Refractory thrombocytopenia is a rare variant of refractory cytopenia with unilineage dysplasia. The dysplasia is generally confined to the megakaryocytic lineage and is characterized by 10% or more dysplastic megakaryocytes of at least 30 megakaryocytes evaluated. Blasts are rarely seen in the blood and are always <1% of the white blood cells and myeloblasts account for less than 5% of the nucleated bone marrow cells. It is associated with refractory thrombocytopenia and rarely bicytopenia. Distinction from chronic autoimmune thrombocytopenia is critical and may be extremely difficult clinically and morphologically.