Immunosuppressed patients have an increased risk for developing extranodal lymphoma, including testicular lymphoma. In AIDS patients, primary testicular lymphoma has been reported as an initial manifestation of the disease. These patients typically present at an early age; their lymphomas usually have aggressive histologic appearance and are associated with poor prognosis. We report a testicular lymphoma consistent with diffuse large B-cell lymphoma (DLBCL) in an AIDS patient and we review the literature on primary testicular lymphoma in AIDS patients.