Chronic recurrent multifocal osteomyelitis with interstitial myositis

Abstract

Chronic recurrent multifocal osteomyelitis (CRMO) is an inflammatory, non-infectious disorder of skeletal system mainly seen in children. We report a case of CRMO presenting with fever and leg pain. The patient was an 11-year-old boy complaining of a fever, swelling and pain on his right foot, and pain on both legs. Although serum levels of CK and aldolase were not increased, MRI imaging suggested polymyositis. Muscle biopsy showed interstitial infiltration of inflammatory cells without any evidences of dermatomyositis or polymyositis. One month later, he complained of a swelling, pain and redness of his left clavicle as recurrently experienced during the recent 6 months, and MRI investigation indicated the diagnosis of osteomyelitis. Bone biopsy was performed and showed chronic inflammatory changes with negative bacterial culture. Multiple bone lesions and muscle uptake of FDG in his legs were revealed by whole body FDG-PET/CT, and he was diagnosed as having CRMO with interstitial myositis. The combinatorial administration of non-steroidal anti-inflammatory drugs and bisphosphonate successfully improved his clinical symptoms and laboratory abnormalities. To our knowledge, there is no report of a patient of CRMO associated with interstitial myositis.

Adult-onset Chronic Recurrent Multifocal Osteomyelitis with High Intensity of Muscles Detected by Magnetic Resonance Imaging, Successfully Controlled with Tocilizumab

“…These reports suggest an association of either myositis or soft-tissue inflammation with osteomyelitis because of the close anatomic relationships of the foci of myositis and osteomyelitis in these cases. A patient with CRMO associated with interstitial myositis has also been reported ( 16 ). In an 11-year-old boy, CRMO associated with myositis of the quadriceps femoris and gastrocnemius muscles, detected on MRI and fluorodeoxyglucose (FDG)-positron emission tomography/CT, was diagnosed, but the osteomyelitis did not involve his femurs and tibias.…”

Chronic recurrent multifocal osteomyelitis with myositis: A case report and review of the literature

“…These findings suggest an association of myositis with osteomyelitis because of the close anatomic relationships of the foci of myositis and osteomyelitis. In contrast, myositis was present not along osteomyelitis in the other three patients . In an 11‐year‐old boy, myositis associated with CRMO was detected on MRI in the quadriceps femoris and gastrocnemius muscles .…”

“…In contrast, myositis was present not along osteomyelitis in the other three patients . In an 11‐year‐old boy, myositis associated with CRMO was detected on MRI in the quadriceps femoris and gastrocnemius muscles . However, osteomyelitis did not involve his femurs and tibias.…”

“…Chronic, recurrent, multifocal osteomyelitis (CRMO) is a rare, autoinflammatory bone disease, characterized by chronic, non‐bacterial osteomyelitis, multifocal bone lesions, and multiple recurrence. Patients with CRMO typically present with bone pain; myositis is rare as a clinical symptom of CRMO . Here, we report a case of CRMO with myositis of the internal obturator muscle.…”

“…Myositis as a clinical symptom of CRMO is very rare, and only five cases have been previously reported (Table ). The muscles in the extremities were affected in most cases.…”