Herein we report a rare case of Epstein-Barr virus-associated primary testicular T-cell lymphoma in a Taiwanese man treated with high-dose chemotherapy and peripheral blood stem cell transplantation. At first, the patient underwent radical orchiectomy, involved-field irradiation, and chemotherapy. Soon after, the lymphoma recurred in the skin, peripheral nerves and central nervous system, for which the patient received high- dose chemotherapy and allogenic peripheral blood stem cell support. Four months later, he died. This case of Epstein-Barr virus-associated primary testicular T-cell lymphoma represents the first to be treated with both high-dose chemotherapy and stem cell transplantation. Relevant studies regarding the treatment of primary testicular T-cell lymphoma are discussed.