Mesonephric neoplasms of the lower female genital tract are rare. To date, there are scarce reports of benign biphasic vaginal mesonephric lesions, and none have included immunohistochemical and/or molecular analysis. A biphasic neoplasm of mesonephric-type was incidentally identified in the vaginal submucosal tissue of a 55-yr-old woman who underwent a right salpingo-oophorectomy for an ovarian cyst. The well-circumscribed, 5 mm nodule exhibited white-tan, firm homogenous cut surfaces. Microscopic examination showed a lobular arrangement of glands with columnar to the cuboidal epithelium and intraluminal eosinophilic secretions, embedded within a myofibromatous stroma. Cytologic atypia and mitotic activity were absent. Immunohistochemical staining for PAX8 and GATA3 demonstrated diffuse expression in the glandular epithelium, CD10 exhibited a patchy luminal expression pattern, while TTF1, ER, PR, p16, and NKX3.1 were negative. Desmin highlighted a subset of the stromal cells, but myogenin was negative. Whole exome sequencing demonstrated variants of unknown significance in multiple genes including PIK3R1 and NFIA . The morphologic and immunohistochemical profiles are consistent with a benign mesonephric neoplasm. This is the first report describing the immunohistochemical and whole exome sequencing results for a benign biphasic vaginal mesonephric neoplasm. To the best of our knowledge, benign mesonephric adenomyofibroma has not been previously reported in this anatomic location.
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