Survival analysis of irish amyotrophic lateral sclerosis patients diagnosed from 1995-2010.
Survival analysis of irish amyotrophic lateral sclerosis patients diagnosed from 1995-2010.
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DOI：
10.1371/journal.pone.0074733
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发表时间：
2013
期刊：
影响因子：
3.7
通讯作者：
Hardiman O
中科院分区：
文献类型：
--
作者：
Rooney J;Byrne S;Heverin M;Corr B;Elamin M;Staines A;Goldacre B;Hardiman O
爱尔兰ALS登记是检查爱尔兰ALS患者生存因素的宝贵资源。考克斯回归已成为生存分析的默认工具，但最近新的灵活的参数生存分析工具，称为罗伊斯顿-帕玛模型已经成为可用。我们采用考克斯比例风险和罗伊斯顿-帕玛灵活的参数模型，以检查影响爱尔兰ALS患者生存的因素。我们进一步研究了时间尺度的选择对考克斯模型和比例风险假设的影响，并扩展了考克斯和Royston-Parmar模型的时变分量。在模型比较中，我们选择了Royston-Parmar比例风险模型作为最佳拟合，该模型不含随时间变化的协变量。使用这个模型，我们证实了已知的生存标志物在ALS中的相关性，包括诊断时的年龄，（风险比（HR）1.34/10年增加; 95% CI 1.26-1.42），诊断延迟（HR 0.96/12周延迟; 95% CI 0.94-0.97），明确ALS（HR 1.47 95% CI 1.17-1.84），延髓发作疾病（HR 1.58 95% CI 1.33-1.87）、利鲁唑使用（HR 0.72 95% CI 0.61-0.85）和ALS诊所就诊（HR 0.74 95% CI 0.64-0.86）。我们的分析探讨了考克斯比例风险和Royston-Parmar灵活的参数方法的优点和缺点。通过包含时变分量，我们能够更深入地了解数据集。不同时间段之间的生存率差异似乎是由于第一个时间段的数据缺失所致。使用年龄作为时间尺度来解释年龄的混杂，解决了违反比例风险假设的问题，但这样做可能会掩盖数据中的缺陷。我们的研究表明，需要测试，并充分探讨，违反考克斯比例风险假设。Royston-Parmar灵活的参数化建模被证明是实现这一目标的有力方法。
The Irish ALS register is a valuable resource for examining survival factors in Irish ALS patients. Cox regression has become the default tool for survival analysis, but recently new classes of flexible parametric survival analysis tools known as Royston-Parmar models have become available. We employed Cox proportional hazards and Royston-Parmar flexible parametric modeling to examine factors affecting survival in Irish ALS patients. We further examined the effect of choice of timescale on Cox models and the proportional hazards assumption, and extended both Cox and Royston-Parmar models with time varying components. On comparison of models we chose a Royston-Parmar proportional hazards model without time varying covariates as the best fit. Using this model we confirmed the association of known survival markers in ALS including age at diagnosis (Hazard Ratio (HR) 1.34 per 10 year increase; 95% CI 1.26–1.42), diagnostic delay (HR 0.96 per 12 weeks delay; 95% CI 0.94–0.97), Definite ALS (HR 1.47 95% CI 1.17–1.84), bulbar onset disease (HR 1.58 95% CI 1.33–1.87), riluzole use (HR 0.72 95% CI 0.61–0.85) and attendance at an ALS clinic (HR 0.74 95% CI 0.64–0.86). Our analysis explored the strengths and weaknesses of Cox proportional hazard and Royston-Parmar flexible parametric methods. By including time varying components we were able to gain deeper understanding of the dataset. Variation in survival between time periods appears to be due to missing data in the first time period. The use of age as timescale to account for confounding by age resolved breaches of the proportional hazards assumption, but in doing so may have obscured deficiencies in the data. Our study demonstrates the need to test for, and fully explore, breaches of the Cox proportional hazards assumption. Royston-Parmar flexible parametric modeling proved a powerful method for achieving this.
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影响因子：
2
作者：
Thiébaut, ACM;Bénichou, J
通讯作者：
Bénichou, J
DOI：
10.3109/17482960802566824
发表时间：
2009-10
期刊：
Amyotrophic lateral sclerosis : official publication of the World Federation of Neurology Research Group on Motor Neuron Diseases
影响因子：
--
作者：
Chiò A;Logroscino G;Hardiman O;Swingler R;Mitchell D;Beghi E;Traynor BG;Eurals Consortium
通讯作者：
Eurals Consortium
影响因子：
2
作者：
Royston, P;Parmar, MKB
通讯作者：
Parmar, MKB
影响因子：
9.9
作者：
Chiò, A;Mora, G;Mutani, R
通讯作者：
Mutani, R
影响因子：
7.2
作者：
Cheung, YB;Gao, F;Khoo, KS
通讯作者：
Khoo, KS