Cryptococcus neoformans (C. neoformans) is a ubiquitous yeast-like fungus that has been a common opportunistic human pathogen, especially in immunocompromised patients. Although skin lesions due to C. neoformans are found in 10-15% of patients with systemic cryptococcosis, primary cutaneous cryptococcosis without systemic infection is rare and now considered a distinct clinical entity. We report a case of primary cutaneous cryptococcosis in a patient with iatrogenic Cushing’s syndrome. A 73-year-old woman presented with pain and discharge from lesions on her left forearm. The patient had been treated with oral corticosteroids for 20 years, and as a result had developed iatrogenic Cushing’s syndrome. A skin fragment of the ulcer was cultured, and the encapsulated fungus were isolated and identified as C. neoformans using a Vitek2 system (Vitek2 ID-YST, bioMérieux, France) and API 20C (bioMérieux). Concurrent blood and urine cultures were negative for growth. At first, she was treated with antibiotics due to suspicion of cellulitis. After surgical resection and treatment with systemic and oral fluconazole, her wound was improved with scar. Primary cutaneous cryptococcosis should be considered when skin lesions are not responsive to antibiotics and accurate identification is important for proper treatment. (Korean J Clin Microbiol 2012;15:70-73)