Identifying domains for CNO and SAPHO: A scoping review to create domains from existing outcomes by the OMERACT CNO and SAPHO working group

Melissa Oliver; Farzana Nuruzzaman; Aleksander Lenert; Arundathi Jayatilleke; Alexander Theos; Natalia Palmou-Fontana; Alissa Skinner; Marinka Twilt; Eveline Y. Wu; Cassyanne Aguiar; Samir Shah; Micol Romano; Suzanne Li; Sivia Lapidus; Jenna King; Sierra Gerber; Bethany Welc; Lindsey Bergstrom; Emily Fox; Matthew Hollander; Gabriele Simonini; Alhanouf Alsaleem; Karmela Kim Chan; Ron Laxer; Karen Onel; Seza Ozen; Fatma Dedeoglu; Polly Ferguson; Philip Helliwell; Beverley Shea; Christian Hedrich; Jonathan Akikusa; Philip Mease; Yongdong Zhao

doi:10.1016/j.semarthrit.2025.152862

Identifying domains for CNO and SAPHO: A scoping review to create domains from existing outcomes by the OMERACT CNO and SAPHO working group

Melissa Oliver
, Farzana Nuruzzaman
, Aleksander Lenert
, Arundathi Jayatilleke
, Alexander Theos
, Natalia Palmou-Fontana
, Alissa Skinner
, Marinka Twilt
, Eveline Y. Wu
, Cassyanne Aguiar
, Samir Shah
, Micol Romano
, Suzanne Li
, Sivia Lapidus
, Jenna King
, Sierra Gerber
, Bethany Welc
, Lindsey Bergstrom
, Emily Fox
, Matthew Hollander

Gabriele Simonini, Alhanouf Alsaleem, Karmela Kim Chan, Ron Laxer, Karen Onel, Seza Ozen, Fatma Dedeoglu, Polly Ferguson, Philip Helliwell, Beverley Shea, Christian Hedrich, Jonathan Akikusa, Philip Mease, Yongdong Zhao

Pediatrics

Indiana University Bloomington
University of Iowa
Temple University
Georgetown University
Hospital Universitario Marques de Valdecilla
OMERACT Patient/parent research partner
University of Calgary
University of North Carolina at Chapel Hill
Children's Hospital of The King's Daughters Health System
Division of Northeast Orthopaedic Alliance
Western University
Hackensack Meridian Health
Children's Mercy Hospitals and Clinics
University of Vermont Medical Center
Azienda Ospedaliero Universitaria Meyer
King Faisal Specialist Hospital and Research Centre
Cornell University
University of Toronto
Hospital for Special Surgery - New York
Hacettepe University
Boston Children's Hospital
University of Leeds
University of Ottawa
Alder Hey Children's NHS Foundation Trust
University of Liverpool
Royal Children's Hospital Melbourne
University of Washington

Research output: Contribution to journal › Article › peer-review

1 Scopus citations

Abstract

Introduction: Chronic nonbacterial osteomyelitis (CNO) and synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome are autoinflammatory bone diseases of unknown etiology that present with bone pain and varying degrees of extraosseous manifestations such as skin, intestinal and joint involvement. Currently, there are no validated outcome measurement sets that represent the input from all collaborating groups. Methods: The OMERACT CNO & SAPHO working group performed a scoping review to identify domains previously used in CNO and SAPHO clinical studies. The list of potential domains was narrowed through a process of binning and winnowing. Results: A scoping review included 260 observational studies published from 1978 -2020 and 220 domains were initially identified. Domains were reduced to 25 through a binning and winnowing process. Domains cover each of the OMERACT core with most domains mapped to life impact and pathophysiological manifestations. Conclusion: We identified 25 potential domains covering health concepts of function, disease manifestations, pain, and impact on mental health and societal participation to be included in the final core domain set. The next step will be to reach a consensus on the final CNO & SAPHO core domain set and begin instrument selection.

