TY - JOUR
T1 - Information anchored reference-based sensitivity analysis for truncated normal data with application to survival analysis
AU - Atkinson, Andrew
AU - Cro, Suzie
AU - Carpenter, James R.
AU - Kenward, Michael G.
N1 - Publisher Copyright:
© 2021 The Authors. Statistica Neerlandica published by John Wiley & Sons Ltd on behalf of Netherlands Society for Statistics and Operations Research.
PY - 2021/11
Y1 - 2021/11
N2 - The primary analysis of time-to-event data typically makes the censoring at random assumption, that is, that—conditional on covariates in the model—the distribution of event times is the same, whether they are observed or unobserved. In such cases, we need to explore the robustness of inference to more pragmatic assumptions about patients post-censoring in sensitivity analyses. Reference-based multiple imputation, which avoids analysts explicitly specifying the parameters of the unobserved data distribution, has proved attractive to researchers. Building on results for longitudinal continuous data, we show that inference using a Tobit regression imputation model for reference-based sensitivity analysis with right censored log normal data is information anchored, meaning the proportion of information lost due to missing data under the primary analysis is held constant across the sensitivity analyses. We illustrate our theoretical results using simulation and a clinical trial case study.
AB - The primary analysis of time-to-event data typically makes the censoring at random assumption, that is, that—conditional on covariates in the model—the distribution of event times is the same, whether they are observed or unobserved. In such cases, we need to explore the robustness of inference to more pragmatic assumptions about patients post-censoring in sensitivity analyses. Reference-based multiple imputation, which avoids analysts explicitly specifying the parameters of the unobserved data distribution, has proved attractive to researchers. Building on results for longitudinal continuous data, we show that inference using a Tobit regression imputation model for reference-based sensitivity analysis with right censored log normal data is information anchored, meaning the proportion of information lost due to missing data under the primary analysis is held constant across the sensitivity analyses. We illustrate our theoretical results using simulation and a clinical trial case study.
KW - Rubin's rules
KW - censoring not at random
KW - informative censoring
KW - reference-based multiple imputation
KW - sensitivity analysis
KW - tobit regression
KW - truncated normal data
UR - http://www.scopus.com/inward/record.url?scp=85107995669&partnerID=8YFLogxK
U2 - 10.1111/stan.12250
DO - 10.1111/stan.12250
M3 - Article
AN - SCOPUS:85107995669
SN - 0039-0402
VL - 75
SP - 500
EP - 523
JO - Statistica Neerlandica
JF - Statistica Neerlandica
IS - 4
ER -