Diagnostic test evaluation methodology: A systematic review of methods employed to evaluate diagnostic tests in the absence of gold standard – An update

Abstract

ObjectiveTo systematically review methods developed and employed to evaluate the diagnostic accuracy of medical test when there is a missing or no gold standard.Study design and settingsArticles that proposed or applied any methods to evaluate the diagnostic accuracy of medical test(s) in the absence of gold standard were reviewed. The protocol for this review was registered in PROSPERO (CRD42018089349).ResultsIdentified methods were classified into four main groups: methods employed when there is a missing gold standard; correction methods (which make adjustment for an imperfect reference standard with known diagnostic accuracy measures); methods employed to evaluate a medical test using multiple imperfect reference standards; and other methods, like agreement studies, and a mixed group of alternative study designs. Fifty-one statistical methods were identified from the review that were developed to evaluate medical test(s) when the true disease status of some participants is unverified with the gold standard. Seven correction methods were identified and four methods were identified to evaluate medical test(s) using multiple imperfect reference standards. Flow-diagrams were developed to guide the selection of appropriate methods.ConclusionVarious methods have been proposed to evaluate medical test(s) in the absence of a gold standard for some or all participants in a diagnostic accuracy study. These methods depend on the availability of the gold standard, its’ application to the participants in the study and the availability of alternative reference standard(s). The clinical application of some of these methods, especially methods developed when there is missing gold standard is however limited. This may be due to the complexity of these methods and/or a disconnection between the fields of expertise of those who develop (e.g. mathematicians) and those who employ the methods (e.g. clinical researchers). This review aims to help close this gap with our classification and guidance tools.

The estimation of diagnostic accuracy of tests for COVID-19: A scoping review

“…While many methods have been described, there are only a few well-defined statistical approaches that use a reference standard in lieu of a gold standard, reviewed elsewhere. 60 Latent class analysis is one commonly used approach in situations in which neither the true error rates of the reference standard nor the true prevalence of the disease are known. This approach uses the results of a set of imperfect tests to estimate parameters related to sensitivity, specificity, and prevalence often using maximum likelihood methods.…”

Should RT‐PCR be considered a gold standard in the diagnosis of COVID‐19?

“…For the moment, whenever possible, it is more useful in clinical practice to evaluate tests by several methods because there is no generally accepted reference standard nor is there a gold test for the diagnosis of COVID-19. 14…”

Validity of Medical Record Abstraction and Electronic Health Record–Generated Reports to Assess Performance on Cardiovascular Quality Measures in Primary Care

“…Bayesian latent class models are traditionally used to assess the validity of medical diagnostic tests, and their application to this research topic is extremely novel. 22 , 23 …”