Extramedullary relapses of acute lymphoblastic leukemia (ALL) in children and young adults are rare and in most cases are localized in the central nervous system, testes, or both. We describe a rare case of extramedullary relapse of ALL in the breast of a 17-year-old girl. The patient, who had been diagnosed with ALL 1 year before and had been in complete remission for 5 months, was admitted to the hospital for investigation of a mass in her left breast. On clinical examination, she had a large, palpable, nontender mass in her left breast. Mammography revealed a very dense mass. On gray-scale and power Doppler sonography, the appearance of the mass was consistent with malignancy. Histopathologic examination of a surgical biopsy specimen permitted accurate diagnosis of ALL.