Leukemia cutis, a form of extramedullary leukemia involving the skin, is a poor prognostic marker and indicator of imminent hematologic relapse and chemotherapeutic resistance in leukemia patients. Although total skin electron beam radiotherapy is commonly recommended for patients with leukemia cutis, its role and effectiveness remains unclear. Here we describe a 19-month-old infant with acute myelogenous leukemia first diagnosed at 15 months of age. It relapsed in her skin at 18 months of age, associated with raised hyperpigmented lesions on her chest and scalp. Radiation treatment to the total skin surface with radiation therapy resulted in relief of her symptoms before relapse in the bone marrow.