Combination Therapy with Methotrexate and Etanercept for Refractory Chronic Recurrent Multifocal Osteomyelitis: Figure 1.

Abstract

To the Editor: Chronic recurrent multifocal osteomyelitis (CRMO) is a form of chronic nonbacterial osteitis (CNO) characterized by one or more lytic bone lesions with no identifiable cause1. The metaphyses of long bones and the clavicle are most frequently affected, although any bone may be involved. Most cases begin in childhood, and follow an intermittent course. Pain is the most common presenting symptom. Affected individuals are sometimes febrile, and acute-phase reactants may be increased, pointing to the inflammatory expression of this condition. Lesions may resolve at or before the time of puberty, but in some cases skeletal deformity and/or disability may occur2. We describe 2 children with CRMO whose symptoms persisted after trials of nonsteroidal antiinflammatory medication, glucocorticoids, and methotrexate (MTX). Combination therapy with MTX and etanercept resulted in amelioration of pain, accompanied in one patient by radiographic improvement in a lytic bone lesion. A 4-year-old boy developed pain in his right foot and refused to walk for 2 months. Plain radiographs showed normal-appearing vertebra. A bone scan disclosed increased uptake in the right talus and navicular bone. He received several courses of intravenous and oral antimicrobials, without improvement. Fever was absent, C-reactive protein was normal, and erythrocyte sedimentation rate (ESR) was mildly increased to 23 mm/h. A second bone scan performed 2 months later showed increased radionuclide uptake in the L4 and L5 vertebra. Focal magnetic resonance imaging (MRI) revealed vertebral abnormalities consistent with CRMO including increased signal intensity of the vertebral marrow (Figure 1A). Talar biopsy was nonrevealing, showing bone fragments with myxoid-appearing fibrous stroma. Naproxen 10 mg/kg/day was given, without improvement. Prednisone 2 mg/kg/day was added for 3 weeks, … [↵][1]Address correspondence to Dr. Eisenstein; E-mail: emeisenstein{at}gmail.com [1]: #xref-corresp-1-1