Vaginal leiomyosarcoma is a very rare malignant lesion of the female genital tract accounting for less than 1% of all vaginal malignancies. Surgery remains the primary treatment in prolonging the overall survival. We report on an unusual recurrent low-grade leiomyosarcoma of the vagina in a 51-year-old woman who underwent four surgical resections over a 7-year period. Optimal debulking was performed after her third recurrence and the patient remained free of recurrence 6 years after the fourth surgery. Extensive and aggressive resection can improve the prognosis of selected patients with local recurrence of vaginal low-grade leiomyosarcoma and can be considered a treatment option.
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