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Year : 2014  |  Volume : 57  |  Issue : 2  |  Page : 347-348
Bilateral ovarian serous cystadenofibromas coexisting with an incidental unilateral Brenner tumor and Walthard cell rests in bilateral Fallopian tubes: An unusual case with diagnostic implications and histogenesis


1 Department of Pathology, Tata Memorial Hospital, Parel, Mumbai, Maharashtra, India
2 Department of Surgical Oncology (Gynaecology), Tata Memorial Hospital, Parel, Mumbai, Maharashtra, India

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Date of Web Publication19-Jun-2014
 

How to cite this article:
Rekhi B, Vinarkar S, Shylasree ST. Bilateral ovarian serous cystadenofibromas coexisting with an incidental unilateral Brenner tumor and Walthard cell rests in bilateral Fallopian tubes: An unusual case with diagnostic implications and histogenesis. Indian J Pathol Microbiol 2014;57:347-8

How to cite this URL:
Rekhi B, Vinarkar S, Shylasree ST. Bilateral ovarian serous cystadenofibromas coexisting with an incidental unilateral Brenner tumor and Walthard cell rests in bilateral Fallopian tubes: An unusual case with diagnostic implications and histogenesis. Indian J Pathol Microbiol [serial online] 2014 [cited 2020 May 31];57:347-8. Available from: http://www.ijpmonline.org/text.asp?2014/57/2/347/134748


Sir,

Brenner tumor is known to coexist with mucinous ovarian tumors. [1] Seidman and Khedmati [2] observed 1.3-4% incidence of coexisting Brenner tumor and mucinous cystadenomas. A serous tumor is rarely found coexisting with a benign Brenner tumor. [3]

A 75-year-old postmenopausal female presented with intermittent abdominal pain over a period of time. She disclosed a medical history of controlled diabetes mellitus; hypertension and was on antiepileptic medications, for the past 20 years, until the last 2 years.

Clinically, her general condition was good. Vaginal examination showed a 5 cm × 5 cm sized firm, immobile mass in the posterior fornix. Uterus was small and anteverted.

Magnetic resonance imaging scan showed a 5.8 cm × 4.5 cm × 4 cm sized solid-cystic, multiloculated mass in her left adnexa. Her celomic antigen (CA) 125, carcinoembryonic antigen and CA19.9 serum levels were normal.

Initial fine-needle aspiration cytology (FNAC) from the tumor mass was reported as adenocarcinoma. Thereafter, intraoperative frozen sections were reported as a serous borderline tumor with coexistent benign Brenner tumor. The patient underwent total abdominal hysterectomy with bilateral salpingo-oophorectomy and infracolic omentectomy.

Grossly, the left ovarian mass measured 6 cm × 3.5 cm × 2.5 cm, with an unremarkable capsular surface. Cut surface demonstrated serous fluid, 0.3 cm thickened cyst wall with papillary excrescences and a solid area measuring 1 cm × 1 cm. The right-sided ovary was cystic measuring 2.5 cm × 1 cm × 1 cm with an unremarkable external surface. On cutting open, serous fluid was identified along with papillary excrescences. Endometrium, isthmus, and cervix were unremarkable [Figure 1].
Figure 1: Gross specimen (fresh state) showing bilateral, solid-cystic ovarian masses. Cut surface of the left-sided tumor is cystic with a discrete solid nodule (arrow). Right-sided tube revealing paratubal cysts

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Microscopically, left ovarian mass showed a benign serous cystadenofibroma with focal hypercellularity, mild atypia and a co-existent benign Brenner tumor [Figure 2]. Immunohistochemically, MIB1/Ki67 was low in the cystadenofibroma and in the benign Brenner tumor. The other ovary showed a small benign serous cystadenofibroma. In addition, both the ovarian masses also showed foci of endosalpingiosis. Bilateral  Fallopian tube More Detailss showed Walthard cell rests [Figure 3]. Right-sided fallopian tube showed paratubal cysts.
Figure 2: Histopathological findings of left ovarian mass, displaying two components. The left-side (delineated with arrow heads) represents the solid nodule, revealing features of benign Brenner tumor, including in the inset (on higher magnification). The other component toward the right is a cystadenofibroma displaying tubal-type lining (inset on the same side) (H and E, 40) (Insets: H and E, 200)

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Coexistent occurrence of ovarian mucinous tumors with benign Brenner tumors is documented. [1],[2] To the best of our knowledge, there is no documented case of bilateral ovarian serous cystadenofibromas, coexistent with unilateral benign Brenner tumor, in peer-reviewed English literature.

