Lester Rachael A, Brown Lindsay C, Eckel Laurence J, Foote Robert T, NageswaraRao Amulya A, Buckner Jan C, Parney Ian F, Wetjen Nicholas M, Laack Nadia N
Mayo Medical School, Mayo Clinic College of Medicine, Rochester, MN, USA.
J Neurooncol. 2014 Nov;120(2):371-9. doi: 10.1007/s11060-014-1561-8. Epub 2014 Aug 13.
Central nervous system primitive neuroectodermal tumors (CNS PNETs) predominantly occur in children and rarely in adults. Because of the rarity of this tumor, its outcomes and prognostic variables are not well characterized. The purpose of this study was to evaluate clinical outcomes and prognostic factors for children and adults with CNS PNET. The records of 26 patients (11 children and 15 adults) with CNS PNET from 1991 to 2011 were reviewed retrospectively. Disease-free survival (DFS) and overall survival (OS) were estimated with the Kaplan-Meier method, and relevant prognostic factors were analyzed. For the cohort, both the 5-year DFS and the OS were 46 %. For pediatric patients, the 5-year DFS was 78 %; for adult patients, it was 22 % (P = 0.004). Five-year OS for the pediatric and adult patients was 67 and 33 %, respectively (P = 0.07). With bivariate analysis including chemotherapy regimen (high dose vs. standard vs. nonstandard) or risk stratification (standard vs. high) and age, the increased risk of disease recurrence in adults persisted. A nonsignificant tendency toward poorer OS in adult patients relative to pediatric patients also persisted. High-dose chemotherapy with stem cell rescue was associated with a statistically significant improvement in OS and a tendency toward improved DFS, although the findings were mitigated when the effect of age was considered. Local recurrence was the primary pattern of treatment failure in both adults and children. Our results suggest that adult patients with CNS PNETs have inferior outcomes relative to the pediatric cohort. Further research is needed to improve outcomes for CNS PNET in populations of all ages.
中枢神经系统原始神经外胚层肿瘤(CNS PNETs)主要发生于儿童,成人中罕见。由于该肿瘤罕见,其预后及预后变量尚不明确。本研究旨在评估儿童和成人CNS PNET的临床预后及预后因素。回顾性分析了1991年至2011年26例CNS PNET患者(11例儿童和15例成人)的病历。采用Kaplan-Meier法估计无病生存期(DFS)和总生存期(OS),并分析相关预后因素。该队列的5年DFS和OS均为46%。儿童患者的5年DFS为78%;成人患者为22%(P = 0.004)。儿童和成人患者的5年OS分别为67%和33%(P = 0.07)。经包括化疗方案(高剂量vs标准剂量vs非标准剂量)或风险分层(标准风险vs高风险)及年龄的双变量分析,成人疾病复发风险增加的情况依然存在。相对于儿童患者,成人患者OS较差的趋势也依然存在。尽管在考虑年龄影响时结果有所减弱,但干细胞救援的高剂量化疗与OS的统计学显著改善及DFS改善趋势相关。局部复发是成人和儿童治疗失败的主要模式。我们的结果表明,成人CNS PNET患者的预后相对于儿童队列较差。需要进一步研究以改善各年龄段人群CNS PNET的预后。