A joint model for hierarchical continuous and zero-inflated overdispersed count data

Many applications in public health, medical and biomedical or other studies demand modelling of two or more longitudinal outcomes jointly to get better insight into their joint evolution. In this regard, a joint model for a longitudinal continuous and a count sequence, the latter possibly overdispersed and zero-inflated (ZI), will be specified that assembles aspects coming from each one of them into one single model. Further, a subject-specific random effect is included to account for the correlation in the continuous outcome. For the count outcome, clustering and overdispersion are accommodated through two distinct sets of random effects in a generalized linear model as proposed by Molenberghs et al. [A family of generalized linear models for repeated measures with normal and conjugate random effects. Stat Sci. 2010;25:325–347]; one is normally distributed, the other conjugate to the outcome distribution. The association among the two sequences is captured by correlating the normal random effects describing the continuous and count outcome sequences, respectively. An excessive number of zero counts is often accounted for by using a so-called ZI or hurdle model. ZI models combine either a Poisson or negative-binomial model with an atom at zero as a mixture, while the hurdle model separately handles the zero observations and the positive counts. This paper proposes a general joint modelling framework in which all these features can appear together. We illustrate the proposed method with a case study and examine it further with simulations.     

A joint marginalized multilevel model for longitudinal outcomes

The shared-parameter model and its so-called hierarchical or random-effects extension are widely used joint modeling approaches for a combination of longitudinal continuous, binary, count, missing, and survival outcomes that naturally occurs in many clinical and other studies. A random effect is introduced and shared or allowed to differ between two or more repeated measures or longitudinal outcomes, thereby acting as a vehicle to capture association between the outcomes in these joint models. It is generally known that parameter estimates in a linear mixed model (LMM) for continuous repeated measures or longitudinal outcomes allow for a marginal interpretation, even though a hierarchical formulation is employed. This is not the case for the generalized linear mixed model (GLMM), that is, for non-Gaussian outcomes. The aforementioned joint models formulated for continuous and binary or two longitudinal binomial outcomes, using the LMM and GLMM, will naturally have marginal interpretation for parameters associated with the continuous outcome but a subject-specific interpretation for the fixed effects parameters relating covariates to binary outcomes. To derive marginally meaningful parameters for the binary models in a joint model, we adopt the marginal multilevel model (MMM) due to Heagerty [13] and Heagerty and Zeger [14] and formulate a joint MMM for two longitudinal responses. This enables to (1) capture association between the two responses and (2) obtain parameter estimates that have a population-averaged interpretation for both outcomes. The model is applied to two sets of data. The results are compared with those obtained from the existing approaches such as generalized estimating equations, GLMM, and the model of Heagerty [13]. Estimates were found to be very close to those from single analysis per outcome but the joint model yields higher precision and allows for quantifying the association between outcomes. Parameters were estimated using maximum likelihood. The model is easy to fit using available tools such as the SAS NLMIXED procedure.     

Score test for testing zero-inflated Poisson regression against zero-inflated generalized Poisson alternatives

In several cases, count data often have excessive number of zero outcomes. This zero-inflated phenomenon is a specific cause of overdispersion, and zero-inflated Poisson regression model (ZIP) has been proposed for accommodating zero-inflated data. However, if the data continue to suggest additional overdispersion, zero-inflated negative binomial (ZINB) and zero-inflated generalized Poisson (ZIGP) regression models have been considered as alternatives. This study proposes the score test for testing ZIP regression model against ZIGP alternatives and proves that it is equal to the score test for testing ZIP regression model against ZINB alternatives. The advantage of using the score test over other alternative tests such as likelihood ratio and Wald is that the score test can be used to determine whether a more complex model is appropriate without fitting the more complex model. Applications of the proposed score test on several datasets are also illustrated.     

Analysis of mixed correlated bivariate zero-inflated count and (k,l)-inflated beta responses with application to social network datasets

This paper presents a new model that monitors the basic network formation mechanisms via the attributes through time. It considers the issue of joint modeling of longitudinal inflated (0, 1)-support continuous and inflated count response variables. For joint model of mentioned response variables, a correlated generalized linear mixed model is studied. The fraction response is inflated in two points k and l (k < l) and a k and l inflated beta distribution is introduced to use as its distribution. Also, the count response is inflated in zero and we use some members of zero-inflated power series distributions, hurdle-at-zero, members of zero-inflated double power series distributions and zero-inflated generalized Poisson distribution as our count response distribution. A full likelihood-based approach is used to yield maximum likelihood estimates of the model parameters and the model is applied to a real social network obtained from an observational study where the rate of the ith node’s responsiveness to the jth node and the number of arrows or edges with some specific characteristics from the ith node to the jth node are the correlated inflated (0, 1)-support continuous and inflated count response variables, respectively. The effect of the sender and receiver positions in an office environment on the responses are investigated simultaneously.     

