We report on a case of refractory anemia with trilineage dysplasia and an unbalanced der(1)t(1;10) that resulted in trisomy of the long arm of chromosome 1 (1q) and monosomy of the short arm of chromosome 10 (10p). Fluorescence in situ hybridization showed that the rearranged chromosome contained the centromeres of both chromosomes 1 and 10, leading to a dic(1;10). To our knowledge, a dicentric chromosome involving chromosomes 1 and 10 has never been described in hematological malignancies.