高危神经母细胞瘤诱导后居里分数的验证：SIOPEN/HR-NBL1 上的儿童肿瘤学组和 SIOPEN 组报告
Validation of Postinduction Curie Scores in High-Risk Neuroblastoma: A Children's Oncology Group and SIOPEN Group Report on SIOPEN/HR-NBL1.
机构信息
Department of Pediatrics and Communicable Diseases, University of Michigan Medical Center, Ann Arbor, Michigan
Department of Radiology, University of Washington School of Medicine/Seattle Children's Hospital, Seattle, Washington.
出版信息
J Nucl Med. 2018 Mar;59(3):502-508. doi: 10.2967/jnumed.117.195883. Epub 2017 Sep 8.
A semiquantitative I-metaiodobenzylguanidine (I-MIBG) scoring method (the Curie score, or CS) was previously examined in the Children's Oncology Group (COG) high-risk neuroblastoma trial, COG A3973, with a postinduction CS of more than 2 being associated with poor event-free survival (EFS). The validation of the CS in an independent dataset, International Society of Paediatric Oncology European Neuroblastoma/High-Risk Neuroblastoma 1 (SIOPEN/HR-NBL1), is now reported. A retrospective analysis of I-MIBG scans obtained from patients who had been prospectively enrolled in SIOPEN/HR-NBL1 was performed. All patients exhibited I-MIBG-avid, International Neuroblastoma Staging System stage 4 neuroblastoma. I-MIBG scans were evaluated at 2 time points, diagnosis ( = 345) and postinduction ( = 330), before consolidation myeloablative therapy. Scans of 10 anatomic regions were evaluated, with each region being scored 0-3 on the basis of disease extent and a cumulative CS generated. Cut points for outcome analysis were identified by Youden methodology. CSs from patients enrolled in COG A3973 were used for comparison. The optimal cut point for CS at diagnosis was 12 in SIOPEN/HR-NBL1, with a significant outcome difference by CS noted (5-y EFS, 43.0% ± 5.7% [CS ≤ 12] vs. 21.4% ± 3.6% [CS > 12], < 0.0001). The optimal CS cut point after induction was 2 in SIOPEN/HR-NBL1, with a postinduction CS of more than 2 being associated with an inferior outcome (5-y EFS, 39.2% ± 4.7% [CS ≤ 2] vs. 16.4% ± 4.2% [CS > 2], < 0.0001). The postinduction CS maintained independent statistical significance in Cox models when adjusted for the covariates of age and gene copy number. The prognostic significance of postinduction CSs has now been validated in an independent cohort of patients (SIOPEN/HR-NBL1), with a postinduction CS of more than 2 being associated with an inferior outcome in 2 independent large, cooperative group trials.
一种半定量的碘-131 间碘苄胍（I-MIBG）评分方法（居里评分，CS）先前已在儿童肿瘤学组（COG）高危神经母细胞瘤试验 COG A3973 中进行了检验，诱导后 CS 超过 2 与不良无事件生存（EFS）相关。现将 CS 在独立数据集国际儿科肿瘤学会欧洲神经母细胞瘤/高危神经母细胞瘤 1（SIOPEN/HR-NBL1）中的验证结果报告如下。对前瞻性纳入 SIOPEN/HR-NBL1 的患者的 I-MIBG 扫描进行了回顾性分析。所有患者均表现为 I-MIBG 摄取阳性、国际神经母细胞瘤分期系统（INSS）分期 4 期神经母细胞瘤。在巩固性清髓性化疗前，对 I-MIBG 扫描进行了 2 个时间点的评估，即诊断时（=345）和诱导后（=330）。对 10 个解剖区域的扫描进行了评估，每个区域根据疾病范围和累积 CS 进行 0-3 分评分。采用 Youden 方法确定用于结果分析的 CS 切点。使用来自 COG A3973 的患者的 CS 进行比较。在 SIOPEN/HR-NBL1 中，CS 在诊断时的最佳切点为 12，CS 差异有显著的结果意义（5 年 EFS，CS≤12 组为 43.0%±5.7%，CS>12 组为 21.4%±3.6%，<0.0001）。在 SIOPEN/HR-NBL1 中，诱导后 CS 的最佳切点为 2，诱导后 CS 超过 2 与不良结局相关（5 年 EFS，CS≤2 组为 39.2%±4.7%，CS>2 组为 16.4%±4.2%，<0.0001）。当按年龄和基因拷贝数调整协变量时，诱导后 CS 在 Cox 模型中仍具有独立的统计学意义。诱导后 CS 在两个独立的大型合作组试验中的预后意义已在独立患者队列（SIOPEN/HR-NBL1）中得到验证，诱导后 CS 超过 2 与不良结局相关。
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