Department of Biostatistics and Bioinformatics, Duke University, Durham, North Carolina, USA.
Duke Global Health Institute, Duke University, Durham, North Carolina, USA.
Stat Med. 2022 May 10;41(10):1862-1883. doi: 10.1002/sim.9333. Epub 2022 Feb 10.
A practical limitation of cluster randomized controlled trials (cRCTs) is that the number of available clusters may be small, resulting in an increased risk of baseline imbalance under simple randomization. Constrained randomization overcomes this issue by restricting the allocation to a subset of randomization schemes where sufficient overall covariate balance across comparison arms is achieved. However, for multi-arm cRCTs, several design and analysis issues pertaining to constrained randomization have not been fully investigated. Motivated by an ongoing multi-arm cRCT, we elaborate the method of constrained randomization and provide a comprehensive evaluation of the statistical properties of model-based and randomization-based tests under both simple and constrained randomization designs in multi-arm cRCTs, with varying combinations of design and analysis-based covariate adjustment strategies. In particular, as randomization-based tests have not been extensively studied in multi-arm cRCTs, we additionally develop most-powerful randomization tests under the linear mixed model framework for our comparisons. Our results indicate that under constrained randomization, both model-based and randomization-based analyses could gain power while preserving nominal type I error rate, given proper analysis-based adjustment for the baseline covariates. Randomization-based analyses, however, are more robust against violations of distributional assumptions. The choice of balance metrics and candidate set sizes and their implications on the testing of the pairwise and global hypotheses are also discussed. Finally, we caution against the design and analysis of multi-arm cRCTs with an extremely small number of clusters, due to insufficient degrees of freedom and the tendency to obtain an overly restricted randomization space.
集群随机对照试验(cRCTs)的一个实际限制是可用的集群数量可能较少,这导致在简单随机化下基线失衡的风险增加。约束随机化通过将分配限制在达到足够的总体协变量平衡的随机化方案的子集来克服这个问题。然而,对于多臂 cRCTs,涉及约束随机化的几个设计和分析问题尚未得到充分研究。受正在进行的多臂 cRCT 的启发,我们详细阐述了约束随机化的方法,并在多臂 cRCTs 中对简单和约束随机化设计下基于模型和基于随机化的检验的统计特性进行了全面评估,同时考虑了不同的设计和基于分析的协变量调整策略的组合。特别是,由于基于随机化的检验在多臂 cRCTs 中尚未得到广泛研究,我们还在线性混合模型框架下为我们的比较开发了最有力的基于随机化的检验。我们的结果表明,在约束随机化下,基于模型和基于随机化的分析都可以在保持名义第一类错误率的情况下获得更高的功效,前提是对基线协变量进行适当的基于分析的调整。然而,基于随机化的分析对分布假设的违反更具鲁棒性。还讨论了平衡度量和候选集大小的选择及其对成对和全局假设检验的影响。最后,我们警告不要对集群数量非常少的多臂 cRCTs 进行设计和分析,因为自由度不足和倾向于获得过于受限的随机化空间。