Alterations in the immunologic response induced by human T cell lymphotrophic virus type I (HTLV-I) predispose the development of disseminated strongyloidiasis. We report a case of an atypical clinical presentation of strongyloidiasis in a patient co-infected with HTLV-I causing scrotal and perineal pain and infertility. Strongyloides stercoralis was found in the analysis of the sperm and specific therapy for strongyloidiasis was associated with disappearance of the symptoms.