Leiomyosarcoma is a malignant smooth-muscle tumor that has a predilection for the gastrointestinal and female genital tract and is a rare entity in the paranasal sinuses. It is locally fast-spreading and highly aggressive, and the prognosis is poor. We report a rare case of leiomyosarcoma of the maxilla in a patient who sought treatment for maxillary swelling, nasal obstruction with no epistaxis, orbital involvement or cervical lymph node metastasis. The patient underwent total maxillectomy followed by radiotherapy. At present after 5 years of follow up, he is symptom free with no recurrence.
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