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İstanbul Üniversitesi, Cerrahpaşa Tıp Fakültesi, İç Hastalıkları, Medikal Onkoloji BD., İstanbul Primary malignant lymphoma of the testis is rare, constituting only about 5% of all testicular neoplasms. The disease has a tendency to spread to atypical sites including contralateral testis, central nervous system, skin and Waldeyer's ring. The outcome of patients with testicular lymphoma remains poor despite aggressive treatment. Orchiectomy combined with prophylactic scrotal radiotherapy plus intrathecal chemotherapy and systemic combination chemotherapy are the preferred treatments. Our two patients had late treatment failure in the contralateral testis, one also had skin infiltration on the left forearm and died of disease progression. Therefore, we need to discuss the clinical presentation, prognosis and appropriate treatment of primary testicular lymphoma in the view of the literature. Keywords :