Azathioprine is a commonly used immunosuppressive agent in renal transplantation. Thiopurine therapy is associated with an increased risk of solid organ malignancies after renal transplantation. There are few reported cases of azathioprine therapy-related myelodysplastic syndrome/acute myeloid leukemia. We describe a case of post-renal transplant patients developing chronic myelomonocytic leukemia-1 associated with monosomy of chromosome 7, possibly related to prolonged exposure of azathioprine.