Hydroxyurea is a chemotherapeutic agent used extensively for myeloproliferative disorders. Cutaneous side effects have been described during long-term hydroxyurea treatment. We described the occurrence of multiple squamous cell skin carcinomas in a patient treated with hydroxyurea for chronic myelogenous leukemia. The lesions were removed and the hematological therapy switched to busulfan. In a previously reported case, the development of cutaneous epithelial cancers required the discontinuation of hydroxyurea, in addition to the surgical excision of the neoplastic lesions. Since squamous cell carcinoma is a malignant cutaneous neoplasm that can metastatize, the surveillance of skin changes is advisable during hydroxyurea treatment.