中枢神经系统非生殖细胞性生殖细胞瘤（CNS-NGGCT）患者的治疗失败模式：临床试验的汇总分析。
Pattern of treatment failures in patients with central nervous system non-germinomatous germ cell tumors (CNS-NGGCT): A pooled analysis of clinical trials.
机构信息
The Hospital for Sick Children, University of Toronto, Toronto, Ontario, Canada.
Department of Oncology, Children's National Hospital, Washington, DC, USA.
出版信息
Neuro Oncol. 2022 Nov 2;24(11):1950-1961. doi: 10.1093/neuonc/noac057.
BACKGROUND
Central Nervous System Non-Germinomatous Germ Cell Tumors (CNS-NGGCT) are rare but curable tumors. Due to their rarity, patients with treatment failures remain a poorly characterized group with unfavorable outcomes. In this study, we sought to characterize patients with treatment failures in a large, prospectively treated cohort.
METHODS
European and North American clinical trials for patients with CNS-NGGCT (SIOP-GCT-96, SFOP-TGM-TC 90/92, COG-ACNS0122, and COG-ACNS1123) were pooled for analysis. Additionally, patients included and treated in the UK and France national registries under strict protocol guidelines were included as an independent, non-overlapping cohort.
RESULTS
A total of 118 patients experienced a treatment failure. Twenty-four patients had progressive disease during therapy, and additional 11 patients were diagnosed with growing teratoma syndrome (GTS). Patients with GTS are significantly younger and present with local failures and negative tumor markers. Eighty-three individuals experienced disease relapses after treatment ended. Patients' metastatic relapses presented significantly earlier than local relapses and were associated with tumor marker elevation (OR: 4.39; P = .026). In our analysis, focal or whole-ventricular radiation therapy was not associated with an increased risk of metastatic relapses.
CONCLUSIONS
Herein, we present the largest pooled dataset of prospectively treated patients with relapsed CNS-NGGCT. Our study identified younger age and negative tumor markers to be characteristic of GTS. Additionally, we elucidated that metastatic relapses occur earlier than local relapses are associated with elevated tumor markers and are not associated with the field of radiation therapy. These findings are of utmost importance for the planning of future clinical trials and the implementation of surveillance strategies in these patients.
背景
中枢神经系统非生殖细胞性生殖细胞瘤（CNS-NGGCT）较为罕见，但可治愈。由于其罕见性，治疗失败的患者仍是一组特征较差且预后不良的人群。在这项研究中，我们试图对大型前瞻性治疗队列中治疗失败的患者进行特征描述。
方法
对欧洲和北美的 CNS-NGGCT 患者临床试验（SIOP-GCT-96、SFOP-TGM-TC 90/92、COG-ACNS0122 和 COG-ACNS1123）进行了汇总分析。此外，还纳入了根据严格方案指南在英国和法国国家登记处接受治疗的独立、非重叠队列中的患者。
结果
共有 118 例患者经历了治疗失败。24 例患者在治疗过程中出现进展性疾病，另有 11 例患者被诊断为生长性细胞瘤综合征（GTS）。患有 GTS 的患者明显更年轻，表现为局部失败和肿瘤标志物阴性。83 例患者在治疗结束后出现疾病复发。患者的转移性复发明显早于局部复发，且与肿瘤标志物升高相关（OR：4.39；P =.026）。在我们的分析中，局部或全脑室放疗与转移性复发风险增加无关。
结论
在此，我们报告了最大的前瞻性治疗 CNS-NGGCT 患者复发数据集。本研究发现，年轻和肿瘤标志物阴性是 GTS 的特征。此外，我们阐明了转移性复发早于局部复发，与肿瘤标志物升高相关，与放疗野无关。这些发现对未来临床试验的规划和这些患者监测策略的实施具有重要意义。
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