A 74-year-old woman presented in April, 2003, with cutaneous lesions of the face by Paecilomyces lilacinus infection. The patient had received predonisolone and azathioprine for 20 months for treatment of autoimmune hemolytic anemia. The lesion first developed on the right lateral eyelid 1.5 years earlier, and gradually enlarged. Physical examination revealed a dark reddish or brownish plaque and scattered papules and abscesses around the plaque on right lateral and lower eyelids, and the cheek. She noted mild tenderness on pressure. Cultures obtained from pus and biopsy specimen showed moulds, and those were identified as Paecilomyces lilacinus. Griseofulvin, 500 mg per day, was not effective for the lesion, so itraconazole, 200-300 mg per day, was administered orally for 11 weeks. Since culture from pus still yielded P. lilacinus despite clinical effectiveness, itraconazole pulse therapy (400 mg daily, 7 days a month) was started. The lesion cleared after three cycles of the pulse therapy.