Background: Chronic Myelogenous Leukemia (CML) is a myeloproliferative disorder characterized by, overproduction of myeloid cells. Hydroxyurea is an antimetabolite which is commonly used in myeloproliferative syndromes. Reported skin changes related to hydroxyurea therapy may range form xerosis to mucocutaneous cancers. Aim: In this study, our aim was to understand and discuss the underlying reason for the development of squamous cell carcinoma (SCC) in patients recieving hydroxyurea and present a case, in whomwe operated to excise the tumoral masses and reconstruct them. Materials and Methods: After recieving hydroxyuea therapy for four years, the patient was diagnosed as squamous cell carcinoma on the upper lip and, on the dorsum of the left hand. She was operated and the tumoral masses were excised. The lesion on the upper lip was reconstructed with nasolabial flap, inner and inferior sides of the flap was skin grafted. The defect on the dorsum of the hand was skin grafted. Results: No complication related to operation occured. Throughout postoperative eight months of follow-up period, the patient had no complaint regarding the operation. Conclusıon: Skin lesions of patients recieving hydroxyurea therapy must be carefully examined and monitored regularly, since these lesions may proceed to skin cancers.