SAPHO—a diagnosis to consider in patients with refractory costochondritis

Abstract

Abstract Background SAPHO syndrome (synovitis, acne, pustulosis, hyperostosis, and osteitis) is a rare, heterogeneous, self-limited disease of unknown etiology. It involves progressive bone and joint damage, and skin and bone lesions may occur at different times in the course of the disease. Skin lesions are characterized by neutrophil dermatosis. Its management is empirical and mainly symptomatic, and nonsteroidal anti-inflammatory drugs are the first-line treatment. Case presentation Forty-seven-year-old female presented with a 7-year history of costochondral pain. It had progressive onset, chronical course, with no other associated symptoms, and no other joint involvement. She was treated with intermittent NSAID (nonsteroidal anti-inflammatory drugs), which provided only partial pain relief; there was bilateral tender swelling of the sternoclavicular region, the skin over the sternoclavicular area was slightly erythematous, but there were no other skin lesions, and based on imaging findings, a diagnosis of SAPHO syndrome was established. The patient received an infusion of zoledronic acid with subsequent complete resolution of her chest wall symptoms, and completely improved after 3 days. Conclusions This case is considered atypical presentation of SAPHO syndrome, without skin changes, long-term persistence of refractory symptoms, and the diagnosis was established by imaging, with complete resolution after zoledronic acid infusion. SAPHO is a differential diagnosis in patients with chronic costochondritis. Therapeutic failure to NSAID is a key to its diagnostic suspicion. Also, early diagnostic suspicion is associated with better outcomes.

Keywords

• Acquired hyperostosis syndrome
• Osteitis
• Hyperostosis
• Chest pain
• Treatment