Primary T-cell lymphoma of the central nervous system lymphoma (T-PCNSL) is an extremely rare tumor. A human T-cell lymphoma virus type I(HTLV-I) associated adult TCL often involves the CNS during its course but disease limited to the CNS is exceptional. We report a case of a 63-year-old male with a highly malignant TCL localized in the bilateral cerebral hemispheres. The patient was HTLV-I positive but no systemic disease was detected after various examinations. We discuss the clinico-pathological features of TCL in the CNS reported in the literature including our case and compare them with those of B-cell lymphomas.