Gathering expert consensus to inform a proposed trial in chronic nonbacterial osteomyelitis (CNO)

Research output: Contribution to journalResearch articleContributedpeer-review

Contributors

  • C. M. Hedrich - , University of Liverpool (UOL), Royal Liverpool Children's NHS Trust: (Author)
  • M. W. Beresford - , University of Liverpool (UOL), Royal Liverpool Children's NHS Trust: (Author)
  • F. Dedeoglu - , Harvard University (Author)
  • G. Hahn - , Institute and Polyclinic of Diagnostic and Interventional Radiology, Technische Universität Dresden (Author)
  • S. R. Hofmann - , Department of Paediatrics (Author)
  • A. F. Jansson - , Ludwig Maximilian University of Munich (Author)
  • R. M. Laxer - , University of Toronto (Author)
  • P. Miettunen - , University of Calgary (Author)
  • H. Morbach - , University Hospital of Würzburg (Author)
  • C. E. Pain - , Royal Liverpool Children's NHS Trust: (Author)
  • A. V. Ramanan - , University of Bristol (Author)
  • E. Roberts - , Royal Liverpool Children's NHS Trust: (Author)
  • A. Schnabel - , Department of Paediatrics (Author)
  • A. Theos - , Georgetown University (Author)
  • L. Whitty - , University of Liverpool (UOL) (Author)
  • Y. Zhao - , University of Washington (Author)
  • P. J. Ferguson - , University of Iowa (Author)
  • H. J. Girschick - , Vivantes Hospitals Berlin (Author)

Abstract

Chronic nonbacterial osteomyelitis (CNO) is an autoinflammatory bone disease that primarily affects children and adolescents. CNO is associated with pain, bone swelling, deformity, and fractures. Its pathophysiology is characterized by increased inflammasome assembly and imbalanced expression of cytokines. Treatment is currently based on personal experience, case series and resulting expert recommendations. Randomized controlled trials (RCTs) have not been initiated because of the rarity of CNO, expired patent protection of some medications, and the absence of agreed outcome measures. An international group of fourteen CNO experts and two patient/parent representatives was assembled to generate consensus to inform and conduct future RCTs. The exercise delivered consensus inclusion and exclusion criteria, patent protected (excludes TNF inhibitors) treatments of immediate interest (biological DMARDs targeting IL-1 and IL-17), primary (improvement of pain; physician global assessment) and secondary endpoints (improved MRI; improved PedCNO score which includes physician and patient global scores) for future RCTs in CNO.

Details

Original languageEnglish
Article number109344
Number of pages7
JournalClinical immunology
Volume251 (2023)
Publication statusPublished - 23 Apr 2023
Peer-reviewedYes

External IDs

PubMed 37098355

Keywords

ASJC Scopus subject areas

Keywords

  • CNO, CRMO, Consensus, Osteomyelitis, Outcome, Study, Treatment, Trial, Cytokines, Pain/complications, Humans, Osteomyelitis/drug therapy, Adolescent, Antirheumatic Agents/therapeutic use, Child, Chronic Disease

Library keywords