分别为 2.6%+/-0.7% 和 4.6%+/-1.1%。标准化发病率为 8.3（95% 置信区间，5.0-13.0）。 5 名患者在诊断后 5 年内出现 SMN。急性髓系白血病/骨髓增生异常综合征 (n=4)、肉瘤 (n=7) 和癌 (n=5) 的中位潜伏期分别为 3.6、9 和 24.2 年。 9 名患者死于 SMN，其中包括所有急性髓系白血病/骨髓增生异常综合征患者。结论：神经母细胞瘤患者继发性肿瘤的风险增加。风险适应疗法的修改可能会改变受影响的患者群体和 SMN 的发生率。未来的研究有必要将 SMN 与治疗暴露联系起来，并评估诊断后 30 年后 SMN 的风险。
Background: Most prior studies evaluating subsequent malignant neoplasms (SMNs) in patients with neuroblastoma are restricted to long-term survivors and/or their treatment exposures. This study investigates SMNs in patients diagnosed with neuroblastoma at our institution.Methods: Records of 646 patients treated for neuroblastoma at St Jude Children's Research Hospital between 1961 and 2005 were reviewed. Data from patients with SMNs were analyzed and the 20- and 30-year cumulative incidence of SMNs and standardized incidence ratio were calculated.Results: Twenty-one patients had a SMN. The 20-and 30-year cumulative incidences of a SMN were 2.6%+/-0.7% and 4.6%+/-1.1%, respectively. The standardized incidence ratio was 8.3 (95% confidence interval, 5.0-13.0). Five patients developed a SMN within 5 years from diagnosis. The median latency for the development of acute myeloid leukemia/myelodysplastic syndrome (n=4), sarcomas (n=7), and carcinomas (n=5) were 3.6, 9, and 24.2 years, respectively. Nine patients died from their SMN, including all with acute myeloid leukemia/myelodysplastic syndrome.Conclusions: Patients with neuroblastoma have an increased risk of secondary neoplasia. Modification of risk-adapted therapies will likely alter the affected patient population and the incidence of SMNs. Future studies are necessary to link SMNs to treatment exposures and to evaluate the risk of SMNs beyond 30 years from diagnosis.