Approaches to multiplicity in publicly funded pragmatic randomised controlled trials: a survey of clinical trials units and a rapid review of published trials

Katie E Pike*, Barnaby C Reeves, Chris A Rogers

*Corresponding author for this work

Research output: Contribution to journalArticle (Academic Journal)peer-review

6 Citations (Scopus)
121 Downloads (Pure)

Abstract

Background
Opinions and practices vary around the issue of performing multiple statistical tests in randomised controlled trials (RCTs). We carried out a study to collate information about opinions and practices using a methodological systematic rapid review and a survey, specifically of publicly funded pragmatic RCTs that are not seeking marketing authorisation. The aim was to identify the circumstances under which researchers would make a statistical adjustment for multiplicity.

Methods
A methodological systematic review was performed extracting information from articles reporting primary analyses of pragmatic RCTs in one of seven high quality medical journals, in January to June (inclusive) 2018. A survey (Survey Monkey) eliciting opinions and practices around multiplicity was distributed to the 47 registered clinical trials units (CTUs) in the UK.

Results
One hundred and thirty-eight RCTs were included in the systematic review, and survey responses were received from 27/47 (57%) CTUs. Both the systematic review and survey indicated that adjusting for multiplicity was considered most important for multiple treatment comparisons; adjustment was performed for 11/23 (48%) published trials, and 24/27 (89%) CTU statisticians reported they would consider adjustment. Opinions and practices varied around adjustment for multiplicity arising from multiple primary outcomes and interim analyses. Adjustment was considered less important for multiplicity due to multiple secondary outcomes (adjustment performed for 17/136 [13%] published trials and 3/27 [11%] CTU statisticians would consider adjustment) and subgroup analyses (8/85 [9%] published trials adjusted and 6/27 CTU [22%] statisticians would consider adjustment).

Conclusions
There is variation in opinions about adjustment for multiplicity among both statisticians reporting RCTs and applied statisticians working in CTUs. Further guidance is needed on the circumstances in which adjustment should be considered in relation to primary trial hypotheses, and if there are any situations in which adjustment would be recommended in the context of secondary analyses.
Original languageEnglish
Article number39
Number of pages11
JournalBMC Medical Research Methodology
Volume22
Issue number1
Early online date6 Feb 2022
DOIs
Publication statusPublished - 6 Feb 2022

Bibliographical note

Funding Information:
Thank you to the researchers at each CTU for completing the survey, and the kind assistance of the UKCRC CTU statistics group in distributing the survey.

Funding Information:
This research is funded by the National Institute for Health Research (NIHR Doctoral Research Fellowship, Miss Katie Pike, DRF-2017-10-126).

Publisher Copyright:
© 2022, The Author(s).

Keywords

  • Rapid review
  • Survey
  • multiple testing
  • Randomised controlled trials

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