PURPOSE: Pilomatrixoma is a benign, usually asymptomatic tumor. It presents clinically as a solitary superficial subcutaneous nodule measuring between 0.5 cm and 5 cm in diameter on the head or upper extremeties and has not been reported after skin graft. The objective of this article is to report our experience in treating pilomatrixoma which occurred after split thickness skin graft on the lower extremity. METHODS: A 56-year-old female was treated in August 2005 with a 0.5 X 0.5 cm firm subcutaneous nodule at recipient site of split thickness skin graft on the left medial thigh. The tumor was successfully removed by complete excision and histologic examination was followed. RESULTS: The diagnosis was pilomatrixoma which was characterized by a dual population of proliferating basophilic cells and diagnostic shadow cells. CONCLUSION: The tumor was successfully treated by complete resection. The authors report this very rare case of pilomatrixoma which occurred at recipient site of split thickness skin graft.