Infant leukemias are rare in childhood leukemias and have poor prognostic features. Most infant leukemias are presented with MLL rearrangement at diagnosis. t (5; 15) has been reported very rarely in infant leukemias. In addition, the relationship between congenital anomalies and leukemia is controversial. Here we report a case with t (5; 15) (p15; q11.2) infant leukemia and ectrodactyly.