Bivariate zero-inflated negative binomial regression model with applications

Count data often display excessive number of zero outcomes than are expected in the Poisson regression model. The zero-inflated Poisson regression model has been suggested to handle zero-inflated data, whereas the zero-inflated negative binomial (ZINB) regression model has been fitted for zero-inflated data with additional overdispersion. For bivariate and zero-inflated cases, several regression models such as the bivariate zero-inflated Poisson (BZIP) and bivariate zero-inflated negative binomial (BZINB) have been considered. This paper introduces several forms of nested BZINB regression model which can be fitted to bivariate and zero-inflated count data. The mean–variance approach is used for comparing the BZIP and our forms of BZINB regression model in this study. A similar approach was also used by past researchers for defining several negative binomial and zero-inflated negative binomial regression models based on the appearance of linear and quadratic terms of the variance function. The nested BZINB regression models proposed in this study have several advantages; the likelihood ratio tests can be performed for choosing the best model, the models have flexible forms of marginal mean–variance relationship, the models can be fitted to bivariate zero-inflated count data with positive or negative correlations, and the models allow additional overdispersion of the two dependent variables.     

Joint modeling of mixed skewed continuous and ordinal longitudinal responses: a Bayesian approach

In this paper, a joint model for analyzing multivariate mixed ordinal and continuous responses, where continuous outcomes may be skew, is presented. For modeling the discrete ordinal responses, a continuous latent variable approach is considered and for describing continuous responses, a skew-normal mixed effects model is used. A Bayesian approach using Markov Chain Monte Carlo (MCMC) is adopted for parameter estimation. Some simulation studies are performed for illustration of the proposed approach. The results of the simulation studies show that the use of the separate models or the normal distributional assumption for shared random effects and within-subject errors of continuous and ordinal variables, instead of the joint modeling under a skew-normal distribution, leads to biased parameter estimates. The approach is used for analyzing a part of the British Household Panel Survey (BHPS) data set. Annual income and life satisfaction are considered as the continuous and the ordinal longitudinal responses, respectively. The annual income variable is severely skewed, therefore, the use of the normality assumption for the continuous response does not yield acceptable results. The results of data analysis show that gender, marital status, educational levels and the amount of money spent on leisure have a significant effect on annual income, while marital status has the highest impact on life satisfaction.     

Likelihood estimation for longitudinal zero-inflated power series regression models

In this paper, a zero-inflated power series regression model for longitudinal count data with excess zeros is presented. We demonstrate how to calculate the likelihood for such data when it is assumed that the increment in the cumulative total follows a discrete distribution with a location parameter that depends on a linear function of explanatory variables. Simulation studies indicate that this method can provide improvements in obtaining standard errors of the estimates. We also calculate the dispersion index for this model. The influence of a small perturbation of the dispersion index of the zero-inflated model on likelihood displacement is also studied. The zero-inflated negative binomial regression model is illustrated on data regarding joint damage in psoriatic arthritis.     

Estimation of Median in Two-Phase Sampling Using Two Auxiliary Variables

In recent years, zero-inflated count data models, such as zero-inflated Poisson (ZIP) models, are widely used as the count data with extra zeros are very common in many practical problems. In order to model the correlated count data which are either clustered or repeated and to assess the effects of continuous covariates or of time scales in a flexible way, a class of semiparametric mixed-effects models for zero-inflated count data is considered. In this article, we propose a fully Bayesian inference for such models based on a data augmentation scheme that reflects both random effects of covariates and mixture of zero-inflated distribution. A computational efficient MCMC method which combines the Gibbs sampler and M-H algorithm is implemented to obtain the estimate of the model parameters. Finally, a simulation study and a real example are used to illustrate the proposed methodologies.     

Prediction of transplant-free survival in idiopathic pulmonary fibrosis patients using joint models for event times and mixed multivariate longitudinal data

We implement a joint model for mixed multivariate longitudinal measurements, applied to the prediction of time until lung transplant or death in idiopathic pulmonary fibrosis. Specifically, we formulate a unified Bayesian joint model for the mixed longitudinal responses and time-to-event outcomes. For the longitudinal model of continuous and binary responses, we investigate multivariate generalized linear mixed models using shared random effects. Longitudinal and time-to-event data are assumed to be independent conditional on available covariates and shared parameters. A Markov chain Monte Carlo algorithm, implemented in OpenBUGS, is used for parameter estimation. To illustrate practical considerations in choosing a final model, we fit 37 different candidate models using all possible combinations of random effects and employ a deviance information criterion to select a best-fitting model. We demonstrate the prediction of future event probabilities within a fixed time interval for patients utilizing baseline data, post-baseline longitudinal responses, and the time-to-event outcome. The performance of our joint model is also evaluated in simulation studies.     

