Ovarian serous cystadenofibromas associated with a low-grade serous carcinoma of the peritoneum

Ovarian serous cystadenofibromas associated with a low-grade serous carcinoma of the peritoneum

Annals of Diagnostic Pathology 17 (2013) 302–304 Contents lists available at SciVerse ScienceDirect Annals of Diagnostic Pathology Ovarian serous c...

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Annals of Diagnostic Pathology 17 (2013) 302–304

Contents lists available at SciVerse ScienceDirect

Annals of Diagnostic Pathology

Ovarian serous cystadenofibromas associated with a low-grade serous carcinoma of the peritoneum Stacy A. Hinson ⁎, Elvio G. Silva, K. Pinto Department of Pathology, Baylor University Medical Center, Dallas, TX

a r t i c l e Keywords: Ovary Cystadenofibroma Serous neoplasms

i n f o

a b s t r a c t Ovarian serous cystadenofibromas are benign neoplasms that sometimes have focal areas of borderline serous tumor and rarely have been associated with epithelial proliferations in the peritoneum, resembling implants. We are reporting 2 cases of ovarian serous cystadenofibromas with serous peritoneal lesions of higher grade than the ovarian tumor: 1 case had a serous carcinoma and another 1 a serous borderline tumor. © 2013 Elsevier Inc. All rights reserved.

In 1997, Tornos et al [1] reported in an abstract 9 cases of ovarian serous cystadenofibromas that were associated with proliferating extraovarian serous lesions, resembling implants. These cases were in both peri- and postmenopausal women, and in 7 of the 9 patients, the ovarian tumors were bilateral. It was proposed that women with ovarian serous cystadenofibromas had an increased risk of peritoneal lesions of low-grade serous proliferations. These lesions were proposed to be independent because there was no borderline tumor in the ovary. There were no cases of serous carcinoma in their series. Here, we are reporting 2 cases of bilateral ovarian serous cystadenofibromas (Fig. 1). One is associated with a low-grade serous carcinoma of the peritoneum, whereas the other is associated with borderline serous tumor of the peritoneum.

1. Case report 1 In July of 2009, a 68-year-old postmenopausal gravida 0 para 0 presented to her primary care physician with left-sided abdominal pain consistent with cholelithiasis. At her diagnostic laparoscopy, she was noted to have extensive peritoneal disease, which was biopsied. The pathology on these lesions returned as psammomatous carcinoma. Her CA-125 was elevated at 54, and a computed tomographic scan demonstrated an enlarged periaortic node, a pelvic mass, and omental caking. She was brought back to the operating room approximately 1 month later for total abdominal hysterectomy, bilateral salpingooophorectomy, and extensive enterolysis as well as cholecystectomy and retroperitoneal lymph node dissection. During the operation, gross evidence of carcinomatosis was seen along the small bowel and mesentery as well as the greater curvature of the stomach and the right bladder dome.

⁎ Corresponding author. E-mail address: [email protected] (S.A. Hinson). 1092-9134/$ – see front matter © 2013 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.anndiagpath.2012.05.006

For pathologic evaluation, both ovaries and fallopian tubes were entirely submitted. Serous cyst adenofibromas were found in both ovaries, measuring 3.8 cm on the right and 3.2 cm on the left (Fig. 2). On the ovarian surface, not associated with the cystadenofibromas, there were multiple focal areas of carcinoma similar to the autoimplants seen in borderline tumors (Fig. 3); however, no serous carcinoma was present infiltrating into the ovarian parenchyma. These lesions were associated with psammomatous calcifications. The omentum showed multiple foci of low-grade serous carcinoma associated with psammomatous calcifications (Fig. 4). The foci were up to 1 mm in size and were found in spaces not lined by epithelial cells. A periaortic lymph node showed metastatic low-grade serous carcinoma with prominent psammomatous calcifications scattered throughout the cortical tissue and subcapsular lymphatic sinuses. The tumor cells showed infrequent mitotic activity, and the nuclear atypia ranged from mild to moderate. The omental tumor and the lymph node showed diffuse 3+ positive staining for estrogen receptor and only focal weak positive staining for progesterone. Based on the amount of serous epithelial cells present, a diagnosis of serous carcinoma rather than psammomatous carcinoma was rendered. The final diagnosis was primary peritoneal serous carcinoma, low grade. At her 2-year follow-up, the patient is doing well with no evidence of recurrence. After her optimal surgical debulking, she completed 6 cycles of adjuvant chemotherapy with carboplatin and Taxol. She is being followed up on a biannual basis. 2. Case report 2 In July of 2011, a 49-year-old woman presented to her primary care physician with irregular vaginal bleeding. Sonogram revealed bilateral complex adnexal masses. Her CA-125 was mildly elevated at 24. Computed tomography of the abdomen and pelvis confirmed these findings. She was counseled on the need for definitive diagnosis, and the patient was taken to the operating room where

