A 36-year-old man with underlying chronic lymphocytic leukemia had left arm swelling for a duration of 3 months. Clinically, the affected arm was swollen, erythematous and tender. Epicoccum nigrum was isolated from the culture of the tissue that was obtained intraoperatively. He was treated and responded to voriconazole therapy. To the best of our knowledge, this is the first case of intramuscular abscess as a result of E. nigrum infection in an immunocompromised patient.