Journal article
Development and validation of minimal disease activity, disease flare, and minimal clinically important difference for children with chronic nonbacterial osteomyelitis using a consensus and data-driven approach
Annals of the rheumatic diseases
08/08/2026
DOI: 10.1016/j.ard.2026.07.006
PMID: 42570905
Abstract
To create definitions for minimal disease activity (MDA), flare, and minimal clinically important difference (MCID) for chronic nonbacterial osteomyelitis (CNO). It is necessary to establish these criteria before starting clinical effectiveness trials for therapies in CNO.
Three separate cohorts’ samples from an international observational CNO registry were presented to a panel of 20 experts in CNO, including 7 patients/caregivers via online and in-person voting using the group consensus voting technique to define MDA, flare, and MCID, respectively. Experts classified the cases in each cohort as meeting the state for that specific cohort, ie, MDA or not, or flare or not, or meeting MCID or not. A consensus of ≥80% was required.
General surveys identified the most important variables to include to define MDA, flare, and MCID. Clinical improvement of ≥30% in these critical parameters and improvement of the CNO Clinical Disease Activity Score by at least 3 were considered meaningful by providers and consensus data.
This study provides the preliminary definitions of MDA, disease flare, and MCID in CNO, which can serve as targets and potential outcomes from treatments. These definitions must now be validated in other cohorts and tested in clinical trials.
Details
- Title: Subtitle
- Development and validation of minimal disease activity, disease flare, and minimal clinically important difference for children with chronic nonbacterial osteomyelitis using a consensus and data-driven approach
- Creators
- Farzana Nuruzzaman - Stony Brook UniversityNatalie Rosenwaser - University of WashingtonXing Wang - Center for Infectious Disease ResearchJonathan D. Akikusa - Royal Children's HospitalMatthew L. Basiaga - Mayo ClinicAva Klein - University of WashingtonIan Muse - University of WashingtonErin Balay-Dustrude - University of WashingtonMegan Nguyen - University of WashingtonEmily Deng - University of WashingtonAleksander Lenert - University of IowaLindsey Bergstrom - National Patient Safety FoundationFatma Dedeoglu - Boston Children's HospitalBin Huang - Cincinnati Children's Hospital Medical CenterJenna King - National Patient Safety FoundationSivia Lapidus - Hackensack University Medical CenterTzielan C. Lee - Stanford UniversityCassandra M. Levesque - National Patient Safety FoundationLillian Lim - Stollery Children's HospitalKimberly Martin - National Patient Safety FoundationElizabeth Murray - National Patient Safety FoundationMelissa S. Oliver - Riley Hospital for ChildrenKaren B. Onel - Hospital for Special SurgerySeza Ozen - Hacettepe UniversityLauren Potts - National Patient Safety FoundationSara M. Stern - University of UtahRobin Villaverda - National Patient Safety FoundationEveline Y. Wu - University of North Carolina at Chapel HillRonald M. Laxer - Hospital for Sick ChildrenPolly J. Ferguson - University of IowaDaniel J. Lovell - Cincinnati Children's Hospital Medical CenterYongdong Zhao - University of WashingtonCHOIR network and CARRA CNO Workgroup
- Resource Type
- Journal article
- Publication Details
- Annals of the rheumatic diseases
- DOI
- 10.1016/j.ard.2026.07.006
- PMID
- 42570905
- NLM abbreviation
- Ann Rheum Dis
- ISSN
- 0003-4967
- eISSN
- 1468-2060
- Publisher
- Elsevier B.V
- Language
- English
- Electronic publication date
- 08/08/2026
- Academic Unit
- Stead Family Department of Pediatrics; Rheumatology, Allergy, and Immunology; Immunology; Internal Medicine
- Record Identifier
- 9985218633702771
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