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Management of Wolfiann Duct Carcinoma with Cytoreductive Surgery and Hyperthermic Intraperitoneal Chemotherapy

Wolfiann duct carcinoma is a rare neoplasia that arises from the remnant of the mesonephric duct after its degeneration. It was first reported in 1973 by Kariminejad and Scully1 and was regarded as a nonmalignant lesion. After further cases were diagnosed, this original assumption of the disease was shown to be false. This carcinoma usually has low malignant potential but has been documented to have recurrence in the form of aggressive carcinomatosis. After further studies, recurrence rates after initial resection of Wolfiann duct carcinoma were found to be as high as 11 per cent. They were also found to be associated with a 5 per cent mortality rate.2 There has not been a distinct guideline set forth for the management of recurrent disease. Some groups advocate that complete debulking surgery and systemic chemotherapy may produce positive outcomes, but there has not been consensus reached.3 The low number of patients presenting with this disease process limits the ability to test a multitude of varied treatments for this disease. Cytoreductive surgery (CRS) and hyperthermic intraperitoneal chemotherapy (HIPEC) has become widely used in the treatment of primary or secondary neoplasms of the peritoneal cavity, including pseudomyxoma peritonei, peritoneal mesothelioma, and peritoneal carcinomatosis from colorectal origin. The traditional surgical management of complete tumor debulking for Wolfiann duct carcinoma and subsequent systemic chemotherapy fails to take advantage of the benefit of HIPEC. HIPEC uses the peritoneal lining as a barrier from systemic circulation. This barrier enables maximum tolerated doses of intraperitoneal chemotherapy to be much higher than those tolerated systemically. The addition of heat also enhances the chemotherapy’s cytotoxic effects.4 With the benefit of CRS and HIPEC established as an accepted modality of treatment of other peritoneal neoplastic process, we deduced that the treatment of the rare entity of recurrent Wolfiann duct carcinoma with CRS and HIPEC might show some added benefit. Our case demonstrates the treatment of a 50-yearold female with recurrent Wolfiann duct carcinoma with carcinomatosis. She initially presented with abdominal pain, and a 5-cm complex adnexal mass was found along with an elevated CA-125. She underwent total laparoscopic hysterectomy, bilateral salphingooophorectomy, and lymph node dissection. She was initially given no adjuvant therapy. During a follow-up visit, three years later, her CA-125 was noted to be elevated, and a CT scan showed multiple peritoneal metastases (Fig. 1). Biopsy was performed which was consistent with recurrence of the primary tumor. She was then treated with bevacizumab for six cycles. In June 2016, she had normalization of her CA-125 and stable residual disease was seen on the CT scan. She was then started on maintenance bevacizumab. Her CA-125 was noted to increase at a subsequent followup, and she underwent a repeat CT scan, which showed continued stable peritoneal disease. She then underwent