Vaginal leiomyosarcoma is a rare malignancy for which little data exists to guide treatment decisions. We describe a patient diagnosed with primary vaginal leiomyosarcoma who underwent hysterectomy and upper vaginectomy followed by whole pelvic radiation. Approximately 3 months after her initial treatment, she presented with an isolated pulmonary recurrence, which resolved after 12 cycles of chemotherapy. Nineteen months later, a second recurrence was found at the same site. This metastasis was resected and she has remained without evidence of further disease for more than 24 months. Similar to patients diagnosed with uterine sarcomas, resection of pulmonary metastases may offer women with recurrent vaginal leiomyosarcoma improved survival with good quality of life. Thoracotomy should be considered for women diagnosed with pulmonary recurrences of this disease.
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