ArticlePloS one2015
Elicitation of expert prior opinion: application to the MYPAN trial in childhood polyarteritis nodosa.
Article in PloS one, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 19 papers.
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Who cites it
19 citing papers in PubMed, 50 citations in OpenAlex.
- Remote, bivariate prior elicitation for a Bayesian non-inferiority randomized controlled trial.Trials · 2026Trial
- Bayesian prior elicitation on the efficacy of medical therapies in perianal fistulizing Crohn's disease.Journal of Crohn's & colitis · 2026Article
- Bayesian trial of adalimumab versus secukinumab for children with juvenile idiopathic arthritis associated uveitis or chronic anterior uveitis.Pediatric rheumatology online journal · 2025Article
- Bayesian sample size determination using commensurate priors to leverage preexperimental data.Biometrics · 2023Article
- Bayesian adaptive design for pediatric clinical trials incorporating a community of prior beliefs.BMC medical research methodology · 2022Article
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- Prior Elicitation for Use in Clinical Trial Design and Analysis: A Literature Review.International journal of environmental research and public health · 2021Review
- Prior elicitation of the efficacy and tolerability of Methotrexate and Mycophenolate Mofetil in Juvenile Localised Scleroderma.AMRC open research · 2021Article
- Expert Elicitation for Latent Growth Curve Models: The Case of Posttraumatic Stress Symptoms Development in Children With Burn Injuries.Frontiers in psychology · 2020Article
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- Key issues in Rett syndrome: emotional, behavioural and autonomic dysregulation (EBAD) - a target for clinical trials.Orphanet journal of rare diseases · 2018Review
- Using a meta-narrative literature review and focus groups with key stakeholders to identify perceived challenges and solutions for generating robust evidence on the effectiveness of treatments for rare diseases.Orphanet journal of rare diseases · 2018Review
- [Hypertension and intermittent convulsions for one month in a school-age child].Zhongguo dang dai er ke za zhi = Chinese journal of contemporary pediatrics · 2017Article
- Article
- Optic Neuropathy Secondary to Polyarteritis Nodosa, Case Report, and Diagnostic Challenges.Frontiers in neurology · 2017Article
- Therapeutic advances in the treatment of vasculitis.Pediatric rheumatology online journal · 2016Review
- Rett Syndrome: Crossing the Threshold to Clinical Translation.Trends in neurosciences · 2016Review
- Bayesian methods for the design and interpretation of clinical trials in very rare diseases.Statistics in medicine · 2014Article
Corrections and comments
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Authors and funding
22 authors at 19 institutions in 9 countries.
Funding
Abstract
objectivesDefinitive sample sizes for clinical trials in rare diseases are usually infeasible. Bayesian methodology can be used to maximise what is learnt from clinical trials in these circumstances. We elicited expert prior opinion for a future Bayesian randomised controlled trial for a rare inflammatory paediatric disease, polyarteritis nodosa (MYPAN, Mycophenolate mofetil for polyarteritis nodosa).
methodsA Bayesian prior elicitation meeting was convened. Opinion was sought on the probability that a patient in the MYPAN trial treated with cyclophosphamide would achieve disease remission within 6-months, and on the relative efficacies of mycophenolate mofetil and cyclophosphamide. Expert opinion was combined with previously unseen data from a recently completed randomised controlled trial in ANCA associated vasculitis.
resultsA pan-European group of fifteen experts participated in the elicitation meeting. Consensus expert prior opinion was that the most likely rates of disease remission within 6 months on cyclophosphamide or mycophenolate mofetil were 74% and 71%, respectively. This prior opinion will now be taken forward and will be modified to formulate a Bayesian posterior opinion once the MYPAN trial data from 40 patients randomised 1:1 to either CYC or MMF become available.
conclusionsWe suggest that the methodological template we propose could be applied to trial design for other rare diseases.
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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.