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Philadelphia chromosome-positive acute myeloid leukemia (Ph+AML) is a rare disease characterized by a poor prognosis with resistance to standard chemotherapy. We report a patient with Ph+AML with a minor BCR-ABL-positive mRNA transcript who achieved a hematologic, cytogenetic, and major molecular complete response after cytarabine-based chemotherapy followed by imatinib. After more than 6 months of continuous imatinib therapy, the patient is in continuous complete remission. Our results show that imatinib mesylate is effective in treating Ph+AML.