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Assessing Disease Activity in Pediatric Chronic Nonbacterial Osteomyelitis: A Proposal for Composite Scoring,
Christiane Reiser1, Jens Klotsche2, Anja Schnabel3
1Division of Pediatric Rheumatology, Department of Pediatrics and Autoinflammation Reference Centre Tübingen, University Hospital Tübingen, Germany, and Department of Pediatrics, Landeskrankenhaus Bregenz, Bregenz, Austria.
Composite scores incorporating MRI lesions and physician assessments show promise for evaluating chronic nonbacterial osteomyelitis (CNO) disease activity. These metrics offer a more objective approach than subjective measures alone.
Area of Science:
- Rheumatology
- Pediatric Rheumatology
- Autoinflammatory Diseases
Background:
- Chronic nonbacterial osteomyelitis (CNO) lacks validated criteria for assessing disease activity (DA), inactive disease, or remission.
- Current DA assessment in CNO relies heavily on subjective patient, rheumatologist, and radiologist judgments.
- Emerging composite measures aim to provide more objective evaluations of CNO DA.
Purpose of the Study:
- To propose novel composite scores for CNO DA assessment, incorporating physician global assessment (PGDA) and MRI findings.
- To evaluate the performance of the previously published CNO composite DA score (CDAS) and the Pediatric CNO (PedCNO) response score.
- To introduce and suggest a PedCNO 90% improvement (PedCNO90) category for evaluating treatment response.
Main Methods:
- Analysis of newly diagnosed CNO patients (2015-2020) from the National Pediatric Rheumatologic Database.
- Assessment of clinical course and DA measures, including physician global assessment of DA (PGDA) and patient-reported outcomes (PAG/PAP), over up to 4 years of follow-up (YFU).
- Evaluation of existing composite scores (CNO CDAS, PedCNO response score) and proposed new scores using registry data.
Main Results:
- Composite scores integrating MRI lesions and PGDA demonstrated more pronounced changes over time compared to the CNO CDAS.
- Clinical and MRI lesion scores showed significant changes from baseline to 3 YFU (P = 0.003/P = 0.004).
- The PedCNO90 response score increased during follow-up, with approximately half of the remaining patients achieving PedCNO90 by 4 YFU.
Conclusions:
- Composite scores that include MRI lesions and PGDA are promising tools for objectively describing CNO DA.
- The specific composition of DA scores is crucial for the validity and utility of future CNO research.
- These findings support the development of more standardized and objective methods for CNO management.
