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Published on: December 31, 2017
Chronic nonbacterial osteomyelitis in children: a retrospective multicenter study
Daniela Kaiser1, Isabel Bolt2, Michael Hofer3
1Department of Pediatric Rheumatology, Children's Hospital, Kantonsspital Luzern, CH-6000, Luzern 16, Switzerland. daniela.kaiser@luks.ch.
Insights
Pediatric nonbacterial inflammatory bone diseases like chronic nonbacterial osteomyelitis (CNO) and SAPHO syndrome often present with persistent symptoms and may require advanced treatments beyond NSAIDs. Early diagnosis is crucial, as some cases show significant delays.
Area of Science:
- Pediatric Rheumatology
- Pediatric Orthopedics
- Pediatric Inflammation
Background:
- Nonbacterial inflammatory bone diseases in children encompass chronic nonbacterial osteomyelitis (CNO) and synovitis acne pustulosis hyperostosis osteitis (SAPHO) syndrome.
- Understanding their clinical presentation, treatment, and outcomes is vital for effective management.
Purpose of the Study:
- To determine the clinical presentation, current treatment strategies, and outcomes of pediatric patients diagnosed with nonbacterial inflammatory bone diseases.
- To analyze the course and response to therapy in children with CNO and SAPHO syndrome.
Main Methods:
- A retrospective multicenter study was conducted using data from the Swiss Pediatric Rheumatology Working Group registry.
- Patient charts were reviewed for disease presentation, treatment details, disease course, and patient outcomes.
Main Results:
- The study included 41 children with multifocal CNO, unifocal CNO, or SAPHO syndrome, diagnosed between 1995 and 2010.
- Most patients (27/41) had a chronic persistent disease course; 57% achieved disease control with NSAIDs, while others required advanced therapies like corticosteroids, methotrexate, or TNF α inhibitors.
- A significant diagnostic delay was noted in 22% of cases presenting with unifocal lesions.
Conclusions:
- Nonbacterial inflammatory bone diseases in children frequently present as chronic persistent conditions rather than recurrent forms.
- Pelvic osteomyelitis is significantly associated with features of juvenile spondylarthritis.
- Effective management may necessitate a stepwise approach, escalating therapy based on treatment response.
Background:
To determine the clinical presentation, current treatment and outcome of children with nonbacterial inflammatory bone disease.
Methods:
Retrospective multicenter study of patients entered into the Swiss Pediatric Rheumatology Working Group registry with a diagnosis of chronic nonbacterial osteomyelitis (CNO) and synovitis acne pustulosis hyperostosis osteitis (SAPHO) syndrome. The charts were reviewed for informations about disease presentation, treatment, course and outcome.
Results:
Forty-one children (31 girls and 10 boys) from 6 pediatric hospitals in Switzerland diagnosed between 1995 and 2010 were included in the study. The diagnosis was multifocal CNO (n = 33), unifocal CNO (n = 4) and SAPHO syndrome (n = 4). Mean age at onset of CNO was 9.5 years (range 1.4-15.6) and mean follow-up time was 52 months (range 6-156 months). Most patients (n = 27) had a chronic persistent disease course (>6 months), 8 patients had a course with one or more relapses and 6 patients had only one episode of CNO. Forty nine percent had received at least one course of antibiotics. In 57% treatment with nonsteroidal anti-inflammatory drugs (NSAID) was sufficient to control the disease. Twelve out of 16 children with NSAID failure subsequently received corticosteroids, methotrexate, TNF α inhibitors, bisphosphonates or a combination of these drugs.
Conclusions:
In a multicenter cohort of 41 children 22% started with unifocal lesion with a significant diagnostic delay. A higher proportion presented with chronic persistent disease than with a recurrent form. An osteomyelitis in the pelvic region is significantly associated with other features of juvenile spondylarthritis.
