Paecilomyces Sp. are saprophytic fungi, which have rarely been pathogen for human. Herein, we report a case of cutaneous infection with Paecilomyces lilacinus with unusual presentations in a healthy young woman. A biopsy provided an initial diagnosis of fungal elements in the tissue. Multiple positive fungal cultures were obtained from the biopsied tissue. Microscopic and macroscopic examination of the biopsy revealed the presence of Paecilomyces lilacinus. This case was successfully treated by prescribing oral ketoconazole (200 mg/day).