Abstract: A 24-year-old female presented with purple plaques on the face for 5 months. A 77- year-old male presented with nodule, plaques over the whole body for 5 months, and had a history of "psoriasis" for 18 years. Histologic examination showed a diffuse infiltrate composed of blastic lymphocytes and a grenz zone below the epidermis. Immunohistochemical staining showed positivity of CD123, CD56, CD4 and negativity of CD3 and CD20. The two cases were diagnosed as blastic plasmacytoid dendritic cell neoplasm. The case one only survived for 16 months while the case two lost follow-up.