Noncompletion and nonpublication of trials studying rare diseases: A cross-sectional analysis.
Chris A ReesNatalie PicaMichael C MonuteauxFlorence T BourgeoisPublished in: PLoS medicine (2019)
In this study, over half of clinical trials initiated for rare diseases were either discontinued or not published 4 years after completion, resulting in large numbers of patients with rare diseases exposed to interventions that did not lead to informative findings. Concerted efforts are needed to ensure that participation of patients in rare disease trials advances scientific knowledge and treatments for rare diseases.