Winther J F, Boice J D, Frederiksen K, Bautz A, Mulvihill J J, Stovall M, Olsen J H
Institute of Cancer Epidemiology, Danish Cancer Society, DK-2100 Copenhagen, Denmark.
Clin Genet. 2009 Jan;75(1):50-6. doi: 10.1111/j.1399-0004.2008.01109.x. Epub 2008 Nov 17.
Offspring of childhood cancer survivors may be at risk of genetic disease due to the mutagenic cancer treatments received by their parents. Congenital malformations were evaluated in a population-based cohort study of 1715 offspring of 3963 childhood cancer survivors and 6009 offspring of 5657 survivors' siblings. The Danish Central Population Register, Cancer Registry and Hospital Register were used to identify study subjects and congenital malformations. Gonadal and uterine radiation doses were characterized based on standard radiation-treatment regimens. The prevalence of congenital malformations at birth in offspring of survivors (44 cases, 2.6%) was slightly higher but not statistically different from that of offspring of siblings (140 cases, 2.3%) [prevalence proportion ratio (PPR), 1.1; 95% confidence interval, 0.8-1.5] or of the general population (observed-to-expected ratio, 1.2; 0.9-1.6). Including malformations diagnosed later in life did not change the ratios appreciably. The risk for malformations was slightly higher in the offspring of irradiated parents than in that of non-irradiated parents (PPR 1.2 vs 1.0) but was unrelated to gonadal dose. This study provides evidence that cancer therapy of children does not increase the risk for malformations in their offspring. Continued monitoring of genetic risks among their offspring, however, is warranted.
儿童癌症幸存者的后代可能因父母接受的致突变性癌症治疗而面临患遗传疾病的风险。在一项基于人群的队列研究中,对3963名儿童癌症幸存者的1715名后代以及5657名幸存者兄弟姐妹的6009名后代的先天性畸形进行了评估。丹麦中央人口登记处、癌症登记处和医院登记处被用于识别研究对象和先天性畸形。根据标准放疗方案对性腺和子宫的辐射剂量进行了特征描述。幸存者后代出生时先天性畸形的患病率(44例,2.6%)略高于兄弟姐妹后代(140例,2.3%),但在统计学上无差异[患病率比例比(PPR)为1.1;95%置信区间为0.8 - 1.5],也与一般人群无异(观察到的与预期的比例为1.2;0.9 - 1.6)。纳入后期诊断出的畸形并未明显改变这些比例。受辐射父母的后代出现畸形的风险略高于未受辐射父母的后代(PPR为1.2对1.0),但与性腺剂量无关。这项研究提供了证据,表明儿童癌症治疗不会增加其后代出现畸形的风险。然而,仍有必要继续监测其后代的遗传风险。