Liu H C, Liang D C, Chen S H, Liu F L, Chang P Y, Sheu J C, Wang N L
Department of Pediatrics, Mackay Memorial Hospital, Taipei, Taiwan.
Pediatr Hematol Oncol. 1998 May-Jun;15(3):223-8. doi: 10.3109/08880019809028788.
Yolk sac tumor is the most frequent germ cell tumor of testis in children. For stage I yolk sac tumor of testis in children younger than 2 years, high inguinal orchiectomy alone has been the standard treatment, with a cure rate of at least 75%. Here, we compare the treatment results of receiving chemotherapy or no chemotherapy after orchiectomy, to analyze the role of chemotherapy. From February 1987 to January 1997, 22 children younger than 2 years, with stage I yolk sac tumor of testis, were included in the study. All patients had high inguinal orchiectomy without retroperitoneal lymphadenectomy. Initial diagnostic imaging studies included computed tomographic scan of abdomen, chest radiography, and long bone survey. Clinical stage I was defined as a tumor completely resected with no evidence of local regional lymph node involvement or distant metastases. Serum alpha-fetoprotein (AFP) was assessed at diagnosis. After orchiectomy, diagnosis, and staging, patients were stratified into two treatment groups, with or without chemotherapy, according to the decision of the parents. Ten children received chemotherapy consisting of cisplatin, vinblastine, and bleomycin (PVB, modified "Einhorn regimen") for 12 weeks. The remaining 12 patients were followed up according to a "wait and see" policy. Determination of AFP was performed monthly during the first postoperative year, every other month during the second year, every 3 months during the third year, every 6 months during the fourth year, and yearly until the fifth postoperative year at least. The duration of follow-up ranged from 3 months to 119 months (median, 53 months). The Kaplan-Meier plot estimated an overall survival rate of 91.6% at 7 years after diagnosis. Among the 12 patients without chemotherapy, 2 children had relapses at 4 and 6 months after diagnosis, respectively. One was cured with PVB chemotherapy. The other patient died with refractory lung metastasis, in spite of intensive multimodality salvage therapy. The Kaplan-Meier plot showed a survival rate of 80% at 7 years and a relapse-free survival rate of 81.8% at 5 years after diagnosis. All children receiving chemotherapy were alive and free from relapse. There was no significant treatment-related toxicity. Our results may suggest that PVB chemotherapy after orchiectomy is an affective and safe regimen for stage I yolk sac tumor of testis in children younger than 2 years. Instead of four courses of PVB as used here, two or three courses could be enough. To elucidate the necessity for chemotherapy and to determine the number of courses of PVB needed (if chemotherapy is given), a randomized study of more cases is warranted.
卵黄囊瘤是儿童睾丸最常见的生殖细胞肿瘤。对于2岁以下儿童的I期睾丸卵黄囊瘤，单纯高位腹股沟睾丸切除术一直是标准治疗方法，治愈率至少为75%。在此，我们比较睾丸切除术后接受化疗或不接受化疗的治疗结果，以分析化疗的作用。从1987年2月至1997年1月，22例2岁以下患有I期睾丸卵黄囊瘤的儿童被纳入研究。所有患者均接受了高位腹股沟睾丸切除术，未进行腹膜后淋巴结清扫术。初始诊断性影像学检查包括腹部计算机断层扫描、胸部X线摄影和长骨检查。临床I期定义为肿瘤完全切除，无局部区域淋巴结受累或远处转移的证据。诊断时评估血清甲胎蛋白（AFP）。睾丸切除术后、诊断和分期后，根据家长的决定将患者分为两个治疗组，即接受化疗组和不接受化疗组。10名儿童接受了由顺铂、长春碱和博来霉素组成的化疗（PVB，改良的“艾因霍恩方案”），为期12周。其余12名患者根据“观察等待”策略进行随访。术后第一年每月测定AFP，第二年每两个月测定一次，第三年每三个月测定一次，第四年每六个月测定一次，至少直到术后第五年每年测定一次。随访时间为3个月至119个月（中位数为53个月）。Kaplan-Meier曲线估计诊断后7年的总生存率为91.6%。在12名未接受化疗的患者中，分别有2名儿童在诊断后4个月和6个月复发。其中一名经PVB化疗治愈。另一名患者尽管接受了强化多模式挽救治疗，但仍死于难治性肺转移。Kaplan-Meier曲线显示诊断后7年生存率为80%，5年无复发生存率为81.8%。所有接受化疗的儿童均存活且无复发。没有明显的与治疗相关的毒性。我们的结果可能表明，睾丸切除术后的PVB化疗对于小于2岁儿童的I期睾丸卵黄囊瘤是一种有效且安全的方案。此处使用的不是四个疗程的PVB，两个或三个疗程可能就足够了。为了阐明化疗的必要性并确定所需的PVB疗程数（如果给予化疗），有必要进行更多病例的随机研究。
