Paecilomyces is a genus of saprophytic fungus that has been associated, in rare instances, with human disease. We report two cases in which Paecilomyces lilacinus was isolated from cutaneous and subcutaneous lesions in an immunocompromised and an immunocompetent host. The first case was a subcutaneous infection due to P. lilacinus in a patient with a renal transplant and diabetes mellitus. The second case was an immunocompetent young woman who developed a cutaneous infection, with no identified predisposing factors. A biopsy from each patient provided an initial diagnosis of fungal elements in the tissues under examination and multiple positive fungal cultures were obtained from the tissue biopsy samples. Both microscopic and macroscopic examinations of the biopsy revealed the presence of P. lilacinus. Each of the two cases was successfully treated with oral ketoconazole (200 mg/day) and itraconazole. We also review previously reported cases in which the clinical history and response to therapy were noted.