Modeling the rate of HIV testing from repeated binary data amidst potential never-testers.

Many longitudinal studies with a binary outcome measure involve a fraction of subjects with a homogeneous response profile. In our motivating data set, a study on the rate of human immunodeficiency virus (HIV) self-testing in a population of men who have sex with men (MSM), a substantial proportion of the subjects did not self-test during the follow-up study. The observed data in this context consist of a binary sequence for each subject indicating whether or not that subject experienced any events between consecutive observation time points, so subjects who never self-tested were observed to have a response vector consisting entirely of zeros. Conventional longitudinal analysis is not equipped to handle questions regarding the rate of events (as opposed to the odds, as in the classical logistic regression model). With the exception of discrete mixture models, such methods are also not equipped to handle settings in which there may exist a group of subjects for whom no events will ever occur, i.e. a so-called "never-responder" group. In this article, we model the observed data assuming that events occur according to some unobserved continuous-time stochastic process. In particular, we consider the underlying subject-specific processes to be Poisson conditional on some unobserved frailty, leading to a natural focus on modeling event rates. Specifically, we propose to use the power variance function (PVF) family of frailty distributions, which contains both the gamma and inverse Gaussian distributions as special cases and allows for the existence of a class of subjects having zero frailty. We generalize a computational algorithm developed for a log-gamma random intercept model (Conaway, 1990. A random effects model for binary data. Biometrics46, 317-328) to compute the exact marginal likelihood, which is then maximized to obtain estimates of model parameters. We conduct simulation studies, exploring the performance of the proposed method in comparison with competitors. Applying the PVF as well as a Gaussian random intercept model and a corresponding discrete mixture model to our motivating data set, we conclude that the group assigned to receive follow-up messages via SMS was self-testing at a significantly lower rate than the control group, but that there is no evidence to support the existence of a group of never-testers.