Papillary Cystadenoma of the Fallopian Tube Not Associated with von Hippel-Lindau Disease: A Case Report

This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/ by-nc/3.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited. Papillary cystadenoma (PC) is an epithelial tumor believed to be of mesonephric origin 1 that is found rarely in the female genital tract. Since the original report by Gersell and King in 1988, 2 only 14 cases have been reported in the English literature. All of the reported cases occurred in the mesosalpinx or the broad ligament. Most of the tumors (12 out of 14) were associated with von Hippel-Lindau disease (VHL), similar to epididymal tumors. 6 We report a case of PC within the wall of the fallopian tube (rather than in the mesosalpinx or the broad ligament) in a patient with no clinical history of VHL. A comprehensive review of the literature with regard to differential diagnosis and immunohistochemical profile is provided. A 44-year-old multipara woman presented with a complaint of a menorrhagia for six months. She and her family had no history of VHL. Ultrasonogram showed multiple solid masses on the uterus, suggesting leiomyomas. The laboratory findings were unremarkable, except for a slightly decreased hemoglobin level (11.7 g/dL). Laparoscopy was performed under the diagnosis of uterine leiomyomas. Both fallopian tubes were enlarged and showed adhesion to the uterine serosa, colonic serosa, and pelvic wall. Total hysterectomy with bilateral salpingectomy and adhesiolysis was performed. The distal half of the left fallopian tube was slightly enlarged and the outer surface was smooth. On section, a solid grayish-yellow mass that measured 3.9×1.4×1.3 cm was observed along the long axis of the fallopian tube (Fig. 1). On microscopic examination , a papillary cystic tumor was noted within the wall of the fallopian tube pushing against the surface epithelium. The papillae were short and blunted and lined by a single layer of bland nonciliated cuboidal cells with clear to eosinophilic cy-toplasm. The nuclei were round with fine chromatin and occasional inconspicuous nucleoli (Fig. 2). Psammoma bodies and mitotic figures were not found. There was a focus of endometri-osis at the adhesion site. The resected uterus contained multiple leiomyomas and the myometrium showed adenomyosis. The right fallopian tube showed chronic inflammation. Immunohistochemical examination was performed according to a previously reported method. 7 The tumor cells showed strong positivity for Cam5.2, cytokeratin (CK) 7, high-molecular weight CK, …