Centre for Childhood Cancer Survivor Studies, Institute of Applied Health Research, University of Birmingham, Birmingham, UK; Section of Cancer Surveillance, International Agency for Research on Cancer, Lyon, France; Cancer and Radiation Team, U1018 INSERM, Villejuif, France; Epidemiology and Biostatistics Section, Gaslini Children Hospital, Genova, Italy; 2nd Department of Pediatrics, Semmelweis University, Budapest, Hungary; Kepler Universitätsklinikum, Linz, Austria; Survivorship Unit, Danish Cancer Society Research Center, Copenhagen, Denmark; Boyne Research Institute, Drogheda, Ireland; Department of Pediatric Oncology, Emma Children's Hospital/Academic Medical Center, Amsterdam, the Netherlands; Department of Pediatrics, Skane University Hospital, Lund University, Lund, Sweden; German Childhood Cancer Registry, Institute of Medical Biostatistics, Epidemiology and Informatics, University Medical Center, Mainz, Germany; Children's Hospital, Landspitali University Hospital, Reykjavik, Iceland; Pediatric Hematology Unit, San Gerardo Hospital, Monza, Italy; Swiss Childhood Cancer Registry, Institute of Social and Preventive Medicine, University of Bern, Bern, Switzerland; Childhood Cancer Registry of Piedmont, Cancer Epidemiology Unit, Department of Medical Sciences, University of Turin and AOU Città della Salute e della Scienza di Torino, Torino, Italy; Norwegian National Advisory Unit on Solid Tumors in Children; Great North Children's Hospital, Newcastle upon Tyne Hospitals NHS Foundation Trust, and Northern Institute of Cancer Research, Newcastle University, Newcastle upon Tyne, UK; Pediatric Oncology Unit, Fondazione IRCCS Istituto Nazionale dei Tumori, Milano, Italy; Norwegian Cancer Registry and Department of Pediatric Medicine, Oslo University Hospital, Oslo, Norway; Faculty of Medicine, Institute of Clinical Medicine, University of Oslo, Norway; Hungarian Childhood Cancer Registry, 2nd Department of Pediatrics, Semmelweis University, Budapest, Hungary; Department of Pediatrics and Adolescent Medicine, Turku University and Turku University Hospital, Turku, Finland; Institute of Oncology, Ljubljana, Slovenia; Department of Pediatrics, University Children's Hospital of Bern, University of Bern, Bern, Switzerland.
J Natl Cancer Inst. 2018 Feb 1;110(2). doi: 10.1093/jnci/djx165.
We investigate the risks of subsequent primary bone cancers after childhood and adolescent cancer in 12 European countries. For the first time, we satisfactorily address the risks beyond 40 years from diagnosis and beyond 40 years of age among all survivors.
This largest-ever assembled cohort comprises 69 460 five-year survivors of cancer diagnosed before age 20 years. Standardized incidence ratios, absolute excess risks, and multivariable-adjusted relative risks and relative excess risks were calculated. All statistical tests were two-sided.
Overall, survivors were 21.65 times (95% confidence interval = 18.97 to 24.60 times) more likely to be diagnosed with a subsequent primary bone cancer than expected from the general population. The greatest excess numbers of bone cancers were observed after retinoblastoma, bone sarcoma, and soft tissue sarcoma. The excess number of bone cancers declined linearly with both years since diagnosis and attained age (all P < .05). Beyond 40 years from diagnosis and age 40 years, there were at most 0.45 excess bone cancers among all survivors per 10 000 person-years at risk; beyond 30 years from diagnosis and age 30 years, there were at most 5.02 excess bone cancers after each of retinoblastoma, bone sarcoma, and soft tissue sarcoma, per 10 000 person-years at risk.
For all survivors combined and the cancer groups with the greatest excess number of bone cancers, the excess numbers observed declined with both age and years from diagnosis. These results provide novel, reliable, and unbiased information about risks and risk factors among long-term survivors of childhood and adolescent cancer.
我们研究了 12 个欧洲国家 69460 名 5 岁以下儿童和青少年癌症幸存者在诊断后发生原发性骨癌的风险。我们首次令人满意地解决了 40 年以上和所有幸存者 40 岁以上的风险。
这是迄今为止最大规模的队列研究,包含 69460 名 5 岁以下癌症幸存者。我们计算了标准化发病比、绝对超额风险、多变量调整后的相对风险和相对超额风险。所有统计检验均为双侧检验。
总的来说,癌症幸存者诊断出原发性骨癌的风险比普通人群高 21.65 倍(95%置信区间为 18.97 至 24.60 倍)。观察到的骨癌超额数量最多的是视网膜母细胞瘤、骨肉瘤和软组织肉瘤。骨癌的超额数量与诊断后年数和达到年龄呈线性下降(均 P <.05)。在诊断后 40 年和 40 岁以后,所有幸存者每 10000 人年风险最多有 0.45 例额外的骨癌;在诊断后 30 年和 30 岁以后,每 10000 人年风险最多有 5.02 例额外的骨癌,包括视网膜母细胞瘤、骨肉瘤和软组织肉瘤。
对于所有幸存者和骨癌超额数量最大的癌症组,观察到的超额数量随年龄和诊断后年数的增加而减少。这些结果为儿童和青少年癌症的长期幸存者提供了关于风险和风险因素的新的、可靠的、无偏倚的信息。