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Chronic myeloid leukemia (CML) is a myeloproliferative neoplasm characterized by the uncontrolled production and proliferation of mature well-differentiated granulocytes. There are no previous reports of atypical CML (aCML) arising in a patient with a history of I BCR-ABL i -positive CML.[3],[7]-[9] Presence of I SETBP1 i mutations in patients with aCML is associated with worse overall survival in one study[15] but not another.[25] Other mutations (ie, I TET2 i , I ASXL1 i ) have been described in aCML and can also be seen in healthy individuals[26],[27] but were not detected in our patient. In summary, we present here a patient with CML in the chronic phase who responded to TKI therapy for nearly 13 years before developing I BCR-ABL i -negative disease that best fits the diagnosis of aCML. [Extracted from the article]