We experienced a 58-year-old Korean man with adult T-cell leukemia/lymphoma with features of histologically anaplastic large cell lymphoma involving the skin and testis. The patient had cutaneous nodules in both extremities and a palpable right testicular mass. Right orchiectomy was performed and specimens of removed testicle and skin nodules showed immunohistologically anaplastic large cell lymphoma with T-cell phenotype, and CD30 antigen was positive. A human T-cell lymphotropic virus type 1 (HTLV-1) antibody titer was over 1 : 256 and integration of HTLV-1 proviral DNA pX gene was identified in the peripheral blood mononuclear cells and lymphoma tissue by polymerase chain reaction. Peripheral blood and bone marrow did not show any evidence of characteristic neoplastic T-cells.