著者
Hisashi Noma Munechika Misumi Shiro Tanaka
出版者
Japan Epidemiological Association
雑誌
Journal of Epidemiology (ISSN:09175040)
巻号頁・発行日
pp.JE20210509, (Released:2022-06-25)
参考文献数
40
被引用文献数
1 3

Background: In case-cohort studies with binary outcomes, ordinary logistic regression analyses have been widely used because of their computational simplicity. However, the resultant odds ratio estimates cannot be interpreted as relative risk measures unless the event rate is low. The risk ratio and risk difference are more favorable outcome measures that are directly interpreted as effect measures without the rare disease assumption.Methods: We provide pseudo-Poisson and pseudo-normal linear regression methods for estimating risk ratios and risk differences in analyses of case-cohort studies. These multivariate regression models are fitted by weighting the inverses of sampling probabilities. Also, the precisions of the risk ratio and risk difference estimators can be improved using auxiliary variable information, specifically by adapting the calibrated or estimated weights, which are readily measured on all samples from the whole cohort. Finally, we provide computational code in R (R Foundation for Statistical Computing, Vienna, Austria) that can easily perform these methods.Results: Through numerical analyses of artificially simulated data and the National Wilms Tumor Study data, accurate risk ratio and risk difference estimates were obtained using the pseudo-Poisson and pseudo-normal linear regression methods. Also, using the auxiliary variable information from the whole cohort, precisions of these estimators were markedly improved.Conclusion: The ordinary logistic regression analyses may provide uninterpretable effect measure estimates, and the risk ratio and risk difference estimation methods are effective alternative approaches for case-cohort studies. These methods are especially recommended under situations in which the event rate is not low.
著者
Hisashi Noma
出版者
Japan Epidemiological Association
雑誌
Journal of Epidemiology (ISSN:09175040)
巻号頁・発行日
pp.JE20220251, (Released:2023-01-14)
参考文献数
8

Background: The logistic regression analysis proposed by Schouten et al. (Stat Med. 1993;12:1733–1745) has been a standard method in current statistical analysis of case-cohort studies, and it enables effective estimation of risk ratio from selected subsamples, with adjustment of potential confounding factors. Schouten et al. (1993) also proposed the standard error estimate of the risk ratio estimator can be calculated by the robust variance estimator, and this method has been widely adopted.Methods and Results: We show that the robust variance estimator does not account for the duplications of case and subcohort samples and generally has certain bias, i.e., inaccurate confidence intervals and P-values are possibly obtained. To address the invalid statistical inference problem, we provide an alternative bootstrap-based valid variance estimator. Through simulation studies, the bootstrap method consistently provided more precise confidence intervals compared with those provided by the robust variance method, while retaining adequate coverage probabilities.Conclusion: The robust variance estimator has certain bias, and inadequate conclusions might be deduced from the resultant statistical analyses. The proposed bootstrap variance estimator can provide more accurate and precise interval estimates. The bootstrap method would be an alternative effective approach in practice to provide accurate evidence.
著者
Hisashi Noma Kengo Nagashima Shogo Kato Satoshi Teramukai Toshi A. Furukawa
出版者
Japan Epidemiological Association
雑誌
Journal of Epidemiology (ISSN:09175040)
巻号頁・発行日
vol.32, no.10, pp.441-448, 2022-10-05 (Released:2022-10-05)
参考文献数
30
被引用文献数
1 3

Background: In meta-analysis, the normal distribution assumption has been adopted in most systematic reviews of random-effects distribution models due to its computational and conceptual simplicity. However, this restrictive model assumption is possibly unsuitable and might have serious influences in practices.Methods: We provide two examples of real-world evidence that clearly show that the normal distribution assumption is explicitly unsuitable. We propose new random-effects meta-analysis methods using five flexible random-effects distribution models that can flexibly regulate skewness, kurtosis and tailweight: skew normal distribution, skew t-distribution, asymmetric Subbotin distribution, Jones–Faddy distribution, and sinh–arcsinh distribution. We also developed a statistical package, flexmeta, that can easily perform these methods.Results: Using the flexible random-effects distribution models, the results of the two meta-analyses were markedly altered, potentially influencing the overall conclusions of these systematic reviews.Conclusion: The restrictive normal distribution assumption in the random-effects model can yield misleading conclusions. The proposed flexible methods can provide more precise conclusions in systematic reviews.