Primary bone lymphomas comprise <1 % of all malignant lymphomas, and there have been only a limited number of reports on primary adult T-cell leukemia/lymphoma (ATLL) of bone. Here, we report two cases of primary ATLL of bone. The first case was a 41-year-old woman with multiple bone tumors. She was diagnosed with ATLL of the skull through biopsy and treated with chemotherapy. Although the bone lesions showed transient improvement, the subsequent central nervous system (CNS) invasion of ATLL occurred and she died 7 months after diagnosis. The second case was a 65-year-old man with right coxodynia. He was diagnosed with ATLL through right femoral biopsy, and the lesion improved with chemotherapy. However, CNS invasion of ATLL developed during chemotherapy, and he died 10 months after diagnosis. Both the patients with primary ATLL of the bone reported here experienced CNS invasion. Thus, ATLL treatment should aim to prevent CNS invasion at an early stage.
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