Bayesian zero-inflated generalized Poisson regression model: estimation and case influence diagnostics

Count data with excess zeros arises in many contexts. Here our concern is to develop a Bayesian analysis for the zero-inflated generalized Poisson (ZIGP) regression model to address this problem. This model provides a useful generalization of zero-inflated Poisson model since the generalized Poisson distribution is overdispersed/underdispersed relative to Poisson. Due to the complexity of the ZIGP model, Markov chain Monte Carlo methods are used to develop a Bayesian procedure for the considered model. Additionally, some discussions on the model selection criteria are presented and a Bayesian case deletion influence diagnostics is investigated for the joint posterior distribution based on the Kullback–Leibler divergence. Finally, a simulation study and a psychological example are given to illustrate our methodology.     

GEE-based zero-inflated generalized Poisson model for clustered over or under-dispersed count data

The zero-inflated regression models such as zero-inflated Poisson (ZIP), zero-inflated negative binomial (ZINB) or zero-inflated generalized Poisson (ZIGP) regression models can model the count data with excess zeros. The ZINB model can handle over-dispersed and the ZIGP model can handle the over or under-dispersed count data with excess zeros as well. Moreover, the count data may be correlated because of data collection procedure or special study design. The clustered sampling approach is one of the examples in which the correlation among subjects could be defined. In such situations, a marginal model using generalized estimating equation (GEE) approach can incorporate these correlations and lead up to the relationships at the population level. In this study, the GEE-based zero-inflated generalized Poisson regression model was proposed to fit over and under-dispersed clustered count data with excess zeros.     

Decision tree approaches for zero-inflated count data

There have been many methodologies developed about zero-inflated data in the field of statistics. However, there is little literature in the data mining fields, even though zero-inflated data could be easily found in real application fields. In fact, there is no decision tree method that is suitable for zero-inflated responses. To analyze continuous target variable with decision trees as one of data mining techniques, we use F-statistics (CHAID) or variance reduction (CART) criteria to find the best split. But these methods are only appropriate to a continuous target variable. If the target variable is rare events or zero-inflated count data, the above criteria could not give a good result because of its attributes. In this paper, we will propose a decision tree for zero-inflated count data, using a maximum of zero-inflated Poisson likelihood as the split criterion. In addition, using well-known data sets we will compare the performance of the split criteria. In the case when the analyst is interested in lower value groups (e.g. no defect areas, customers who do not claim), the suggested ZIP tree would be more efficient.     

A Bayesian Approach for Zero-Inflated Count Regression Models by Using the Reversible Jump Markov Chain Monte Carlo Method and an Application

In this study, estimation of the parameters of the zero-inflated count regression models and computations of posterior model probabilities of the log-linear models defined for each zero-inflated count regression models are investigated from the Bayesian point of view. In addition, determinations of the most suitable log-linear and regression models are investigated. It is known that zero-inflated count regression models cover zero-inflated Poisson, zero-inflated negative binomial, and zero-inflated generalized Poisson regression models. The classical approach has some problematic points but the Bayesian approach does not have similar flaws. This work points out the reasons for using the Bayesian approach. It also lists advantages and disadvantages of the classical and Bayesian approaches. As an application, a zoological data set, including structural and sampling zeros, is used in the presence of extra zeros. In this work, it is observed that fitting a zero-inflated negative binomial regression model creates no problems at all, even though it is known that fitting a zero-inflated negative binomial regression model is the most problematic procedure in the classical approach. Additionally, it is found that the best fitting model is the log-linear model under the negative binomial regression model, which does not include three-way interactions of factors.     

零膨胀计数数据的联合建模及变量选择

零膨胀计数数据破坏了泊松分布的方差-均值关系，可由取值服从泊松分布的数据和取值为零(退化分布)的数据各占一定比例所构成的混合分布所解释。本文基于自适应弹性网技术， 研究了零膨胀计数数据的联合建模及变量选择问题.对于零膨胀泊松分布，引入潜变量，构造出零膨胀泊松模型的完全似然, 其中由零膨胀部分和泊松部分两项组成.考虑到协变量可能存在共线性和稀疏性，通过对似然函数加自适应弹性网惩罚得到目标函数,然后利用EM算法得到回归系数的稀疏估计量，并用贝叶斯信息准则BIC来确定最优调节参数.本文也给出了估计量的大样本性质的理论证明和模拟研究，最后把所提出的方法应用到实际问题中。     

Bayesian joint modeling of correlated counts data with application to adverse birth outcomes

In disease mapping, health outcomes measured at the same spatial locations may be correlated, so one can consider joint modeling the multivariate health outcomes accounting for their dependence. The general approaches often used for joint modeling include shared component models and multivariate models. An alternative way to model the association between two health outcomes, when one outcome can naturally serve as a covariate of the other, is to use ecological regression model. For example, in our application, preterm birth (PTB) can be treated as a predictor for low birth weight (LBW) and vice versa. Therefore, we proposed to blend the ideas from joint modeling and ecological regression methods to jointly model the relative risks for LBW and PTBs over the health districts in Saskatchewan, Canada, in 2000–2010. This approach is helpful when proxy of areal-level contextual factors can be derived based on the outcomes themselves when direct information on risk factors are not readily available. Our results indicate that the proposed approach improves the model fit when compared with the conventional joint modeling methods. Further, we showed that when no strong spatial autocorrelation is present, joint outcome modeling using only independent error terms can still provide a better model fit when compared with the separate modeling.     

