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Response to Early-onset Pamidronate Treatment in Chronic Nonbacterial Osteomyelitis: A Retrospective Single-center
Caroline Marie Andreasen1,2, Anne Grethe Jurik3,4, Mia B Glerup3,4
1From the Department of Rheumatology, the Department of Radiology, and the Department of Pediatrics and Adolescent Medicine, Aarhus University Hospital; the Department of Clinical Medicine, Aarhus University, Aarhus, Denmark. carand@rm.dk.
Insights
Early-onset pamidronate treatment may improve chronic nonbacterial osteomyelitis (CNO) in children, showing reduced bone lesions. However, some children with extended CNO experienced relapses despite treatment.
Area of Science:
- Pediatric rheumatology
- Pediatric orthopedics
- Inflammatory bone disease
Background:
- Chronic nonbacterial osteomyelitis (CNO) is a sterile inflammatory bone disorder with unpredictable progression.
- Assessing disease activity and treatment response is crucial for managing CNO in children.
Purpose of the Study:
- To evaluate clinical and radiological disease activity in pediatric CNO patients.
- To assess the efficacy of early-onset pamidronate treatment in children with extended CNO.
Main Methods:
- Retrospective single-center study of 51 children meeting Bristol Criteria for CNO.
- Whole-body MRI (WB-MRI) used for radiological assessment at diagnosis and annually.
- Clinical disease activity assessed annually; extended CNO defined by multifocal/spinal involvement unresponsive to NSAIDs.
Main Results:
- Extended CNO patients (n=32) received a 2-year pamidronate regimen.
- Significant decline in radiologically active lesions, including spinal lesions, within the first year of pamidronate (p=0.01).
- After 1 year, 38% of extended CNO patients were clinically inactive, but 67% relapsed; limited CNO patients showed better outcomes.
Conclusions:
- Pamidronate may improve clinical and radiological disease activity in extended CNO, particularly after one year.
- Some children with extended CNO continued to have active disease even after two years of pamidronate treatment.
- Further research is needed to optimize treatment strategies for chronic nonbacterial osteomyelitis in children.
Objectives:
Chronic nonbacterial osteomyelitis (CNO) is a sterile inflammatory bone disorder with an unpredictable disease course. The objective was to assess clinical and radiological disease activity in children with CNO including response to early-onset pamidronate treatment.
Methods:
A single-center retrospective study was conducted of children fulfilling the Bristol Criteria for CNO. At the time of diagnosis, whole-body magnetic resonance imaging (WB-MRI) or local MRI was performed to assess radiological disease activity. Children with multifocal or spinal bone inflammation and clinical disease activity not responding to nonsteroidal antiinflammatory drugs were categorized as having extended CNO. Clinical disease activity was assessed annually.
Results:
Fifty-one children were included. Median followup time was 4 years (interquartile range 3-7). Children categorized with extended CNO (n = 32) were treated in an early-onset 2-year pamidronate regimen. In extended CNO, WB-MRI was performed at time of diagnosis, and at years 1 and 2 in 88%, 84%, and 91% of cases, respectively. During the first year, the total number of radiologically active lesions and number of spinal lesions per patient declined (p = 0.01). Clinically inactive disease was recorded in 12/32 children (38%). However, 8/12 children (67%) experienced clinical relapse. In limited CNO (n = 19), 10/19 children (53%) presented with clinically inactive disease after 1 year and did not experience clinical relapse.
Conclusion:
Pamidronate might contribute to improvement in clinical and radiological disease activity in extended CNO, especially after 1 year of treatment. However, children with continuously active disease after 2 years of pamidronate treatment were seen.
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