Abstract
IntroductionChronic nonbacterial osteomyelitis (CNO) is an inflammatory disorder of unknown etiology. In children and adolescents CNO predominantly affects the metaphyses of the long bones, but lesions can occur at any site of the skeleton. Prospectively followed cohorts using a standardized protocol in diagnosis and treatment have rarely been reported.MethodsThirty-seven children diagnosed with CNO were treated with naproxen continuously for the first 6 months. If assessment at that time revealed progressive disease or no further improvement, sulfasalazine and short-term corticosteroids were added. The aims of our short-term follow-up study were to describe treatment response in detail and to identify potential risk factors for an unfavorable outcome.ResultsNaproxen treatment was highly effective in general, inducing a symptom-free status in 43% of our patients after 6 months. However, four nonsteroidal anti-inflammatory drug (NSAID) partial-responders were additionally treated with sulfasalazine and short-term corticosteroids. The total number of clinical detectable lesions was significantly reduced. Mean disease activity estimated by the patient/physician and the physical aspect of health-related quality of life including functional ability (global assessment/childhood health assessment questionnaire and childhood health assessment questionnaire) and pain improved significantly. Forty-one percent of our patients showed radiological relapses, but 67% of them were clinically silent.ConclusionsMost children show a favorable clinical course in the first year of anti-inflammatory treatment with NSAIDs. Relapses and new radiological lesions can occur at any time and at any site in the skeleton but may not be clinically symptomatic. Whole-body magnetic resonance imaging proved to be very sensitive for initial and follow-up diagnostics.
Highlights
Chronic nonbacterial osteomyelitis (CNO) is an inflammatory disorder of unknown etiology
The CNO core set of outcome variables is composed of the following five measures: erythrocyte sedimentation rate (ESR), number of radiological lesions, severity of disease estimated by the physician, severity of disease estimated by the patient or parent, and the childhood health assessment questionnaire (CHAQ)
184 clinical foci were detected over 1 year - resulting in a mean of 1.0 (2.1 at time of diagnosis, 1.1 after 1 month, 0.8 after 3 months, 0.6 after 6 months, and 0.5 after 1 year of treatment) per patient, showing a significantly lower number in follow-ups (analysis of variance (ANOVA), P < 0.05)
Summary
Chronic nonbacterial osteomyelitis (CNO) is an inflammatory disorder of unknown etiology. Followed cohorts using a standardized protocol in diagnosis and treatment have rarely been reported. Chronic nonbacterial osteomyelitis (CNO) is an inflammatory, non-infectious disorder of the musculoskeletal system of unknown etiology. Both single and multiple lesions and recurrence have been described [1,2,3]. There have been attempts to classify patients into defined groups (unifocal nonrecurrent, unifocal recurrent, multifocal nonrecurrent, multifocal recurrent) in order to set up diagnostic criteria and to find prognostic indicators [12,13]. Until now no standardized diagnostic criteria and therapeutic guidelines or standards exist. There are no generally accepted treatment protocols available
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