Establishment of an outcome measurement set for Chronic Nonbacterial Osteomyelitis and Synovitis, Acne, Pustulosis, Hyperostosis and Osteitis syndrome through the OMERACT Framework

The CNO/SAPHO OMERACT working group aims to develop a core domain set and core outcome measurement set for all clinical trials of patients with CNO and SAPHO. We completed a scoping review, virtual online focus groups, and an online discussion board and compiled a list of candidate core domain items. Next steps include working on narrowing down this list and distributing Delphi surveys.

Contributors

Melissa Oliver (PI), Philip Mease (supervisor), Yongdong (Dan) Zhao (supervisor), Jonathan Akikusa (supervisor), Christian Hedrich (supervisor), Bethany Welc (patient/parent research partner), Alex Theos (patient/parent research partner), and Beverly Shea (methodology expert)

Further Study Information

Current Stage: Ongoing
Date: January 2019 - December 2029
Funding source(s): Childhood Arthritis and Rheumatology Research Alliance-Arthritis Foundation (2022-2024)


Health Area

Disease Category: Rheumatology

Disease Name: SAPHO (synovitis, acne, pustulosis, hyperostosis, and osteitis) syndrome, Chronic Nonbacterial Osteomyelitis

Target Population

Age Range: 5 - 70

Sex: Either

Nature of Intervention: Any

Stakeholders Involved

- Clinical experts
- Conference participants
- Consumers (caregivers)
- Consumers (patients)
- Families
- Patient/ support group representatives
- Pharmaceutical industry representatives
- Researchers
- Service providers

Study Type

- COS for clinical trials or clinical research
- COS for practice
- Recommendations for outcome measures (measurement/how)

Method(s)

- Consensus conference
- Consensus meeting
- Delphi process
- Focus group(s)
- Interview
- Literature review

The OMERACT CNO/SAPHO working group has generated their preliminary list of a core domain set with definitions for each candidate item and is ready to start the Delphi survey process. Our working group started in 2019 with a scoping review performed. Please see the attached draft manuscript for more details on the scoping review. This scoping review generated over 200 candidate domains for the core domain set. This list was narrowed down to 91 over a year through discussions at our bi-monthly working group virtual meetings. Duplicates were removed and similar items were grouped and categorized. Then a smaller group that more rigorously brought the list down to 42 items through a similar process. Following this in 2022 we held the qualitative methods portion of the project with virtual focus groups and an online discussion board. The focus group sessions initially generated 79 candidate domain items and the online discussion board generated 28 items. The focus group list was narrowed down to 44 items again through discussions at our virtual bi-monthly working group meetings. Going into our SIG session at the OMERACT 2023 meeting in Colorado Springs, CO, USA, we had 42 items from the scoping review, 44 items from the focus group, and 28 items from the online discussion board. During the SIG session, we had two breakout groups and additionally narrowed down the list through discussion. We also had 2 working group members participate virtually and individually reduced the list. Immediately following the SIG session, we successfully brought the list down to 35 candidate domain items. I then reviewed the SIG session breakout results along with the 2 individual working group member’s results and a few additional items were ultimately added to the list, bringing it back to 48 items. For the past year, this list was again reviewed and discussed by the working group at our bimonthly meetings. Similar items were grouped (binned), any unclear or duplicate items were removed and the final 36 items were drafted with their preliminary definitions.

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