目的 分析复发性颅外恶性生殖细胞肿瘤(MGCTs)患儿的生存率及预后因素，探索复发性颅外MGCTs患儿可行的挽救性治疗手段。 方法 回顾性研究。研究对象为2010年1月至2020年1月上海儿童医学中心收治的复发性颅外MGCTs患儿，治疗方案包括手术、化疗和放疗。采用Kaplan－Meier生存分析法及Cox风险回归对患儿进行生存率及预后因素分析。 结果 共收治172例颅外MGCTs患儿，其中21例复发，复发率为12。2%，中位复发时间为治疗结束后11个月，2例复发患儿因不符合入组标准被剔除，最终19例进入统计。19例患儿中男10例，女9例；复发年龄26个月(8～170个月)；随访时间57个月(13～122个月)。复发患儿中16例接受挽救性化疗，14例接受手术完整切除，4例患儿接受放疗。复发患儿4年总体生存(4yr-OS)率为(82。5±9。2)%(19例)，其中初诊时仅手术未行辅助化疗的患儿，复发后再治疗的4yr-OS率为(92。3±7。4)% (13例)；初诊时接受化疗的患儿，复发再治疗后4yr-OS率为(60。0±21。9)% (6例)，二者比较差异有统计学意义(P＝0。002)。单因素及Cox多因素回归分析均提示，化疗3个疗程后甲胎蛋白水平未恢复正常者明显影响最终生存率。 结论 颅外MGCTs患儿复发后通过完整手术切除、化疗、放疗等综合治疗后仍有较高的生存率，初治时仅手术未行辅助化疗者，复发再治疗后的生存率明显优于初治时已接受化疗的患儿。 Objective To study the survival and prognostic factors for the recurrent extracranial malignant germ cell tumors (MGCTs) in children， and to explore feasible salvage treatment。 Methods A retrospective study。Pediatric patients with recurrent extracranial MGCTs diagnosed in Shanghai Children′s Medical Center between January 2010 and January 2020 were retrospectively recruited。Comprehensive treatment regimens included surgery， chemotherapy and radiation。Kaplan-Meier survival analysis and Cox regression model were employed to analyze the survival and prognostic factors for children with recurrent extracranial MGCTs。 Results A total of 172 children with extracranial MGCTs were treated， including 21 (12。2%) recurrent cases。The median time of MGCT recurrence after the end of the first treatment was 11 months。Finally， 19 patients were recruited after excluding 2 non-eligible cases， including 10 boys and 9 girls with the age at recurrence of 26 (8-170) months。The follow-up time was 57 (13-122) months。Salvage chemotherapy， complete resection and radiotherapy were performed in 16， 14 and 4 patients， respectively。The 4-year overall survival (4yr-OS) rate was (82。5±9。2)%(19 cases)。 The 4yr-OS rate was significantly higher in patients managed with surgery but without adjuvant chemotherapy at the initial treatment (13 cases) than those managed with chemotherapy at the initial treatment (6 cases)[(92。3±7。4)% vs。(60。0%±21。9)%， P=0。002]。 Univariant and Cox multivariant regression analyses showed that failure to achieve the normal range of alpha fetoprotein after 3 cycles of chemotherapy significantly influenced the survival of recurrent extracranial MGCTs。 Conclusions For patients with recurrent extracranial MGCTs， comprehensive treatment approaches like complete surgical resection， chemotherapy， and radiotherapy offer a favorable survival rate。Specifically， recurrent and re-treated patients who initially received surgery alone without adjuvant chemotherapy have a higher survival rate compared to those who received chemotherapy during the initial treatment。
A retrospective single-center study of treatment experience of recurrent extracranial malignant germ cell tumor in 19 children
Objective To study the survival and prognostic factors for the recurrent extracranial malignant germ cell tumors (MGCTs) in children, and to explore feasible salvage treatment. Methods A retrospective study.Pediatric patients with recurrent extracranial MGCTs diagnosed in Shanghai Children′s Medical Center between January 2010 and January 2020 were retrospectively recruited.Comprehensive treatment regimens included surgery, chemotherapy and radiation.Kaplan-Meier survival analysis and Cox regression model were employed to analyze the survival and prognostic factors for children with recurrent extracranial MGCTs. Results A total of 172 children with extracranial MGCTs were treated, including 21 (12.2%) recurrent cases.The median time of MGCT recurrence after the end of the first treatment was 11 months.Finally, 19 patients were recruited after excluding 2 non-eligible cases, including 10 boys and 9 girls with the age at recurrence of 26 (8-170) months.The follow-up time was 57 (13-122) months.Salvage chemotherapy, complete resection and radiotherapy were performed in 16, 14 and 4 patients, respectively.The 4-year overall survival (4yr-OS) rate was (82.5±9.2)%(19 cases). The 4yr-OS rate was significantly higher in patients managed with surgery but without adjuvant chemotherapy at the initial treatment (13 cases) than those managed with chemotherapy at the initial treatment (6 cases)[(92.3±7.4)% vs.(60.0%±21.9)%, P=0.002]. Univariant and Cox multivariant regression analyses showed that failure to achieve the normal range of alpha fetoprotein after 3 cycles of chemotherapy significantly influenced the survival of recurrent extracranial MGCTs. Conclusions For patients with recurrent extracranial MGCTs, comprehensive treatment approaches like complete surgical resection, chemotherapy, and radiotherapy offer a favorable survival rate.Specifically, recurrent and re-treated patients who initially received surgery alone without adjuvant chemotherapy have a higher survival rate compared to those who received chemotherapy during the initial treatment.
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