We report our first case of visceral leishmaniosis (VL) presenting as pancytopenia four years after successful kidney transplantation. VL is a parasitic disease, caused by protozoa of the genus Leishmania, transmitted by the phlebotomies sand fly. A 57-year-old female patient underwent a kidney transplantation in November 2013 because of end-stage renal disease resulting from Hypertension. She received induction immunosuppression with antithymocyte globulin and maintenance with tacrolimus,mycophenolate mofetil and prednisone. In January, 2016 she was admitted to the hospital with macroscopic hematuria and graft disfunction when she had a diagnosis of adenovirus nephritis confirmed by PCR. She fully recovered after reduction in immunosuppression. In August , 2017 she presented with fever, myalgia, loss weight, severe weakness, pancytopenia and splenomegaly when she was submitted to a full investigation and a bone marrow biopsy identified leishmaniosis. The patient was treated with liposomal amphotericin for 7 days with progressive resolution of all symptoms. Initial laboratory values were remarkable for Hemoglobin 7,9g/dL, Total leucocytes 1810/µL, platelets 66000/µL. Prior hemoglobin, total leucocytes and platelet values from 6 months earlier were 12,1 g/dL, 3120/µL and 255 000/µL, respectively. One week after the completion of therapy, follow-up laboratory evaluation revealed improvement with Hemoglobin 9,79g/dL, Total leucocytes 4230/µL, platelets 102000/µL. Health authorities proceeded an investigation in her home town that was negative for the sand fly and for affected dogs. We revised the donor data and he was from an endemic region in the country. As humans can be infected asymptomatically for long periods we believe that it was donor transmitted and the viral infection followed by this parasitic reactivation was a consequence of the immunosuppression. Our report identifies that VL should be suspected in renal transplant recipients presenting unexplained fever, splenomegaly and pancytopenia specially from endemic regions, a bone marrow biopsy is the preferred method of diagnosis. The prognosis is good with proper treatment, but the disease may recur.