A 57-year-old Japanese woman developed multiple subcutaneous cysts located on the pubic region, thighs, back, and buttocks. She had been treated with systemic prednisolone and azathioprine for 3 years because of autoimmune hepatitis. Histologic examination revealed that the lesions were pseudocysts with numerous spores and hyphae. Trichophyton rubrum was identified in culture from the content of several examined cysts. The serum beta-D-glucan level was high (313 pg/mL), and a computed tomographic scan of the chest cavity showed infective embolism in the lung. To our best knowledge, this is the first reported case of multiple-cystic tinea profunda,presumably with systemic dermatophyte infection. Systemic T-cell immunosuppression, as represented by a relatively low percentage of memory T cells and negative delayed-type hypersensitivity tests, is considered to cause this rare manifestation of dermatophytosis. A 57-year-old Japanese woman developed multiple subcutaneous cysts located on the pubic region, thighs, back, and buttocks. She had been treated with systemic prednisolone and azathioprine for 3 years because of autoimmune hepatitis. Histologic examination revealed that the lesions were pseudocysts with numerous spores and hyphae. Trichophyton rubrum was identified in culture from the content of several examined cysts. The serum beta-D-glucan level was high (313 pg / mL), and a computed tomographic scan of the chest cavity showed infective embolism in the lung. To our best knowledge, this is the first reported case of multiple-cystic tinea profunda, presumably with systemic dermatophyte infection. Systemic T-cell immunosuppression, as represented by a relatively low percentage of memory T cells and negative delayed-type hypersensitivity tests, is considered to cause this rare manifestation of dermatophytosis.