Extramedullary relapse is a pattern of acute myeloid leukemia (AML) relapse post-allogeneic hematopoietic stem cell transplantation (alloHSCT). We present the case of a 43-year-old female patient with acute mix cell leukemia who had isolated extramedullary relapse develop in both breasts 2 years after alloHSCT, the first reported case in adult patients in the literature to the best of our knowledge. She received further chemotherapy with pemetrexed (Alimta) intravenous injection (500mg/D1, every 3 weeks is one course of treatment). After 2 courses of treatments, her swelled breasts were shrunk remarkably and no obvious side effects were found. We therefore conclude that pemetrexed treatment may be an effective regimen for breast extramedullary leukemia. Review of the literature about extramedullary relapse and breast involvement as well as the treatment of these cases were further discussed.

Disclosures: No relevant conflicts of interest to declare.

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