Original language	English
Article number	152862
Journal	Seminars in Arthritis and Rheumatism
Volume	75
DOIs	https://doi.org/10.1016/j.semarthrit.2025.152862
State	Published - Dec 2025

Keywords

Autoinflammatory disease
Chronic nonbacterial osteomyelitis
Core domain set
OMERACT
Scoping review

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10.1016/j.semarthrit.2025.152862

Cite this

Oliver, M., Nuruzzaman, F., Lenert, A., Jayatilleke, A., Theos, A., Palmou-Fontana, N., Skinner, A., Twilt, M., Wu, E. Y., Aguiar, C., Shah, S., Romano, M., Li, S., Lapidus, S., King, J., Gerber, S., Welc, B., Bergstrom, L., Fox, E., ... Zhao, Y. (2025). Identifying domains for CNO and SAPHO: A scoping review to create domains from existing outcomes by the OMERACT CNO and SAPHO working group. Seminars in Arthritis and Rheumatism, 75, Article 152862. https://doi.org/10.1016/j.semarthrit.2025.152862

@article{7a4e552e500240c6a6869bcc89c0cbe0,

title = "Identifying domains for CNO and SAPHO: A scoping review to create domains from existing outcomes by the OMERACT CNO and SAPHO working group",

abstract = "Introduction: Chronic nonbacterial osteomyelitis (CNO) and synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome are autoinflammatory bone diseases of unknown etiology that present with bone pain and varying degrees of extraosseous manifestations such as skin, intestinal and joint involvement. Currently, there are no validated outcome measurement sets that represent the input from all collaborating groups. Methods: The OMERACT CNO \& SAPHO working group performed a scoping review to identify domains previously used in CNO and SAPHO clinical studies. The list of potential domains was narrowed through a process of binning and winnowing. Results: A scoping review included 260 observational studies published from 1978 -2020 and 220 domains were initially identified. Domains were reduced to 25 through a binning and winnowing process. Domains cover each of the OMERACT core with most domains mapped to life impact and pathophysiological manifestations. Conclusion: We identified 25 potential domains covering health concepts of function, disease manifestations, pain, and impact on mental health and societal participation to be included in the final core domain set. The next step will be to reach a consensus on the final CNO \& SAPHO core domain set and begin instrument selection.",

keywords = "Autoinflammatory disease, Chronic nonbacterial osteomyelitis, Core domain set, OMERACT, Scoping review",

author = "Melissa Oliver and Farzana Nuruzzaman and Aleksander Lenert and Arundathi Jayatilleke and Alexander Theos and Natalia Palmou-Fontana and Alissa Skinner and Marinka Twilt and Wu, \{Eveline Y.\} and Cassyanne Aguiar and Samir Shah and Micol Romano and Suzanne Li and Sivia Lapidus and Jenna King and Sierra Gerber and Bethany Welc and Lindsey Bergstrom and Emily Fox and Matthew Hollander and Gabriele Simonini and Alhanouf Alsaleem and Chan, \{Karmela Kim\} and Ron Laxer and Karen Onel and Seza Ozen and Fatma Dedeoglu and Polly Ferguson and Philip Helliwell and Beverley Shea and Christian Hedrich and Jonathan Akikusa and Philip Mease and Yongdong Zhao",

note = "Publisher Copyright: {\textcopyright} 2025 Elsevier Inc. All rights are reserved, including those for text and data mining, AI training, and similar technologies.",

year = "2025",

month = dec,

doi = "10.1016/j.semarthrit.2025.152862",

language = "English",

volume = "75",

journal = "Seminars in Arthritis and Rheumatism",

issn = "0049-0172",

}

Oliver, M, Nuruzzaman, F, Lenert, A, Jayatilleke, A, Theos, A, Palmou-Fontana, N, Skinner, A, Twilt, M, Wu, EY, Aguiar, C, Shah, S, Romano, M, Li, S, Lapidus, S, King, J, Gerber, S, Welc, B, Bergstrom, L, Fox, E, Hollander, M, Simonini, G, Alsaleem, A, Chan, KK, Laxer, R, Onel, K, Ozen, S, Dedeoglu, F, Ferguson, P, Helliwell, P, Shea, B, Hedrich, C, Akikusa, J, Mease, P & Zhao, Y 2025, 'Identifying domains for CNO and SAPHO: A scoping review to create domains from existing outcomes by the OMERACT CNO and SAPHO working group', Seminars in Arthritis and Rheumatism, vol. 75, 152862. https://doi.org/10.1016/j.semarthrit.2025.152862

TY - JOUR

T1 - Identifying domains for CNO and SAPHO

T2 - A scoping review to create domains from existing outcomes by the OMERACT CNO and SAPHO working group

AU - Oliver, Melissa

AU - Nuruzzaman, Farzana

AU - Lenert, Aleksander

AU - Jayatilleke, Arundathi

AU - Theos, Alexander

AU - Palmou-Fontana, Natalia

AU - Skinner, Alissa

AU - Twilt, Marinka

AU - Wu, Eveline Y.

