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Prevalence estimation subject to misclassification: the mis-substitution bias and some remedies.

ABSTRACT: We consider the problem of estimating the prevalence of a disease under a group testing framework. Because assays are usually imperfect, misclassification of disease status is a major challenge in prevalence estimation. To account for possible misclassification, it is usually assumed that the sensitivity and specificity of the assay are known and independent of the group size. This assumption is often questionable, and substitution of incorrect values of an assay's sensitivity and specificity can result in a large bias in the prevalence estimate, which we refer to as the mis-substitution bias. In this article, we propose simple designs and methods for prevalence estimation that do not require known values of assay sensitivity and specificity. If a gold standard test is available, it can be applied to a validation subsample to yield information on the imperfect assay's sensitivity and specificity. When a gold standard is unavailable, it is possible to estimate assay sensitivity and specificity, either as unknown constants or as specified functions of the group size, from group testing data with varying group size. We develop methods for estimating parameters and for finding or approximating optimal designs, and perform extensive simulation experiments to evaluate and compare the different designs. An example concerning human immunodeficiency virus infection is used to illustrate the validation subsample design.

SUBMITTER: Zhang Z

PROVIDER: S-EPMC4184986 | biostudies-literature | 2014 Nov

REPOSITORIES: biostudies-literature

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Prevalence estimation subject to misclassification: the mis-substitution bias and some remedies.

Zhang Zhiwei Z Liu Chunling C Kim Sungduk S Liu Aiyi A

Statistics in medicine 20140718 25

We consider the problem of estimating the prevalence of a disease under a group testing framework. Because assays are usually imperfect, misclassification of disease status is a major challenge in prevalence estimation. To account for possible misclassification, it is usually assumed that the sensitivity and specificity of the assay are known and independent of the group size. This assumption is often questionable, and substitution of incorrect values of an assay's sensitivity and specificity ca ...[more]

PMID: 25043925

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Project description:BACKGROUND:Regional prevalence estimation requires epidemiologic data with substantial local detail. National health surveys may lack in sufficient local observations due to limited resources. Therefore, corresponding prevalence estimates may not capture regional morbidity patterns with the necessary accuracy. Health insurance records represent alternative data sources for this purpose. Fund-specific member populations have more local observations than surveys, which benefits regional prevalence estimation. However, due to national insurance market regulations, insurance membership can be informative for morbidity. Regional fund-specific prevalence proportions are selective in the sense that the morbidity structure of a fund's member population cannot be extrapolated to the national population. This implies a selection bias that marks a major obstacle for statistical inference. We provide a methodology to adjust fund-specific selectivity and perform regional prevalence estimation from health insurance records. The methodology is applied to estimate regional cohort-referenced diabetes mellitus type 2 prevalence in Germany. METHODS:Records of the German Public Health Insurance Company from 2014 and Diagnosis-Related Group Statistics data are combined within a benchmarked multi-level model. The fund-specific selectivity is adjusted in a two-step procedure. Firstly, the conditional expectation of the insurance company's regional prevalence given related inpatient diagnosis frequencies of its members is quantified. Secondly, the regional prevalence is estimated by extrapolating the conditional expectation using corresponding inpatient diagnosis frequencies of the Diagnosis-Related Group Statistics as benchmarks. Model assumptions are validated via Monte Carlo simulation. Variable selection is performed via multivariate methods. The optimal model fit is determined by analysis of variance. 95% confidence intervals for the estimates are constructed via semiparametric bootstrapping. RESULTS:The national diabetes mellitus type 2 prevalence is estimated at 8.70% with a 95% confidence interval of [8.48%, 9.35%]. This indicates an adjustment of the original fund-specific prevalence from -?32.79 to -?25.93%. The estimated disease distribution shows significant morbidity differences between regions, especially between eastern and western Germany. However, the cohort-referenced estimates suggest that these differences can be partially explained by regional demography. CONCLUSIONS:The proposed methodology allows regional prevalence estimation in remarkable detail despite fund-specific selectivity. This enhances and encourages the use of health insurance records for future epidemiologic studies.

Dataset Information

Prevalence estimation subject to misclassification: the mis-substitution bias and some remedies.

Publications

Prevalence estimation subject to misclassification: the mis-substitution bias and some remedies.

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