A case of acute monocytic leukemia with an apparent isolated skin relapse is reported. The cutaneous involvement was associated with a morphological bone marrow remission but a cytogenetic relapse was present. Regression of the skin lesions was obtained with a protocol including daunoblastine, aracytin and thioguanine, but the patient relapsed and died a few months later without achieving another remission. The relation between cutaneous and medullary disease is discussed.