Chronic Nonbacterial Osteomyelitis of the Jaw in a 3-Year-Old Girl
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Published on: May 23, 2020
Shigeru Makino1, Hideo Oshige2, Jun Shinozuka1
1Department of Pediatrics, Uji-Tokushukai Medical Center, Kyoto 611-0041, Japan.
Insights
Diagnosing chronic nonbacterial osteomyelitis (CNO) in young children, especially when isolated to the jaw, is difficult. This case highlights successful treatment of pediatric CNO with alendronate and NSAIDs.
Area of Science:
- Pediatric rheumatology
- Osteology
- Medical imaging
Background:
- Chronic nonbacterial osteomyelitis (CNO) is a rare autoinflammatory bone disease.
- Diagnosis is challenging, particularly in young children and when isolated to the jaw.
- Distinguishing CNO from bacterial osteomyelitis (BOM) is critical for appropriate treatment.
Observation:
- A 3-year-old female presented with isolated CNO of the jaw.
- Symptoms included jaw pain, trismus, and facial swelling without fever.
- CT imaging revealed mandibular hyperostosis with osteolytic and sclerotic changes.
Findings:
- Initial suspicion of BOM led to antibiotic treatment, which was ineffective.
- CNO diagnosis was confirmed, and treatment with NSAIDs (flurbiprofen) showed limited response.
- Combination therapy with oral alendronate and flurbiprofen achieved successful treatment outcomes.
Implications:
- Physicians must consider CNO in young children presenting with jaw osteomyelitis, even without systemic symptoms.
- Early and accurate diagnosis is essential to avoid prolonged or incorrect treatments.
- Combined alendronate and NSAID therapy may be effective for refractory pediatric CNO of the jaw.
Abstract:
Differential diagnosis of bacterial osteomyelitis (BOM) and chronic nonbacterial osteomyelitis (CNO) is challenging. Pediatric CNO can be diagnosed at around 10 years of age and when CNO cases involve only the jaw, it is difficult to make a diagnosis in a young child. A 3-year-old female developed CNO at the jaw alone. She presented with no fever, right jaw pain, mild trismus, and a preauricular facial swelling around the right mandible. Computed tomography (CT) revealed a hyperostotic right mandible, with osteolytic and sclerotic changes associated with periosteal reaction. At first, we suspected BOM and antibiotics were administered. Subsequently, CNO was diagnosed, and the patient received flurbiprofen (a nonsteroidal anti-inflammatory drug (NSAIDs)). Lack of a sufficient response led to successful treatment with a combination of oral alendronate and flurbiprofen. Physicians should be aware of CNO, a rare autoinflammatory noninfectious bone disease of unknown etiology, even in young children, although the disease mostly affects older children and adolescents.
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