Raleigh David R, Tomlin Bryan, Buono Benedict Del, Roddy Erika, Sear Katherine, Byer Lennox, Felton Erin, Banerjee Anu, Torkildson Joseph, Samuel David, Horn Biljana, Braunstein Steve E, Haas-Kogan Daphne A, Mueller Sabine
Department of Radiation Oncology, University of California San Francisco, San Francisco, CA, USA.
Department of Economics, California State University Chanel Islands, Camarillo, CA, USA.
J Neurooncol. 2017 Jan;131(2):359-368. doi: 10.1007/s11060-016-2307-6. Epub 2016 Oct 24.
Pediatric embryonal brain tumor patients treated with craniospinal irradiation (CSI) are at risk for adverse effects, with greater severity in younger patients. Here we compare outcomes of CSI vs. high-dose chemotherapy (HD), stem cell transplant (SCT) and delayed CSI in newly diagnosed patients. Two hundred one consecutive patients treated for medulloblastoma (72 %), supratentorial primitive neuroectodermal tumor (sPNET; 18 %) or pineoblastoma (10 %) at two institutions between 1988 and 2014 were retrospectively identified. Progression free survival (PFS) and overall survival (OS) were estimated using the Kaplan-Meier method and compared by log-rank tests. Adjuvant CSI regimens were used for 56 % of patients (upfront-CSI), and HD/SCT regimens were used in 32 % of patients. HD/SCT patients were significantly younger than those receiving upfront-CSI (2.9 vs. 7.8 years; P < 0.0001). There were no differences in metastases, extent of resection, or CSI dose between upfront-CSI and HD/SCT patients, but median follow-up was shorter in the HD/SCT group (6.2 vs. 3.9 years; P = 0.007). There were no significant outcome differences between upfront-CSI and HD/SCT patients who received CSI as a prophylaxis or following relapse (OS 66 % vs. 61 %, P = 0.13; PFS 67 % vs. 62 %, P = 0.12). Outcomes were equivalent when restricting analyses to HD/SCT patients who received prophylactic CSI prior to relapse (OS 66 % vs. 65 %, P = 0.5; PFS 67 % vs. 74 %, P = 0.8). At last follow-up, 48 % of HD/SCT patients had received neither definitive nor salvage radiotherapy. In this retrospective cohort, outcomes with adjuvant HD/SCT followed by delayed CSI are comparable to upfront-CSI for carefully surveyed pediatric embryonal brain tumor patients. Future prospective studies are required to validate this finding, and also to assess the impact of delayed CSI on neurocognitive outcomes.
接受颅脊髓照射(CSI)治疗的小儿胚胎性脑肿瘤患者存在不良反应风险,年龄越小,严重程度越高。在此,我们比较了新诊断患者中CSI与大剂量化疗(HD)、干细胞移植(SCT)以及延迟CSI的治疗效果。回顾性分析了1988年至2014年间在两家机构接受治疗的201例连续患者,这些患者分别患有髓母细胞瘤(72%)、幕上原始神经外胚层肿瘤(sPNET;18%)或松果体母细胞瘤(10%)。采用Kaplan-Meier方法估计无进展生存期(PFS)和总生存期(OS),并通过对数秩检验进行比较。56%的患者采用辅助CSI方案( upfront-CSI),32%的患者采用HD/SCT方案。接受HD/SCT的患者明显比接受 upfront-CSI的患者年龄小(2.9岁对7.8岁;P < 0.0001)。 upfront-CSI组和HD/SCT组在转移情况、切除范围或CSI剂量方面无差异,但HD/SCT组的中位随访时间较短(6.2年对3.9年;P = 0.007)。接受CSI作为预防或复发后治疗的 upfront-CSI组和HD/SCT组患者在治疗效果上无显著差异(OS分别为66%对61%,P = 0.13;PFS分别为67%对62%,P = 0.12)。当将分析限制在复发前接受预防性CSI的HD/SCT患者时,治疗效果相当(OS分别为66%对65%,P = 0.5;PFS分别为67%对74%,P = 0.8)。在最后一次随访时,48%的HD/SCT患者既未接受确定性放疗也未接受挽救性放疗。在这个回顾性队列中,对于经过仔细评估的小儿胚胎性脑肿瘤患者,辅助HD/SCT后延迟CSI的治疗效果与 upfront-CSI相当。未来需要进行前瞻性研究来验证这一发现,并评估延迟CSI对神经认知结果的影响。