Department of Biostatistics, Institute of Basic Medical Sciences, University of Oslo, Oslo, Norway.
Stat Med. 2012 Dec 10;31(28):3731-47. doi: 10.1002/sim.5441. Epub 2012 Jun 29.
The Armitage-Doll model with random frailty can fail to describe incidence rates of rare cancers influenced by an accelerated biological mechanism at some, possibly short, period of life. We propose a new model to account for this influence. Osteosarcoma and Ewing sarcoma are primary bone cancers with characteristic age-incidence patterns that peak in adolescence. We analyze Surveillance, Epidemiology and End Result program incidence data for whites younger than 40 years diagnosed during the period 1975-2005, with an Armitage-Doll model with compound Poisson frailty. A new model treating the adolescent growth spurt as the accelerated mechanism affecting cancer development is a significant improvement over that model. We also model the incidence rate conditioning on the event of having developed the cancers before the age of 40 years and compare the results with those predicted by the Armitage-Doll model. Our results support existing evidence of an underlying susceptibility for the two cancers among a very small proportion of the population. In addition, the modeling results suggest that susceptible individuals with a rapid growth spurt acquire the cancers sooner than they otherwise would have if their growth had been slower. The new model is suitable for modeling incidence rates of rare diseases influenced by an accelerated biological mechanism.
Armitage-Doll 带有随机脆弱性的模型可能无法描述受某些可能较短的生命时期加速生物学机制影响的罕见癌症的发病率。我们提出了一个新的模型来解释这种影响。骨肉瘤和尤文肉瘤是原发性骨癌，其特征性的年龄发病率模式在青春期达到高峰。我们分析了 1975 年至 2005 年期间诊断的年龄小于 40 岁的白人患者的监测、流行病学和最终结果计划发病数据，采用带有复合泊松脆弱性的 Armitage-Doll 模型。将青春期生长突增视为影响癌症发展的加速机制的新模型明显优于该模型。我们还对在 40 岁之前发生癌症的事件进行了发病风险建模，并将结果与 Armitage-Doll 模型的预测进行了比较。我们的结果支持在人口中非常小的一部分人群中存在两种癌症潜在易感性的现有证据。此外，建模结果表明，生长突增较快的易感个体比生长较慢的个体更容易患上癌症。该新模型适用于受加速生物学机制影响的罕见疾病发病率的建模。
