A stepped wedge design for testing an effect of intranasal insulin on cognitive development of children with Phelan-McDermid syndrome: A comparison of different designs

Stat Methods Med Res. 2017 Apr;26(2):766-775. doi: 10.1177/0962280214558864. Epub 2014 Nov 19.

Abstract

This paper compares the power of the parallel group design, the matched-pairs design, and several options for the stepped wedge and delayed start designs for testing a possible effect of intranasal insulin with respect to placebo on developmental growth of children with a rare disorder like Phelan-McDermid syndrome. A subject-specific linear mixed effects model for the primary outcome developmental age in a longitudinal setting with five time points was assumed. Monte Carlo simulation studies with small sample sizes were applied since the rare disorder prohibits large trials. The stepped wedge designs, which were initially preferred for ethical reasons, appear to be competitive in power to other designs and were in some settings even the best. The assumed statistical model also demonstrates that all of the designs can be viewed as a stepped wedge or delayed treatment design. Our results show that the stepped wedge design is an appropriate alternative for randomized controlled trials on developmental growth with small numbers of participants under the formulated statistical conditions.

Keywords: Developmental growth; Phelan-McDermid syndrome; delayed start design; random coefficient model; small trial designs; stepped wedge design.

Publication types

  • Comparative Study
  • Review
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Administration, Intranasal
  • Biostatistics / methods
  • Child
  • Child Development / drug effects
  • Chromosome Deletion
  • Chromosome Disorders / drug therapy*
  • Chromosome Disorders / psychology*
  • Chromosomes, Human, Pair 22
  • Cognition / drug effects
  • Computer Simulation
  • Humans
  • Insulin / administration & dosage*
  • Linear Models
  • Longitudinal Studies
  • Monte Carlo Method
  • Randomized Controlled Trials as Topic / methods*
  • Randomized Controlled Trials as Topic / statistics & numerical data
  • Sample Size

Substances

  • Insulin

Supplementary concepts

  • Telomeric 22q13 Monosomy Syndrome