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Arsenic trioxide (As2O3) with or without interferon-α (IFN) was given to 4 patients with relapsed/refractory adult T-cell leukemia/lymphoma (ATLL). Treatment with As2O3 and IFN showed an encouraging response in two moderately-aggressive ATLL patients, while As2O3 alone was ineffective in two patients with very aggressive and rapidly progressing ATLL. Further studies are needed to clarify the role of As2O3 and its usefulness when combined with IFN for the treatment of ATLL.