Results: Using appropriate controls (n=14), we identified a total of 104 CpG sites hypermethylated in Ewing sarcoma. Most of hypermethylated genes are related with cell adhesion, cell regulation, development, and signal transduction. We compared the methylation mean of each tumor according to the survival data. The methylation mean was significantly higher in the alive patient group (0.25±0.03) compared to the dead patient group (0.22±0.05) (p=0.0322). However, the methylation mean was not significantly correlated with age, sex, or tumor location. We selected the most popular hypermethylated genes, GDF10, OSM, APC, and HOXA11, but, their methylation levels were not significantly correlated with the survival data. Conclusion: We have characterized the DNA methylation profile of Ewing sarcomas and detected 104 CpG sites that were significantly hypermethylated in Ewing sarcomas. These might therefore play an important role in the development of Ewing sarcomas.