Bayesian Analysis of Diagnostic Test Accuracy When Disease State is Unverified for Some Subjects

Studies of the accuracy of medical tests to diagnose the presence or absence of disease can suffer from an inability to verify the true disease state in everyone. When verification is missing at random (MAR), the missing data mechanism can be ignored in likelihood-based inference. However, this assumption may not hold even approximately. When verification is nonignorably missing, the most general model of the distribution of disease state, test result, and verification indicator is overparameterized. Parameters are only partially identified, creating regions of ignorance for maximum likelihood estimators. For studies of a single test, we use Bayesian analysis to implement the most general nonignorable model, a reduced nonignorable model with identifiable parameters, and the MAR model. Simple Gibbs sampling algorithms are derived that enable computation of the posterior distribution of test accuracy parameters. In particular, the posterior distribution is easily obtained for the most general nonignorable model, which makes relatively weak assumptions about the missing data mechanism. For this model, the posterior distribution combines two sources of uncertainty: ignorance in the estimation of partially identified parameters, and imprecision due to finite sampling variability. We compare the three models on data from a study of the accuracy of scintigraphy to diagnose liver disease.

[1]  R A Greenes,et al.  Assessment of diagnostic tests when disease verification is subject to selection bias. , 1983, Biometrics.

[2]  L. Joseph,et al.  Bayesian Approaches to Modeling the Conditional Dependence Between Multiple Diagnostic Tests , 2001, Biometrics.

[3]  S G Baker,et al.  Evaluating multiple diagnostic tests with partial verification. , 1995, Biometrics.

[4]  W. Wong,et al.  The calculation of posterior distributions by data augmentation , 1987 .

[5]  Huiman X Barnhart,et al.  A global sensitivity analysis of performance of a medical diagnostic test when verification bias is present , 2003, Statistics in medicine.

[6]  D. Rubin INFERENCE AND MISSING DATA , 1975 .

[7]  L. Joseph,et al.  Bayesian estimation of disease prevalence and the parameters of diagnostic tests in the absence of a gold standard. , 1995, American journal of epidemiology.

[8]  Geert Molenberghs,et al.  Sensitivity analysis for incomplete contingency tables: the Slovenian plebiscite case , 2001 .

[9]  Nicole A. Lazar,et al.  Statistical Analysis With Missing Data , 2003, Technometrics.

[10]  Bradley P. Carlin,et al.  Bayesian measures of model complexity and fit , 2002 .

[11]  R. Little,et al.  The prevention and treatment of missing data in clinical trials. , 2012, The New England journal of medicine.

[12]  P. Albert,et al.  A Cautionary Note on the Robustness of Latent Class Models for Estimating Diagnostic Error without a Gold Standard , 2004, Biometrics.

[13]  M. Tan,et al.  Random effects models in latent class analysis for evaluating accuracy of diagnostic tests. , 1996, Biometrics.

[14]  David J. Spiegelhalter,et al.  Introducing Markov chain Monte Carlo , 1995 .

[15]  Roderick J. A. Little,et al.  Statistical Analysis with Missing Data: Little/Statistical Analysis with Missing Data , 2002 .

[16]  B. Craig,et al.  Estimating disease prevalence in the absence of a gold standard , 2002, Statistics in medicine.

[17]  D. Drum,et al.  Hepatic scintigraphy in clinical decision making. , 1972, Journal of nuclear medicine : official publication, Society of Nuclear Medicine.

[18]  Xiao-Hua Zhou,et al.  Maximum likelihood estimators of sensitivity and specificity corrected for verification bias , 1993 .

[19]  Joseph L. Gastwirth,et al.  Bayesian Inference for Medical Screening Tests: Approximations Useful for the Analysis of Acquired Immune Deficiency Syndrome , 1991 .