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Published on: February 9, 2011
Pediatric chronic nonbacterial osteomyelitis
Arturo Borzutzky1, Sara Stern, Andreas Reiff
1Program in Rheumatology, Division of Immunology, Children’s Hospital Boston, Boston, Massachusetts 02115, USA.
Insights
Chronic nonbacterial osteomyelitis (CNO) in children often coexists with autoimmune diseases. Immunosuppressive therapies, including disease-modifying antirheumatic drugs and biologics, showed higher efficacy than NSAIDs for CNO treatment.
Area of Science:
- Pediatric rheumatology
- Pediatric orthopedics
- Immunology
Background:
- Chronic nonbacterial osteomyelitis (CNO) is a rare pediatric condition with limited data on its natural history and optimal management.
- Understanding CNO's characteristics is crucial for improving patient outcomes.
Purpose of the Study:
- To retrospectively review a large cohort of pediatric patients diagnosed with CNO.
- To assess the clinical features, natural history, and treatment responses in children with CNO.
Main Methods:
- Retrospective chart review of 70 children diagnosed with CNO across three US tertiary care centers (1985-2009).
- Data extraction included clinical, laboratory, histopathologic, and radiologic findings.
- Evaluation of treatment responses to various agents: NSAIDs, sulfasalazine, methotrexate, TNFα inhibitors, and corticosteroids.
Main Results:
- Half of the pediatric CNO patients had comorbid autoimmune diseases, associated with more bone lesions and higher inflammatory markers.
- Treatment efficacy varied significantly by agent; corticosteroids (95%), TNFα inhibitors (91%), and methotrexate (91%) showed the highest response rates.
- Disease-modifying antirheumatic drugs (DMARDs) and biologics were more effective than NSAIDs.
Conclusions:
- Autoimmunity is a significant comorbidity in pediatric CNO, linked to multifocal disease and the need for immunosuppressive therapy.
- DMARDs and biologics represent more effective treatment options for pediatric CNO compared to NSAIDs.
Background And Objectives:
Little information is available concerning the natural history and optimal treatment of chronic nonbacterial osteomyelitis (CNO). We conducted a retrospective review to assess the clinical characteristics and treatment responses of a large cohort of pediatric CNO patients.
Methods:
Children diagnosed with CNO at 3 tertiary care centers in the United States between 1985 and 2009 were identified. Their charts were reviewed, and clinical, laboratory, histopathologic, and radiologic data were extracted.
Results:
Seventy children with CNO (67% female patients) were identified. Median age at onset was 9.6 years (range 3-17), and median follow-up was 1.8 years (range 0-13). Half of the patients had comorbid autoimmune diseases, and 49% had a family history of autoimmunity. Patients with comorbid autoimmune diseases had more bone lesions (P < .001), higher erythrocyte sedimentation rate (P < .05), and higher use of second line therapy (P = .02). Treatment response to nonsteroidal antiinflammatory drugs (NSAIDs), sulfasalazine, methotrexate, tumor necrosis factor α inhibitors, and corticosteroids was evaluated. The only significant predictor of a positive treatment response was the agent used (P < .0001). Estimated probability of response was 57% for NSAIDs, 66% for sulfasalazine, 91% for methotrexate, 91% for tumor necrosis factor α inhibitors, and 95% for corticosteroids.
Conclusions:
In a US cohort of 70 children with CNO, coexisting autoimmunity was a risk factor for multifocal involvement and treatment with immunosuppressive agents. Disease-modifying antirheumatic drugs and biologics were more likely to lead to clinical improvement than NSAIDs.
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