Background Superficial leiomyosarcoma is an exceedingly uncommon malignant tumor which could be located either to cutaneous or subcutaneous tissues. Increased mass size and depth, advanced tumor staging and inadequate surgical excision are the main prognostic factors for poor result. Case presentation We report a rare case of a 71-year-old man with an extensive exophytic lesion (12 × 10 cm) in the anterior-medial side of the proximal right tibia. The lesion was painless and consistently neglected by the patient until a skin trauma caused ulceration of the affected area. Magnetic Resonance Imaging revealed a soft-tissue mass which was well defined from the surrounding bone and muscles. As initial biopsy in another hospital hadn't clarified the true nature of the lesion, new samples were taken and the diagnosis of leiomyosarcoma was established. Laboratory examination showed no distant metastasis and wide excision of the neoplasm was decided. After tumor resection, the remaining soft tissue and skin defect was covered with a gastrocnemius myocutaneous flap. The postoperative period was uneventful and wound healing was followed by local radiotherapy and systemic chemotherapy. At 3 years follow up, no recurrence or metastasis was identified and the patient was able to walk and stand without impairment of his ambulatory status. Conclusion Proper surgical management of soft tissue leiomyosarcoma continues to remain the cornerstone of treatment efficacy and the most important prognostic factor for patients' survival. Reconstruction of the remaining soft tissue defect should be always performed at the same operative time when removal of giant size tumors leaves an uncovered cavity with an inadequate sleeve of muscular and skin tissues.
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