Secondary malignant neoplasms after high-dose chemotherapy and autologous stem cell rescue for high-risk neuroblastoma
Secondary malignant neoplasms after high-dose chemotherapy and autologous stem cell rescue for high-risk neuroblastoma
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DOI：
10.1002/pbc.25033
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发表时间：
2014-08-01
影响因子：
3.2
通讯作者：
Hijiya, Nobuko
中科院分区：
文献类型：
--
作者：
Martin, Alissa;Schneiderman, Jennifer;Hijiya, Nobuko
背景 高危神经母细胞瘤的结果仍然很差。现代治疗方案采用强烈诱导，然后进行清髓性巩固化疗和自体干细胞拯救 (ASCR)，提高了生存率，但长期后遗症，包括继发性恶性肿瘤 (SMN) 的发展，现在才刚刚出现。方法 我们回顾性回顾了 87 名高危神经母细胞瘤患者的数据，这些患者在 1991 年 1 月至 2011 年 7 月期间接受强化诱导化疗，随后接受 ASCR，遵循两个机构方案之一：芝加哥试点 1 (CP1；n=12) 和芝加哥试点 2 (CP2；n=75)。结果 所有 87 名患者的 15 年总生存率为 33.9%（95% 置信区间 [CI]，23.1-45.0%）。 SMN 的 10 年和 15 年累计发生率分别为 16.5%（95%CI，7.2-38.0%）和 34.2%（95%CI，18.6-63.1%），没有证据表明 15 年出现平台期。 10 名发生 SMN 的患者中，有 6 名（CP1 中 n=2，CP2 中 n=8）患有血液系统恶性肿瘤，包括急性髓系白血病 (AML)/骨髓增生异常综合征 (MDS)。实体瘤包括甲状腺乳头状癌、软骨肉瘤、肝细胞癌和胆道腺癌。结论 与较早的神经母细胞瘤研究相比，在该队列中观察到 SMN 的发病率显着较高，尤其是血液恶性肿瘤，这可能是由于这些患者接触表鬼臼毒素和接受高累积剂量的烷化剂。随着生存时间的延长，发生 SMN 的风险持续增加，并且在 15 年时并未达到稳定水平。尽管患者数量相对较少，但我们的研究强调需要对接受现代疗法治疗的幸存者进行终身随访。儿科血癌2014； 61：1350-1356。 (c) 2014 年 Wiley 期刊公司。
Background Outcomes for high-risk neuroblastoma remain poor. Modern treatment protocols utilizing intense induction followed by myeloablative consolidation chemotherapy with autologous stem cell rescue (ASCR) have improved survival rates, but the long-term sequelae, including development of secondary malignant neoplasms (SMN), are just now surfacing. Methods We retrospectively reviewed data from 87 patients with high-risk neuroblastoma who were treated with intensive induction chemotherapy followed by ASCR between January 1991 and July 2011 following one of two institutional protocols: Chicago Pilot 1 (CP1; n=12) and Chicago Pilot 2 (CP2; n=75). Results The 15-year overall survival rate for all 87 patients was 33.9% (95% confidence interval [CI], 23.1-45.0%). The 10- and 15-year cumulative incidence of SMN was 16.5% (95%CI, 7.2-38.0%) and 34.2% (95%CI, 18.6-63.1%), respectively, without evidence of a plateau at 15 years. Six of the 10 patients (n=2 in CP1 and n=8 in CP2) who developed SMN had hematologic malignancies including acute myeloid leukemia (AML)/myelodysplastic syndrome (MDS). Solid tumors included thyroid papillary carcinoma, chondrosarcoma, hepatocellular carcinoma, and biliary adenocarcinoma. Conclusion A significantly higher incidence of SMN, especially hematological malignancies, was observed in this cohort compared to older neuroblastoma studies, potentially due to exposure to epipodophyllotoxins and a high cumulative dose of alkylating agents these patients received. The risk of developing an SMN continued to increase with survival time and did not reach the plateau at 15 years. Although the number of the patients is relatively small, our study emphasizes the need for life-long follow-up of survivors who were treated using modern therapy. Pediatr Blood Cancer 2014; 61:1350-1356. (c) 2014 Wiley Periodicals, Inc.