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Female adnexal tumor of probable wolffian origin: a clinicopathological case report and a possible new treatment
  1. H. STEED*,
  2. A. OZA,
  3. W. B. CHAPMAN,
  4. M. YARON* and
  5. D. DE PETRILLO*
  1. *Department of Obstetrics and Gynecology, Division of Gynecology Oncology, University of Toronto, Princess Margaret Hospital, Toronto, Ontario
  2. Department of Medical Oncology, University of Toronto, Princess Margaret Hospital, Toronto, Ontario
  3. Department of Pathology, University of Toronto, Princess Margaret Hospital, Toronto, Ontario
  1. Address correspondence and reprint requests to: Dr Helen Steed, Division of Gynecology Oncology, University of Toronto, Princess Margaret Hospital, 610 University Avenue, Room M-700, Toronto, Ontario, Canada M5G 2M9. Email: helensteed{at}sympatico.ca

Abstract

Female adnexal tumors of probable wolffian origin (FATWOs) are rare tumors arising in the broad ligament from the remnants of the mesonephric duct. We report a case of recurrent disease. A 15-year-old girl who presented with a painful pelvic mass underwent a laparotomy with tumor resection. Pathology findings confirmed a FATWO. The tumor recurred within 2 years and was treated with multiple chemotherapy regimens, including a platinum-based drug, and surgery for progressive disease. The tumor was positive for c-kit oncogene (CD 117). Gleevac therapy, a tyrosine kinase inhibitor, was prescribed, and she developed severe persistent lower abdominal pain 2 months later. She underwent a hysterectomy and debulking of retroperitoneal masses. Pathology showed evidence of tumor necrosis, suggesting a possible beneficial effect, and she was recommenced on Gleevac in an effort to prevent recurrences. She is currently asymptomatic, without evidence of disease 10 months after surgery, continuing on Gleevac therapy. FATWOs are very rare tumors. Most cases are benign but have the potential to recur and metastasize. There is limited knowledge about the optimal treatment for this neoplasm. Our patient's favorable response to Gleevac therapy supports the concept of targeted molecular therapy in patients with c-kit-positive FATWO tumors.

  • FATWO
  • Gleevac
  • wolffian

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