Systematic reviews aim to summarize all the available evidence relevant to a particular research question. If appropriate, the data from identified studies are quantitatively combined in a meta-analysis. Often only few studies regarding a particular research question exist. In these settings the estimation of the between-study heterogeneity is challenging. Furthermore, the assessment of publication bias is difficult as standard methods such as visual inspection or formal hypothesis tests in funnel plots do not provide adequate guidance. Previously, Henmi and Copas (Statistics in Medicine 2010, 29: 2969--2983) proposed a confidence interval for the overall effect in random-effects meta-analysis that is robust to publication bias to some extent. As is evident from their simulations, the confidence intervals have improved coverage compared with standard methods. To our knowledge, the properties of their method has never been assessed for meta-analyses including fewer than five studies. In this manuscript, we propose a variation of the method by Henmi and Copas employing an improved estimator of the between-study heterogeneity, in particular when dealing with few studies only. In a simulation study, the proposed method is compared to several competitors. Overall, we found that our method outperforms the others in terms of coverage probabilities. In particular, an improvement compared with the proposal by Henmi and Copas is demonstrated. The work is motivated and illustrated by a systematic review and meta-analysis in paediatric immunosuppression following liver transplantations.
The purpose of the present paper is to assess the efficacy of confidence intervals for Rosenthal's fail-safe number. Although Rosenthal's estimator is highly used by researchers, its statistical properties are largely unexplored. First of all, we developed statistical theory which allowed us to produce confidence intervals for Rosenthal's fail-safe number.This was produced by discerning whether the number of studies analysed in a meta-analysis is fixed or random. Each case produces different variance estimators. For a given number of studies and a given distribution, we provided five variance …
We propose simple formulas of confidence intervals for the Wald statistic, likelihood ratio statistic, and score statistic for a network meta-analysis. In addition, we consider resolutions for concerns that network meta-analyses with a small number of studies cannot hold a nominal confidence level. For a bias adjustment in analyses with a small number of studies, a Bartlett-type adjustment is a well-known method. Many Bartlett-type adjustment-type methods are based on maximum likelihood estimators. However, the network meta-analysis often uses the restricted maximum likelihood estimators that …
Meta-analyses of correlation coefficients are an important technique to integrate results from many cross-sectional and longitudinal research designs. Uncertainty in pooled estimates is typically assessed with the help of confidence intervals, which can double as hypothesis tests for two-sided hypotheses about the underlying correlation. A standard approach to construct confidence intervals for the main effect is the Hedges-Olkin-Vevea Fisher-z (HOVz) approach, which is based on the Fisher-z transformation. Results from previous studies (Field, 2005; Hafdahl and Williams, 2009), however, indic…
Frequentist prediction intervals for random-effects meta-analysis via confidence-distribution propagation
The Institute of Statistical Mathematics（日本统计科学研究所）；The Graduate University for Advanced Studies（高级研究院）；Institute of Medical Biometry and Statistics, Medical Faculty and Medical Center – University of Freiburg（弗赖堡大学医学院医学中心医学生物统计学与统计研究所）
Publication bias is a major concern in conducting systematic reviews and meta-analyses. Various sensitivity analysis or bias-correction methods have been developed based on selection models and they have some advantages over the widely used bias-correction method of the trim-and-fill method. However, likelihood methods based on selection models may have difficulty in obtaining precise estimates and reasonable confidence intervals or require a complicated sensitivity analysis process. In this paper, we develop a simple publication bias adjustment method utilizing information on conducted but st…
Meta-analysis is an important statistical technique for synthesizing the results of multiple studies regarding the same or closely related research question. So-called meta-regression extends meta-analysis models by accounting for studylevel covariates. Mixed-effects meta-regression models provide a powerful tool for evidence synthesis, by appropriately accounting for betweem-study heterogeneity. In fact, modelling the study effect in terms of random effects and moderators not only allows to examine the impact of the moderators, but often leads to more accurate estimates of the involved parame…
For a number of researchers a number of publications for each author is simulated using the zeta distribution and then for each publication a number of citations per publication simulated. Bootstrap confidence intervals indicate that the difference between the average of ratios and the ratio of averages are not significant, and there are no significant differences in the distributions in realistic problems when using the two-sample Kolmogorov-Smirnov test to compare distributions. It was found that the log-logistic distribution which is a general form for the ratio of two correlated Pareto ran…
The area under the curve (AUC) of summary receiver operating characteristic (SROC) curve is a primary statistical outcome for meta-analysis of diagnostic test accuracy studies (DTA). However, its confidence interval has not been reported in most of DTA meta-analyses, because no certain methods and statistical packages have been provided. In this article, we provide a bootstrap algorithm for computing the confidence interval of the AUC. Also, using the bootstrap framework, we can conduct a bootstrap test for assessing significance of the difference of AUCs for multiple diagnostic tests. In addi…
Multivariate meta-analysis is gaining prominence in evidence synthesis research because it enables simultaneous synthesis of multiple correlated outcome data, and random-effects models have generally been used for addressing between-studies heterogeneities. However, coverage probabilities of confidence regions or intervals for standard inference methods for random-effects models (e.g., restricted maximum likelihood estimation) cannot retain their nominal confidence levels in general, especially when the number of synthesized studies is small because their validities depend on large sample appr…
In meta-analyses, publication bias is a well-known, important and challenging issue because the validity of the results from a meta-analysis is threatened if the sample of studies retrieved for review is biased. One popular method to deal with publication bias is the Copas selection model, which provides a flexible sensitivity analysis for correcting the estimates with considerable insight into the data suppression mechanism. However, rigorous testing procedures under the Copas selection model to detect bias are lacking. To fill this gap, we develop a score-based test for detecting publication…
