Abstract
This case report and literature review describes a 60-year-old woman from Southern Italy, diagnosed with synovitis, acne, pustulosis, hyperostosis and osteitis (SAPHO) syndrome in 2015, who achieved sust ained remission through leflunomide therapy. Initially, the patient presented with severe sternoclavicular pain, osteitis and palmoplantar pustulosis. Following leflunomide initiation, the patient experienced significant clinical improvement, maintaining remission for ~5 years, even after a dose reduction to 20 mg on alternate days. No adverse events were reported during the entire treatment period. This case is contextualized within a concise review of the literature on leflunomide use in SAPHO, highlighting its potential as a long-term, low-dose therapeutic option with a favorable safety profile.