Addressing an idiosyncrasy in estimating survival curves using double sampling in the presence of self-selected right censoring.
We investigate the use of follow-up samples of individuals to estimate survival curves from studies that are subject to right censoring from two sources: (i) early termination of the study, namely, administrative censoring, or (ii) censoring due to lost data prior to administrative censoring, so-called dropout. We assume that, for the full cohort of individuals, administrative censoring times are independent of the subjects' inherent characteristics, including survival time. To address the loss to censoring due to dropout, which we allow to be possibly selective, we consider an intensive second phase of the study where a representative sample of the originally lost subjects is subsequently followed and their data recorded. As with double-sampling designs in survey methodology, the objective is to provide data on a representative subset of the dropouts. Despite assumed full response from the follow-up sample, we show that, in general in our setting, administrative censoring times are not independent of survival times within the two subgroups, nondropouts and sampled dropouts. As a result, the stratified Kaplan-Meier estimator is not appropriate for the cohort survival curve. Moreover, using the concept of potential outcomes, as opposed to observed outcomes, and thereby explicitly formulating the problem as a missing data problem, reveals and addresses these complications. We present an estimation method based on the likelihood of an easily observed subset of the data and study its properties analytically for large samples. We evaluate our method in a realistic situation by simulating data that match published margins on survival and dropout from an actual hip-replacement study. Limitations and extensions of our design and analytic method are discussed.
Frangakis
C E
CE
Department of Biostatistics, Johns Hopkins University, Baltimore, Maryland 21205, USA. cfrangak@jhsph.edu
Rubin
D B
DB
D019644:Arthroplasty, Replacement, Hip; D005500:Follow-Up Studies; D006801:Humans; D016013:Likelihood Functions; D010352:Patient Dropouts; D011336:Probability; D013223:Statistics as Topic / Q000379:methods; D013514:Surgical Procedures, Operative; D016019:Survival Analysis; D016896:Treatment Outcome