A hybrid Bayesian hierarchical model combining cohort and case-control studies for meta-analysis of diagnostic tests: Accounting for partial verification bias

Stat Methods Med Res. 2016 Dec;25(6):3015-3037. doi: 10.1177/0962280214536703. Epub 2014 May 26.


To account for between-study heterogeneity in meta-analysis of diagnostic accuracy studies, bivariate random effects models have been recommended to jointly model the sensitivities and specificities. As study design and population vary, the definition of disease status or severity could differ across studies. Consequently, sensitivity and specificity may be correlated with disease prevalence. To account for this dependence, a trivariate random effects model had been proposed. However, the proposed approach can only include cohort studies with information estimating study-specific disease prevalence. In addition, some diagnostic accuracy studies only select a subset of samples to be verified by the reference test. It is known that ignoring unverified subjects may lead to partial verification bias in the estimation of prevalence, sensitivities, and specificities in a single study. However, the impact of this bias on a meta-analysis has not been investigated. In this paper, we propose a novel hybrid Bayesian hierarchical model combining cohort and case-control studies and correcting partial verification bias at the same time. We investigate the performance of the proposed methods through a set of simulation studies. Two case studies on assessing the diagnostic accuracy of gadolinium-enhanced magnetic resonance imaging in detecting lymph node metastases and of adrenal fluorine-18 fluorodeoxyglucose positron emission tomography in characterizing adrenal masses are presented.

Keywords: Bayesian method; cohort and case–control studies; diagnostic test; meta-analysis; partial verification bias.

MeSH terms

  • Bayes Theorem*
  • Bias
  • Case-Control Studies
  • Cohort Studies
  • Diagnostic Tests, Routine*
  • Humans
  • Lymphatic Metastasis / diagnostic imaging
  • Magnetic Resonance Imaging
  • Meta-Analysis as Topic*
  • Sensitivity and Specificity