Untangle the structural and random zeros in statistical modelings

Count data with structural zeros are common in public health applications. There are considerable researches focusing on zero-inflated models such as zero-inflated Poisson (ZIP) and zero-inflated Negative Binomial (ZINB) models for such zero-inflated count data when used as response variable. However, when such variables are used as predictors, the difference between structural and random zeros is often ignored and may result in biased estimates. One remedy is to include an indicator of the structural zero in the model as a predictor if observed. However, structural zeros are often not observed in practice, in which case no statistical method is available to address the bias issue. This paper is aimed to fill this methodological gap by developing parametric methods to model zero-inflated count data when used as predictors based on the maximum likelihood approach. The response variable can be any type of data including continuous, binary, count or even zero-inflated count responses. Simulation studies are performed to assess the numerical performance of this new approach when sample size is small to moderate. A real data example is also used to demonstrate the application of this method.     

A Bayesian analysis of clustered discrete and continuous outcomes

Many study designs yield a variety of outcomes from each subject clustered within an experimental unit. When these outcomes are of mixed data types, it is challenging to jointly model the effects of covariates on the responses using traditional methods. In this paper, we develop a Bayesian approach for a joint regression model of the different outcome variables and show that the fully conditional posterior distributions obtained under the model assumptions allow for estimation of posterior distributions using Gibbs sampling algorithm.     

A general joint model for longitudinal measurements and competing risks survival data with heterogeneous random effects

This article studies a general joint model for longitudinal measurements and competing risks survival data. The model consists of a linear mixed effects sub-model for the longitudinal outcome, a proportional cause-specific hazards frailty sub-model for the competing risks survival data, and a regression sub-model for the variance–covariance matrix of the multivariate latent random effects based on a modified Cholesky decomposition. The model provides a useful approach to adjust for non-ignorable missing data due to dropout for the longitudinal outcome, enables analysis of the survival outcome with informative censoring and intermittently measured time-dependent covariates, as well as joint analysis of the longitudinal and survival outcomes. Unlike previously studied joint models, our model allows for heterogeneous random covariance matrices. It also offers a framework to assess the homogeneous covariance assumption of existing joint models. A Bayesian MCMC procedure is developed for parameter estimation and inference. Its performances and frequentist properties are investigated using simulations. A real data example is used to illustrate the usefulness of the approach.     

Joint generalized estimating equations for multivariate longitudinal binary outcomes with missing data: an application to acquired immune deficiency syndrome data

Summary.  In a large, prospective longitudinal study designed to monitor cardiac abnormalities in children born to women who are infected with the human immunodeficiency virus, instead of a single outcome variable, there are multiple binary outcomes (e.g. abnormal heart rate, abnormal blood pressure and abnormal heart wall thickness) considered as joint measures of heart function over time. In the presence of missing responses at some time points, longitudinal marginal models for these multiple outcomes can be estimated by using generalized estimating equations (GEEs), and consistent estimates can be obtained under the assumption of a missingness completely at random mechanism. When the missing data mechanism is missingness at random, i.e. the probability of missing a particular outcome at a time point depends on observed values of that outcome and the remaining outcomes at other time points, we propose joint estimation of the marginal models by using a single modified GEE based on an EM-type algorithm. The method proposed is motivated by the longitudinal study of cardiac abnormalities in children who were born to women infected with the human immunodeficiency virus, and analyses of these data are presented to illustrate the application of the method. Further, in an asymptotic study of bias, we show that, under a missingness at random mechanism in which missingness depends on all observed outcome variables, our joint estimation via the modified GEE produces almost unbiased estimates, provided that the correlation model has been correctly specified, whereas estimates from standard GEEs can lead to substantial bias.     

A unified approach to multilevel sample selection models

Abstract

We propose a unified approach for multilevel sample selection models using a generalized result on skew distributions arising from selection. If the underlying distributional assumption is normal, then the resulting density for the outcome is the continuous component of the sample selection density and has links with the closed skew-normal distribution (CSN). The CSN distribution provides a framework which simplifies the derivation of the conditional expectation of the observed data. This generalizes the Heckman’s two-step method to a multilevel sample selection model. Finite-sample performance of the maximum likelihood estimator of this model is studied through a Monte Carlo simulation.