Biologic treatment for chronic recurrent multifocal osteomyelitis: report of four cases and review of the literature

Chronic recurrent multifocal osteomyelitis (CRMO) is a rare non-infectious inflammatory disorder with unpredictable clinical course, characterized by acute exacerbations and spontaneous remissions. There are no randomized-controlled trials about treatment options. Non-steroidal anti-inflammatory drugs (NSAID) are the first-line treatment option; glucocorticoids seem to be effective; positive outcomes have been obtained with bisphosphonates. In the last few years successful use of biologic agents like anti-TNF agents has been reported. We report the cases of 3 children suffering from CRMO who were treated with NSAID, steroid, bisphosphonates and eventually received etanercept and 1 case without vertebral involvement treated with etanercept after NSAID and steroid; all cases showed clinical improvement. The mean ages at symptoms onset and diagnosis were 8 and 10 years and 10 months, respectively. Two patients presented with back pain and three had vertebral lesions. Mean interval from diagnosis to the onset of anti-TNF treatment was 14 months. According to our small experience, we suggest considering therapy with etanercept for the treatment of severe cases with persistently active disease despite multiple treatments.