Paecilomyces lilacinus is an emerging fungal pathogen that is highly resistant to many antifungal drugs. Skin and subcutaneous soft tissue infections caused by this organism are very unusual. Most cases occur in patients with impaired host defenses or following surgical procedures. There has been only one previous report of a histologically confirmed cutaneous infection due to Paecilomyces lilacinus in a patient without predisposing factors. Described here is the second histopathologically proven case of Paecilomyces lilacinus cutaneous infection in a healthy patient without any apparent portal of entry. Prolonged antifungal chemotherapy with itraconazole led to resolution of the skin lesion. This case of sporadic cutaneous infection due to Paecilomyces lilacinus is believed to be the first reported in Europe and the first histopathologically proven case successfully treated with itraconazole.