Introduction Autoimmune disorders, including autoimmune cytopenias, are more common in patients with myelodysplastic syndrome (MDS) and may share with MDS the same steps of pathogenesis. Some patients with MDS have antibodies against red cells. Case report We describe herein a 79-year-old patient who presented with fatigue, jaundice and pancytopenia. She was diagnosed with warm-antibody autoimmune hemolytic anemia (AIHA) and synchronous MDS. Management and outcome: In our patient, AIHA responded to the hypomethylating agent 5-azacitidine used for the treatment of MDS. Six months later, the patient remains in clinico-laboratory remission for both MDS and AIHA. Discussion/conclusions Our case indirectly suggests that 5-azacitidine led to a decrease in autoantibody production by the auto-reactive B-cell clone in MDS leading in turn to a diminished rate of autoimmune hemolysis. If our observation is accurate, we believe that similar reports will populate the scientific literature in the future years.

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