Global sensitivity analysis is often impracticable for complex and time demanding numerical models, as it requires a large number of runs. The reduced-basis approach provides a way to replace the original model by a much faster to run code. In this paper, we are interested in the information loss induced by the approximation on the estimation of sensitivity indices. We present a method to provide a robust error assessment, hence enabling significant time savings without sacrifice on precision and rigourousness. We illustrate our method with an experiment where computation time is divided by a …
Publication bias occurs when the publication of research results depends not only on the quality of the research but also on its nature and direction. The consequence is that published studies may not be truly representative of all valid studies undertaken, and this bias may threaten the validity of systematic reviews and meta-analyses - on which evidence-based medicine increasingly relies. Multivariate meta-analysis has recently received increasing attention for its ability reducing potential bias and improving statistical efficiency by borrowing information across outcomes. However, detectin…
Prospective registration of study protocols in clinical trial registries is a useful way to minimize the risk of publication bias in meta-analysis, and several clinical trial registries are available nowadays. However, they are mainly used as a tool for searching studies and information submitted to the registries has not been utilized as efficiently as it could. In addressing publication bias in meta-analyses, sensitivity analysis with the Copas selection model is a more objective alternative to widely-used graphical methods such as the funnel-plot and the trim-and-fill method. Despite its ab…
The present study investigates the performance of several statistical tests to detect publication bias in diagnostic meta-analysis by means of simulation. While bivariate models should be used to pool data from primary studies in diagnostic meta-analysis, univariate measures of diagnostic accuracy are preferable for the purpose of detecting publication bias. In contrast to earlier research, which focused solely on the diagnostic odds ratio or its logarithm ($\lnω$), the tests are combined with four different univariate measures of diagnostic accuracy. For each combination of test and univariat…
The validity of conclusions from meta-analysis is potentially threatened by publication bias. Most existing procedures for correcting publication bias assume normality of the study-specific effects that account for between-study heterogeneity. However, this assumption may not be valid, and the performance of these bias correction procedures can be highly sensitive to departures from normality. Further, there exist few measures to quantify the magnitude of publication bias based on selection models. In this paper, we address both of these issues. First, we explore the use of heavy-tailed distri…
In an interesting recent work, Kuzborskij and Szepesvári derived a confidence bound for functions of independent random variables, which is based on an inequality that relates concentration to squared perturbations of the chosen function. Kuzborskij and Szepesvári also established the PAC-Bayes-ification of their confidence bound. Two important aspects of their work are that the random variables could be of unbounded range, and not necessarily of an identical distribution. The purpose of this note is to advertise/discuss these interesting results, with streamlined proofs. This expository note …
In meta-analysis of diagnostic test accuracy, summary receiver operating characteristic (SROC) is a recommended method to summarize the discriminant capacity of a diagnostic test in the presence of study-specific cutoff values and the area under the SROC (SAUC) gives the aggregate measure of test accuracy. SROC or SAUC can be estimated by bivariate modelling of pairs of sensitivity and specificity over the primary diagnostic studies. However, publication bias is a major threat to the validity of estimates in meta-analysis. To address this issue, we propose to adopt sensitivity analysis to make…
Publication bias and p-hacking are two well-known phenomena that strongly affect the scientific literature and cause severe problems in meta-analyses. Due to these phenomena, the assumptions of meta-analyses are seriously violated and the results of the studies cannot be trusted. While publication bias is almost perfectly captured by the weighting function selection model, p-hacking is much harder to model and no definitive solution has been found yet. In this paper we propose to model both publication bias and p-hacking with selection models. We derive some properties for these models, and we…
Simulation studies are commonly used to evaluate the performance of newly developed meta-analysis methods. For methodology that is developed for an aggregated data meta-analysis, researchers often resort to simulation of the aggregated data directly, instead of simulating individual participant data from which the aggregated data would be calculated in reality. Clearly, distributional characteristics of the aggregated data statistics may be derived from distributional assumptions of the underlying individual data, but they are often not made explicit in publications. This paper provides the di…
Meta-analyses of diagnostic test accuracy (DTA) studies have been gathering attention in research in clinical epidemiology and health technology development, and bivariate random-effects model is becoming a standard tool. However, standard inference methods usually underestimate statistical errors and possibly provide highly overconfident results under realistic situations since they ignore the variability in the estimation of variance parameters. To overcome the difficulty, a new improved inference method, namely, an accurate confidence region for the meta-analysis of DTA, by asymptotically e…
Copas' method corrects a pooled estimate from an aggregated data meta-analysis for publication bias. Its performance has been studied for one particular mechanism of publication bias. We show through simulations that Copas' method is not robust against other realistic mechanisms. This questions the usefulness of Copas' method, since publication bias mechanisms are typically unknown in practice.
Scargle (2000) has discussed Rosenthal and Rubin's (1978) "fail-safe number" (FSN) method for estimating the number of unpublished studies in meta-analysis. He concluded that this FSN cannot possibly be correct because a central assumption the authors used conflicts with the very definition of publication bias. While this point has been made by others before (Elsahoff, 1978; Darlington, 1980; Thomas, 1985, Iyengar & Greenhouse, 1988), Scargle showed, by way of a simple 2-parameter model, how far off Rosenthal & Rubin' s estimate can be in practice. However, his results relied on the assumption…
Standard random-effects meta-analysis methods perform poorly when applied to few studies only. Such settings however are commonly encountered in practice. It is unclear, whether or to what extent small-sample-size behaviour can be improved by more sophisticated modeling. We consider several likelihood-based inference methods. Confidence intervals are based on normal or Student-t approximations. We extract an empirical data set of 40 meta-analyses from recent reviews published by the German Institute for Quality and Efficiency in Health Care (IQWiG). Methods are then compared empirically as wel…
