Chronic Recurrent Multifocal Osteomyelitis of the Spine and Mandible: Case Report and Review of the Literature

Abstract

ABSTRACT. At initial presentation, chronic recurrent multifocal osteomyelitis may mimic acute hematogenous osteomyelitis; however, cultures of affected bone are sterile. Nuclear scintigraphy identifies additional foci of involvement that present concurrently or sequentially. Unlike acute bacterial osteomyelitis, chronic recurrent multifocal osteomyelitis seems unaffected by antibiotic therapy and typically responds to treatment with antiinflammatory drugs. Surgical decortication has been reported for refractory cases. The case presented here illustrates the rare involvement of the mandible after initial presentation in the spine of a 4-year-old girl and the refractory nature of the disease over 6 years despite treatment with various medical and surgical therapies. Pediatrics 2004;113:e380 -e384. URL: http://www.pediatrics. org/cgi/content/full/113/4/e380; bone diseases, osteitis, hyperostosis.ABBREVIATIONS. CRMO, chronic recurrent multifocal osteomyelitis; MRI, magnetic resonance imaging; ESR, erythrocyte sedimentation rate; CT, computed tomography; AFB, acid-fast bacilli; Ig, immunoglobulin; PCR, polymerase chain reaction; DSO, diffuse sclerosing osteomyelitis; SAPHO, synovitis, acne, palmoplantar pustulosis, hyperostosis, osteitis syndrome. G iedion et al 1 first described acquired, culturenegative, multifocal osteomyelitis in children in 1972 and named the syndrome "subacute and chronic symmetrical osteomyelitis." In the original report, the disorder involved the metaphyses of long bones either simultaneously or successively. Subsequent publications reported involvement of additional bone sites and referred to the disorder as chronic multifocal osteomyelitis 2 and chronic recurrent multifocal osteomyelitis (CRMO). 3 Pediatric cases with mandibular involvement have been reported in the oral surgery, radiology, orthopedics, and dermatology literature.Unlike the previously reported 4 cases of CRMO with mandibular involvement in the English-language literature, 4 -7 this patient developed CRMO initially in the spine and thereafter developed mandibular involvement. She did not have prior dental infection or skin manifestations associated with CRMO. Additional aspects of interest include the progressive nature of the mandibular involvement and the disease refractoriness to different therapies over 6 years. CASE REPORT Year 1In February 1997, a previously healthy, 4-year, 2-month-old white female was seen in the clinic for left-sided lumbar pain and a fever to 104°F. There was no history of trauma, and the examination was normal. She was treated for muscle strain with ibuprofen. Over the next 3 months, the pain persisted and woke her at night, and she refused to run or jump. A bone scan revealed increased uptake at the left side of the S1 vertebra on delayed images. Magnetic resonance imaging (MRI) revealed a well-circumscribed, irregularly shaped lytic lesion of S1 extending into the sacral ala (Fig 1). Her erythrocyte sedimentation rate (ESR) was 27 mm/hour, and she underwent a computed tomography (CT)-guided perc...

SAPHO osteomyelitis and sarcoid dermatitis in a patient with DiGeorge syndrome

“…The presented case also revealed unusual clinical features, including persistent painful swelling of the right mandible and prolonged elevation of inflammatory parameters, even after successful periodontal surgery. Her excellent responses to NSIADS indicate that her condition may be consistent with chronic recurrent multifocal osteomyelitis [4] or SAPHO syndrome [1,24]. However, her clinical features lack skin manifestation (acne, pustulosis, and psoriasis), joint symptoms (arthritis or synovitis), or multifocal osteitis or osteomyelitis.…”

Chronic Recurrent Multifocal Osteomyelitis Causing an Acute Scoliosis

“…MRI typically shows multifocal increases in signal on the STIR sequences. High signals can present concurrently or sequentially; therefore, interval scanning is essential to monitor progression of the disease [9]. Malignancy, pyogenic infections, and atypical presentations of juvenile arthritis need to be ruled out, correlating the history, clinical findings and biopsy results.…”

Osteomyelitis of the mandible in a group of 33 pediatric patients with congenital insensitivity to pain with anhidrosis

“…This finding suggests an infection rather than inflammation, although a possibility of an acute infection complicating a chronic inflammation cannot be ruled out. There are reports on chronic recurrent multifocal osteomyelitis (CRMO) with mandibular involvement of [34,35].…”