AU - Aguiar, Cassyanne

AU - Shah, Samir

AU - Romano, Micol

AU - Li, Suzanne

AU - Lapidus, Sivia

AU - King, Jenna

AU - Gerber, Sierra

AU - Welc, Bethany

AU - Bergstrom, Lindsey

AU - Fox, Emily

AU - Hollander, Matthew

AU - Simonini, Gabriele

AU - Alsaleem, Alhanouf

AU - Chan, Karmela Kim

AU - Laxer, Ron

AU - Onel, Karen

AU - Ozen, Seza

AU - Dedeoglu, Fatma

AU - Ferguson, Polly

AU - Helliwell, Philip

AU - Shea, Beverley

AU - Hedrich, Christian

AU - Akikusa, Jonathan

AU - Mease, Philip

AU - Zhao, Yongdong

PY - 2025/12

Y1 - 2025/12

N2 - Introduction: Chronic nonbacterial osteomyelitis (CNO) and synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome are autoinflammatory bone diseases of unknown etiology that present with bone pain and varying degrees of extraosseous manifestations such as skin, intestinal and joint involvement. Currently, there are no validated outcome measurement sets that represent the input from all collaborating groups. Methods: The OMERACT CNO & SAPHO working group performed a scoping review to identify domains previously used in CNO and SAPHO clinical studies. The list of potential domains was narrowed through a process of binning and winnowing. Results: A scoping review included 260 observational studies published from 1978 -2020 and 220 domains were initially identified. Domains were reduced to 25 through a binning and winnowing process. Domains cover each of the OMERACT core with most domains mapped to life impact and pathophysiological manifestations. Conclusion: We identified 25 potential domains covering health concepts of function, disease manifestations, pain, and impact on mental health and societal participation to be included in the final core domain set. The next step will be to reach a consensus on the final CNO & SAPHO core domain set and begin instrument selection.

AB - Introduction: Chronic nonbacterial osteomyelitis (CNO) and synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome are autoinflammatory bone diseases of unknown etiology that present with bone pain and varying degrees of extraosseous manifestations such as skin, intestinal and joint involvement. Currently, there are no validated outcome measurement sets that represent the input from all collaborating groups. Methods: The OMERACT CNO & SAPHO working group performed a scoping review to identify domains previously used in CNO and SAPHO clinical studies. The list of potential domains was narrowed through a process of binning and winnowing. Results: A scoping review included 260 observational studies published from 1978 -2020 and 220 domains were initially identified. Domains were reduced to 25 through a binning and winnowing process. Domains cover each of the OMERACT core with most domains mapped to life impact and pathophysiological manifestations. Conclusion: We identified 25 potential domains covering health concepts of function, disease manifestations, pain, and impact on mental health and societal participation to be included in the final core domain set. The next step will be to reach a consensus on the final CNO & SAPHO core domain set and begin instrument selection.

KW - Autoinflammatory disease

KW - Chronic nonbacterial osteomyelitis

KW - Core domain set

KW - OMERACT

KW - Scoping review

UR - https://www.scopus.com/pages/publications/105024856050

U2 - 10.1016/j.semarthrit.2025.152862

DO - 10.1016/j.semarthrit.2025.152862

M3 - Article

C2 - 41270570

AN - SCOPUS:105024856050

SN - 0049-0172

VL - 75

JO - Seminars in Arthritis and Rheumatism

JF - Seminars in Arthritis and Rheumatism

M1 - 152862

ER -

Identifying domains for CNO and SAPHO: A scoping review to create domains from existing outcomes by the OMERACT CNO and SAPHO working group

Abstract

Keywords

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