Synovial sarcomas (SS) account for 5 to 10 percent of soft-tissue sarcomas, and typically arise in the para-articular regions of adolescents and young adults. Nonetheless, SS can rarely occur in other regions of the body. Here we present a first clinical literature report of a patient with a SS arising from the vaginal wall. A 40 year-old patient who present a necrotic polypoid lesion measuring 50 mm and extending from the external urethral meatus to the middle part of the anterior vaginal wall. The Biopsy showed a poorly differentiated SS with abundant necrosis and a SYT-SSX1 mutation. Staging CT scan was negative for distant metastases. The patient, prior to the radical surgery, received neoadjuvant chemotherapy (ifosfamide and epirubicin) for three cycles. She underwent post operative external and brachytherapy (50 Gy) due to close margins (< 1mm) on pathologic specimen. She relapsed eleven and sixteen months later with lung metastases which both times were successfully removed by surgical resection. At twenty-five months from diagnosis, the patient is alive without further evidence of disease. In summary, in the presence of unfavourable prognosis factors, the neoadjuvant chemotherapy could be the primary approach to reduce the tumor size and the risk of distant micro-metastases allowing a less aggressive radical surgery if tumor is located in non-extremity site. Multidisciplinary approach, if no influencing overall survival and disease free survival, may improve the quality of life. In fact in our patient we obtained a complete clinical control in the pelvis avoiding pelvic exenteration with neoadjuvant chemotherapy.

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