Nonbacterial osteomyelitis of the vertebral bodies and frontal bone: A description of a rare clinical case and a review of the literature

Abstract

BACKGROUND: Nonbacterial osteomyelitis is a chronic autoinflammatory skeletal disorder of unknown origin characterized by sterile bone lesions and presenting more frequently in children. Spinal manifestations are often common in nonbacterial osteomyelitis; however, cases with skull involvement, except for the mandible, are generally rare.

CLINICAL CASE: Herein, we report the case of an 11-year-old girl presenting with multifocal thoracic vertebral and frontal bone lesions, which led to destructive sinusitis. Nonbacterial osteomyelitis was diagnosed after a bone biopsy, which showed no evidence of granuloma, malignancy, or histiocytes. The histopathological findings were nonspecific inflammatory changes. Ibandronic acid was used to treat nonbacterial osteomyelitis. Clinical signs begin to improve after the first infusion. After the fourth infusion of ibandronic acid, the inflammation was reduced and frontal bone structure and thoracic vertebrae were restored.

DISCUSSION: Bisphosphonate therapy can be used in nonbacterial osteomyelitis when response to nonsteroidal anti-inflammatory drugs is not optimal. The efficacy of bisphosphonate therapy reaches 75%. However, bisphosphonate therapy in nonbacterial osteomyelitis has not been developed. The paper contained literature about rare cases with skull involvement and problems in bisphosphonate therapy in pediatric nonbacterial osteomyelitis.

CONCLUSIONS: Nonbacterial osteomyelitis is a treatable condition, whose care depends on a referral to a rheumatologist.

Keywords

nonbacterial osteomyelitis / spondylitis / frontitis / ibandronic acid

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