Initial erroneous diagnosis of adenocarcinoma in the present case, based upon occasional cluster of atypical epithelial cells, on FNAC, indicates its limitation in such cases. On review, these cells displayed cilia and were possibly reminiscent of endosalpingiosis. Endosalpingiosis is a known diagnostic pitfall in cytology specimens. [4] The other possibility is that these cells might be representative of epithelial component of the cystadenofibroma.
Figure 3: (a) Foci of endosalpingiosis identified in the wall of ovarian tissue (H and E, 200). (b) Left-sided fallopian tube revealing Walthard cell rests. H and E, 100. Inset: Walthard cell rests seen on higher magnification (H and E, 200)

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Mild nuclear atypia and focal epithelial stratification led to consideration of a borderline/atypical proliferative serous tumor during intraoperative frozen section evaluation. However, on extensive sampling borderline tumor was ruled out. Serous epithelial tumors are known to exhibit focal proliferation and mild atypia. [5] Adenofibromas can rarely coexist with foci of endosalpingiosis, as noted in the present case. [5],[6] The presence of benign Brenner tumor and Walthard cell rests in the wall of bilateral fallopian tubes reinforces Mόllerian histogenesis of this uncommon tumor. Presence of tubal-type epithelium within the adenofibromas further relates to proximity between the epithelium of ovarian tumors and fallopian tube. Earlier, It has been suggested that benign Brenner tumors can give rise to certain mucinous tumors. [2] We believe that rarely, benign Brenner tumor, as a result of Mόllerian metaplasia, can also lead to the development of serous epithelial tumors, as noted in the present case.

A total hysterectomy with salpingo-oophorectomy was justified in the present case, considering the age of the patient and bilateral ovarian disease. [1] However, she was spared of adjuvant chemotherapy.

Finally, this forms the first documentation of bilateral serous cystadenofibromas coexistent with a benign Brenner tumor and Walthard rests, suggestive of Mόllerian histogenesis. The value of optimal tissue sampling in tubo-ovarian masses cannot be overemphasized. This case also explains diagnostic limitation of FNAC in ovarian tumors, especially when endosalpingiosis can be one of the diagnostic pitfalls.

 
   References Top

1.Kotsopoulos IC, Xirou PA, Deligiannis DA, Tsapanos VS. Coexistence of three benign and a borderline tumor in the ovaries of a 52-year-old woman. Eur J Gynaecol Oncol 2013;34:186-8.  Back to cited text no. 1
    
2.Seidman JD, Khedmati F. Exploring the histogenesis of ovarian mucinous and transitional cell (Brenner) neoplasms and their relationship with Walthard cell nests: A study of 120 tumors. Arch Pathol Lab Med 2008;132:1753-60.  Back to cited text no. 2
    
3.Pschera H, Wikström B. Extraovarian Brenner tumor coexisting with serous cystadenoma. Case report. Gynecol Obstet Invest 1991;31:185-7.  Back to cited text no. 3
    
4.Sidawy MK, Silverberg SG. Endosalpingiosis in female peritoneal washings: A diagnostic pitfall. Int J Gynecol Pathol 1987;6:340-6.  Back to cited text no. 4
    
5.Seidman JD, Cho KR, Ronnett BA, Kurman RJ. Surface epithelial tumors of the ovary. In: Kurman RJ, editor. Blaustein's Pathology of the Female Genital Tract. 6 th ed. New York, NY: Springer-Verlag; 2011. p. 680-772.  Back to cited text no. 5
    
6.Rondez R, Kunz J. [Serous cystadenofibroma of the epiploic appendix. A tumor of the secondary müllerian system: Case report and review of the literature]. Pathologe 2000;21:315-8.  Back to cited text no. 6
    

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Correspondence Address:
Bharat Rekhi
Department of Pathology, 8th Floor, Annex Building, Tata Memorial Hospital, Dr. E.B. Road, Parel, Mumbai - 400 012, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.134748

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