Complete loss of Rho(D) antigen from red blood cells (RBCs) of individuals with hematologic disorders, though not frequent, has been reported. This case reports the loss of D antigen on the RBCs of a patient with acute myelogenous leukemia and its reappearance when he was in remission. Loss of D antigen expression coincided with worsening clinical and cytogenetic disease. At the time of D antigen loss, the patient also had cytogenetic abnormalities in the bone marrow cells. When he was in remission, the chromosomal abnormalities were no longer detectable, and his RBCs regained D antigen expression.