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Chronic non-bacterial osteomyelitis masquerading as fibrous dysplasia
Tessa N Campbell1, Bevan Frizzell2, Paul MacMullan1
1Division of Rheumatology, Department of Medicine, University of Calgary, Calgary, Canada.
Insights
Chronic non-bacterial osteomyelitis (CNBO) is rare in adults. This case shows a 30-year-old female successfully treated with pamidronate after NSAID failure, highlighting CNBO diagnostic challenges and adult awareness needs.
Area of Science:
- Rheumatology
- Immunology
- Oral and Maxillofacial Surgery
Background:
- Chronic non-bacterial osteomyelitis (CNBO) is a rare autoinflammatory bone disorder primarily affecting children.
- Diagnosis in adults is challenging due to its rarity and potential misdiagnosis with other conditions.
Observation:
- A 30-year-old female presented with a 6-year history of right jaw pain and swelling.
- Initial diagnoses included osteomyelitis and fibrous dysplasia, necessitating extensive investigations including multiple biopsies and imaging.
- The patient experienced recurrent painful swelling, elevated inflammatory markers, and specific MRI findings consistent with CNBO.
Findings:
- The patient was initially treated with antibiotics and later non-steroidal anti-inflammatory drugs (NSAIDs) with temporary relief.
- She became refractory to NSAIDs, prompting a shift in treatment strategy.
- Successful remission was achieved using pamidronate, a bisphosphonate, with concurrent improvement in imaging.
Implications:
- This case underscores the diagnostic difficulties of adult-onset CNBO and advocates for increased clinical awareness.
- Effective treatment with pamidronate suggests bisphosphonates may be a valuable early intervention for adult CNBO refractory to NSAIDs.
Abstract:
Chronic non-bacterial osteomyelitis is a rare auto-inflammatory bone disease seen predominantly in the paediatric population. We describe a unique case of a 30-year-old female who presented with right-sided jaw pain and intermittent swelling over the course of 6 years. She was initially treated with antibiotics for possible osteomyelitis, then temporarily diagnosed with fibrous dysplasia. She underwent extensive investigations consisting of an infectious workup, numerous imaging modalities, and three separate biopsies of her right jaw. She was ultimately diagnosed with chronic non-bacterial osteomyelitis based upon her history of recurrent episodes of painful swelling, response to non-steroidal anti-inflammatories, previously raised acute phase reactants, and magnetic resonance imaging findings. Unfortunately, she became refractory to non-steroidal anti-inflammatory therapy. Consequently, she was successfully treated with pamidronate, achieving clinical remission with improvement in her imaging findings. This case highlights the difficulty of diagnosis of chronic non-bacterial osteomyelitis and the need for increased awareness of the disease in the adult population. Additionally, the effective treatment with pamidronate supports the use of a bisphosphonate as an early intervention for adult-onset chronic non-bacterial osteomyelitis in patients who have failed non-steroidal anti-inflammatory therapy.
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