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To investigate the clinical significance of granulocytic sarcoma (GS) in adults with acute myeloid leukemia (AML), 434 consecutive patients with AML were analyzed retrospectively. Forty-five patients (10.4%) with GS at diagnosis were younger (< 0.001), presented with higher white blood cell counts (= 0.03) and were more likely to conform to French–American–British M4 (= 0.001) and M5 (= 0.045) classifications than those without GS. In contrast, no significant difference in frequency of cytogenetic abnormalities was found between the GS and non-GS groups. Treatment outcomes in 260 patients (40 with GS) who underwent intensive chemotherapy, excluding patients with acute promyelocytic leukemia, were investigated. Complete remission rates did not differ significantly between the GS and non-GS groups (75.0% vs 79.1%; = 0.192, respectively) or the 5-year overall survival (OS) rates (39.9% vs 38.7%; = 0.749, respectively). However, the GS group had a significantly higher relapse rate than the non-GS group (74.2% vs 55.3%; = 0.048) and a significantly lower 5-year disease-free survival rate (8.2% vs 25.7%, respectively; = 0.005). When considered together with the results of multivariate analysis, which identified the presence of GS as an independent predictor for disease-free survival time, these findings indicate that GS might identify a high-risk subset of patients with AML. (Cancer Sci, doi: 10.1111/j.1349-7006.2012.02324.x, 2012)