Mycotic osteomyelitis is rare and occurs in immunocompromised patients after inoculation of the pathogen at a penetrating trauma site. Mycotic osteomyelitis due to Scedosporium Apiospermum is extremely rare, with only 13 cases of septic arthritis reported previously. Ours is only the third case of S. apiospermum osteomyelitis in an immunocompromised patient and the only patient with a histopathologic diagnosis from an amputation specimen. Recognition of this pathogen may be delayed due to insidious onset and negative joint fluid cultures, often requiring synovial or bone biopsies to establish the diagnosis. Delay in appropriate treatment may result in disseminating infection or even death.