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Fig. 3. There were multiple areas of serous carcinoma with associated psammomatous calcifications on the ovarian surface (inked black) distant and separated from the adenofibroma in case 1.

matous calcifications and epithelial lined spaces containing serous papillary groups with cell detachments measuring up to 1.5 mm were present. These changes were consistent with borderline serous tumor involving peritoneum. In addition, borderline serous tumor with psammomatous calcifications were seen involving the serosa of the uterus (largest focus, 5 mm) (Fig. 5), and the pelvic washings were positive for epithelial cells with psammoma bodies (differential diagnosis: endosalpingiosis vs low-grade serous neoplasm). The patient is not undergoing any further treatment at this time. 3. Discussion

Fig. 1. There were serous adenofibromas present in both the right and left ovaries in both cases (photo A from the first case, photo B from second case).

she underwent total abdominal hysterectomy and bilateral salpingo-oophorectomy. Histologic examination showed bilateral serous cystadenofibromas (20 cm on the left, 13.4 cm on the right). The cyst walls from each ovary showed focal areas of epithelial proliferation; however, no papillary excrescences, cell detachment, or nuclear atypia was seen. At the ovarian surface, not involving the cystadenofibroma, psammo-

Fig. 2. High-power view of the serous adenofibroma from case 1.

These case reports supports the original suggestion that ovarian serous cystadenofibroma in postmenopausal women may have a risk of serous lesions in the peritoneum that are of higher grade. This reflects the nature of 2 independent lesions. A larger study of similar cases needs to be reported to determine the ratio of this occurrence and if there is justification to suggest obtaining biopsies of the peritoneum in postmenopausal women with serous cystadenofibromas of the ovary. It has been a longstanding accepted idea that, in patients with ovarian and peritoneal lesions, the peritoneal lesions are considered

Fig. 4. In case 1, the omentum was involved with low-grade serous neoplastic cells in spaces that are not lined by epithelial cells. There are associated psammomatous calcifications present.

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There is some molecular evidence to support this [3,4]. The omentum and other peritoneal lesions can be considered the primary site independent of the ovarian tumor. Usually, the lesions of the ovary and peritoneum are of the same degree. In borderline ovarian serous tumors, peritoneal implants are usually noninvasive [5]. In high-grade ovarian carcinoma, metastases in the peritoneum are also of high grade and invasive. However, in rare cases, benign lesions in the ovary coexist with malignant lesions in the peritoneum. These 2 cases we are reporting and those included in an abstract published by Tornos et al [1] in 1997 were in peri- or postmenaupausal women. Gross inspection and sampling of biopsies from the peritoneum in cases of serous cystadenofibromas in peri- and postmenopausal women might be considered at the time of frozen section. References Fig. 5. In case 2, the uterine serosal surface was involved with borderline serous tumor up to 5 mm in size.

metastases from the ovary. For example, peritoneal lesions composed of cells with features of a borderline malignancy are present in about 30% of serous borderline tumors and are generally referred to as implants. This is supported by the fact that peritoneal implants occur more commonly in ovarian borderline tumors with exophytic lesions [2]. More recently, the possibility that some peritoneal lesions can exist independently from lesions in the ovary has been proposed.

[1] Tornos C, Burke TW, Gershenson DM, Silva EG. Benign ovarian serous cystadenofibromas associated with peritoneal implants: report of nine cases. AbstractMod Pathol 1997;10(1). [2] Segal GH, Hart WR. Ovarian serous tumors of low malignant potential (serous borderline tumors): the relationship of exophytic surface tumor to peritoneal “implants.”. Am J Surg Pathol 1992;16:577–83. [3] Emerson RE, Wang M, Liu F, et al. Molecular genetic evidence of an independent origin of serous low malignant potential implants and lymph node inclusions. Int J Gynecol Pathol 2007;26:387–94. [4] Kowalski LD, Kanbour AI, Price FV, et al. A case-matched molecular comparison of extraovarian versus primary ovarian adenocarcinoma. Cancer 1997;79:1587–94. [5] Slomovitz BM, Caputo TA, Gretz III HF, et al. A comparative analysis of 57 serous borderline tumors with and without a noninvasive micropapillary component. Am J Surg Pathol 2002;26:592–600.