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Despite the presence of reports on correlation between major congenital defects and cancer, very few studies have investigated the frequency of minor anomalies in childhood malignancy. The aim of this study was to determine the prevalence of minor anomalies in children with hematological malignancy.A total of 62 well-defined minor anomalies were determined in 109 patients. The patients were compared with age- and sex-matched healthy control subjects.The results of this study showed that, 64.22% of the patients and 26.6% of the controls had at least one minor anomaly. Among the minor anomalies detected, pigmented nevi and café-au-lait spots were significantly more frequent in the patients. The prevalence of minor anomalies in the patients was significantly higher than that of the controls in the present study.Our results contribute to the understanding of the role of genetic factors in childhood hematological malignancies. Future studies may be directed toward identifying the developmental pathways and the relevant genes that are involved in the overlap between childhood hematological malignancies and minor anomalies. Pediatr Blood Cancer 2011;56:258-261. © 2010 Wiley-Liss, Inc.