Abstract： A case of leukemia cutis with skin lesions as the primary symptom is reported. A 31-year-old woman presented with mildly painful erythema on the chest and abdomen for three months. Blood routine test was normal in her first visit while pathological examination revealed leukemia cutis. One month later, some purpuras developed on the skin, and at the same time, the second blood routine test and cytomorphologic examination of bone marrow confirmed the diagnosis of acute lymphoblastic leukemia (ALL-L2). Tumor cells were proved to derive from precursor B lymphocytes by immunophenotype analysis of skin biopsy tissue and flow cytometry analysis of peripheral blood. The involvement of central nervous system was confirmed by cerebrospinal fluid test. The patient was relieved from ALL after two times of chemotherapy. However, ALL recurred one month later, and she died of it 2 days later.