Caroline C.C. Hulsker , Maya Schulpen , Annelies M.C. Mavinkurve-Groothuis , Otto Visser , József Zsiros , Marc H.W. Wijnen , Ronald R. de Krijger , Annette H. Bruggink , Leendert H.J. Looijenga , Henrike E. Karim-Kos , Alida F.W. van der Steeg
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引用次数: 0
Abstract
Background
Population-based studies assessing long-term patterns of incidence and disease characteristics of germ cell tumors (GCTs) in children are scarce. We investigated incidence and survival trends of malignant extracranial GCTs in children using population-based nationwide data from the Netherlands.
Methods
All malignant extracranial GCTs diagnosed in patients aged 0–18 years between 1990 and 2018 were selected from the Netherlands Cancer Registry. Incidence rates were calculated as the average annual number of cases per 1 million person-years. Five-year overall survival (OS) was calculated.
Results
A total of 815 cases were identified. Gonadal GCTs (n=665, testis n=485, ovarian n=180) were more common than extragonadal GCTs (n=149). Stage distribution for testicular and extragonadal GCTs shifted between 1990 and 2004 and 2005–2018 towards more localized disease. The overall incidence remained stable over time, but a significant increase was noted for extragonadal GCTs in the 0–9 years age group. Survival of extragonadal GCTs (5-year OS 84.1%, 95% CI 77.1–89.1), in particular mediastinal GCTs (5-year OS 66.7%, 95% CI 45.7–81.1), was lower than that of gonadal GCTs (5-year OS testis 95.0%, 95% CI 92.7–96.7;ovary 97.8%, 95% CI 94.2–99.2). The 5-year OS of our entire cohort was 93.6% (95% CI 91.7–95.1). Five-year OS significantly increased from 89.5% (95% CI 86.1–92.2) in 1990–2004–97.4% (95% CI 95.3–98.5) in 2005–2018.
Conclusions
Although the incidence of all malignant pediatric extracranial GCTs remained stable during 1990–2018, an increase was observed for extragonadal GCTs in younger children (0–9 years). There was a shift towards more localized disease for testicular and extragonadal GCTs. Five-year OS increased over time exceeding 90% (91.4%, 95% CI 82.7–95.8) in the most recent diagnostic period. Mediastinal GCTs had the lowest OS, supporting the need for future research.
背景评估儿童生殖细胞瘤（GCTs）长期发病模式和疾病特征的人群研究很少。我们利用荷兰全国人口数据调查了儿童恶性颅外GCT的发病率和生存趋势。方法从荷兰癌症登记处选取1990年至2018年间0-18岁患者中确诊的所有恶性颅外GCT。发病率按每100万人年的年平均病例数计算。计算了五年总生存率（OS）。性腺 GCT（665 例，睾丸 485 例，卵巢 180 例）比性腺外 GCT（149 例）更为常见。睾丸和睾丸外 GCT 的分期分布在 1990 年至 2004 年和 2005 年至 2018 年期间向更局部化的疾病转变。随着时间的推移，总体发病率保持稳定，但在0-9岁年龄组中，睾丸外GCT的发病率显著增加。性腺外GCT的生存率（5年OS 84.1%，95% CI 77.1-89.1），尤其是纵隔GCT（5年OS 66.7%，95% CI 45.7-81.1）低于性腺GCT（5年OS睾丸95.0%，95% CI 92.7-96.7；卵巢97.8%，95% CI 94.2-99.2）。整个队列的5年生存率为93.6%（95% CI 91.7-95.1）。5年OS从1990-2004年的89.5%（95% CI 86.1-92.2）明显上升至2005-2018年的97.4%（95% CI 95.3-98.5）。结论虽然1990-2018年期间所有恶性小儿颅外GCT的发病率保持稳定，但在年龄较小的儿童（0-9岁）中观察到颅外GCT的发病率有所上升。睾丸和睾丸外GCT的发病率向更局部化转变。五年生存率随着时间的推移而增加，在最近的诊断期超过了90%（91.4%，95% CI 82.7-95.8）。纵隔GCT的OS最低，这表明未来研究的必要性。
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