Background. There are limited reports of thrombosis among myelodysplastic syndrome patients exposed to erythropoiesis stimulating agents. It is not clear whether erythropoiesis stimulating agents are associated with an increased risk of thrombosis in myelodysplastic syndromes, as they are among patients with solid tumors.Design and Methods. The association between use of erythropoiesis stimulating agent and transient thrombosis risk in patients with myelodysplastic syndromes was assessed in a case-crossover study nested within a cohort of incident myelodysplastic syndromes patients. The data source was the U.S. Surveillance, Epidemiology, and End Results Medicare-linked database. Cases were defined as having an incident diagnosis of deep vein thrombosis based on Medicare claims. In an adjusted conditional logistic regression models, odds of exposure to erythropoiesis stimulating agents in the 12 weeks immediately prior to the incident deep vein thrombosis (the hazard period) was compared to the exposure odds during a prior 12-week comparison period, separated from the hazard period by a 24-week gap. Results. Within the cohort of myelodysplastic syndromes (n=7,385) there were 212 incident cases of deep vein thrombosis, the mean age was 76.2 (standard deviation=±8.6) years. Use of erythropoiesis stimulating agents was not associated with deep vein thrombosis in the crude nor the adjusted models (ORadj=1.21, 95% CI: 0.60, 2.43). Central venous catheter placement (ORadj=6.47, 95% CI: 2.37, 17.62) and red blood cell transfusion (ORadj=4.60, 95% CI: 2.29, 9.23) were associated with deep vein thrombosis. Conclusions. Despite the link between use of erythropoiesis stimulating agents and thrombosis among patients with solid tumors, this study provides evidence that their safety profile may be different among patients with myelodysplastic syndromes.