Modeling delay to diagnosis for amyotrophic lateral sclerosis: under reporting and incidence estimates.
Modeling delay to diagnosis for amyotrophic lateral sclerosis: under reporting and incidence estimates.
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DOI：
10.1186/1471-2377-12-160
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发表时间：
2012-12-23
期刊：
影响因子：
2.6
通讯作者：
Pierannunzio D
中科院分区：
文献类型：
--
作者：
Rocchetti I;Taruscio D;Pierannunzio D
本文提供了一种策略，以获得一个可靠的估计发病率的肌萎缩侧索硬化症的基础上的数据，从国家罕见疾病登记处（NRRD）。事实上，未观察到的病例可能是由于怀疑患有疾病（发病）和疾病诊断日期之间可能存在“很长一段时间”。研究了可能影响在发作（疑似）时有条件地发生事件（诊断）的概率的潜在因素。由于我们治疗的是罕见病，社会和经济因素的作用并不明显;潜在的和观察到的因素可能会影响诊断的延迟。我们使用半参数估计的基础上的延迟诊断的分布，以考虑潜在的漏报。特别是，我们建议采用霍维茨-汤普森为基础的估计，以纠正发病率的数字，可以从NRRD，意大利2007-2009年期间得出。通过采用所提出的方法获得的发病率估计值约为每10万居民1例，尽管他们让恢复了很大一部分的漏报，他们仍然远离ALS发病率的国际范围1.5和2.5之间。然而，通过仅观察北方意大利，我们可以得出的发病率估计值与国际上已知的发病率估计值一致。这些结果证实，在报告准确性方面存在着很大的差异，并指出了数据收集系统必须改进的地方。特别是，当可靠的个人特征将是可用的，他们可以用来完善拟议的估计。
This paper provides a strategy to obtain a reliable estimate of the incidence rate for Amyotrophic lateral sclerosis based on data from the National Registry of Rare Diseases (NRRD). In fact, unobserved cases may be due to the fact that “a long time” may intercour between the suspect of having the disease (onset) and the date the disease is diagnosed. Potential factors that may influence the probability of experiencing the event (diagnosis) conditionally on the onset (suspected) are investigated. Since we are treating rare diseases, the role of social and economic factors is not that obvious; latent as well as observed factors may influence the delay to diagnosis. We use a semiparametric estimator based on the distribution of delay to diagnosis to account for potential underreporting. In particular, we propose to adopt an Horvitz-Thompson based estimator to correct the incidence figure that can be derived for the period 2007-2009 from the NRRD, Italy. The incidence estimates obtained by adopting the proposed approach are about 1 case per 100000 inhabitants and despite they let recovering a good part of underreporting, they are still far from ALS incidence international ranges between 1.5 and 2.5. However, by looking only at northern Italy, the incidence estimates we can derive are coherent with those known internationally. These results confirm the existence of substantial differences in reporting accuracy, and point out where the system of data collection must be improved. In particular, when reliable individual characteristics will be available, they could be employed to refine the proposed estimator.
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DOI：
10.1136/jnnp.2009.183525
发表时间：
2010-04
期刊：
Journal of neurology, neurosurgery, and psychiatry
影响因子：
--
作者：
Logroscino G;Traynor BJ;Hardiman O;Chiò A;Mitchell D;Swingler RJ;Millul A;Benn E;Beghi E;EURALS
通讯作者：
EURALS
影响因子：
1.9
作者：
Kraemer, Markus;Buerger, Melanie;Berlit, Peter
通讯作者：
Berlit, Peter
影响因子：
2.9
作者：
KALBFLEISCH, JD;LAWLESS, JF
通讯作者：
LAWLESS, JF
影响因子：
11
作者：
Logroscino, G;Beghi, E;Serlenga, L
通讯作者：
Serlenga, L
影响因子：
2
作者：
Cui, JS;Yip, PSF;Chau, PH
通讯作者：
Chau, PH