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E-JCRT CORRESPONDENCE
Year : 2015  |  Volume : 11  |  Issue : 4  |  Page : 1030

Serous adenofibroma of ovary: An eccentric presentation


1 Department of Pathology, Jawaharlal Nehru Medical College, Datta Meghe Institute of Medical Sciences University, Sawangi, Wardha, Maharashtra, India
2 Department of Obstetrics and Gynecology, Jawaharlal Nehru Medical College, Datta Meghe Institute of Medical Sciences University, Sawangi, Wardha, Maharashtra, India
3 Department of Medicine, Jawaharlal Nehru Medical College, Datta Meghe Institute of Medical Sciences University, Sawangi, Wardha, Maharashtra, India

Date of Web Publication15-Feb-2016

Correspondence Address:
Samarth Shukla
Department of Pathology, Jawaharlal Nehru Medical College, Datta Meghe Institute of Medical Sciences University, Sawangi (Meghe), Wardha - 442 005, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0973-1482.150419

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 > Abstract 

Surface epithelial tumors of the ovary comprise over fourth of the ovarian neoplasms. Serous adenofibromas are lesser known variants of serous surface epithelial tumors. Though these tumors have a benign fate, yet they can be misinterpreted clinically and radiologically due to their borderline or malignant gross morphological as well as clinical presentation. The cell of origin of surface epithelial tumors of the ovary is histological debated; however, it is now ascertained to have origins from the ovarian cortical and surface lining. Adenofibromas are known to progress in an indolent manner with metachronous behavior for years. The present case is of an ovarian serous adenofibroma in a young female masquerading as peritoneal carcinomatosis.

Keywords: Epithelial tumors, peritoneal carinomatosis, serous adenofibromas


How to cite this article:
Shukla S, Srivastava D, Acharya S, Dhote S, Vagha S. Serous adenofibroma of ovary: An eccentric presentation. J Can Res Ther 2015;11:1030

How to cite this URL:
Shukla S, Srivastava D, Acharya S, Dhote S, Vagha S. Serous adenofibroma of ovary: An eccentric presentation. J Can Res Ther [serial online] 2015 [cited 2019 Sep 20];11:1030. Available from: http://www.cancerjournal.net/text.asp?2015/11/4/1030/150419


 > Introduction Top


Surface epithelial-stromal tumors are the most common neoplasms of the ovary, and majority occur in women of between fourth to sixth decade. Benign serous tumors of the ovary account for approximately 16% of all ovarian epithelial neoplasms and approximately 30-50% are bilateral. In some serous neoplasms, the fibroblastic stromal component is unduly prominent, appearing grossly as solid, white, nodular foci in an otherwise typical cystic neoplasm. These, too, can be separated into benign (adenofibroma and cystadenofibroma), borderline, and malignant (adenofibrocarcinoma and cystadenofibrocarcinoma) types. [1],[2] The benign serous tumors often arise in the surface or the cortex of the ovaries, often these tumors are metachronous in origin with intervals that range from three to 14 years. Similar tumors in extraovarian sites occasionally accompany benign serous tumors.


 > Clinical history Top


A 25-year-old female, female, para 3, gravid 3 presented with palpable mass in the pelvic area, with fullness of lower abdomen and vaginal bleeding since last 15 days. She has been in good health prior to the present symptommatology. She was examined in the outpatient gynecological clinics, where per abdominal examination revealed a bilateral pelvic mass of approximately 16-18 weeks size, she had a normal uterine size for age, per vaginal examination showed a healthy cervix located mildly high and vagina was healthy, bimanual recto vaginal examination also showed palpable nodules.

Computed tomography (CT) abdomen (spiral axial scans) of abdomen showed evidence of lobulated solid cystic lesions (multiple 20 to 30 of varying sizes nodules), involving both the ovaries. The lesion were extending anterior to the uterus and completely surrounding the uterus. Uterus measured 5.2 × 3.9 × 3.2 cms. Both ovaries were not visualized separate from the lesions. There was evidence of mild ascitis, with suspicion of peritoneal involvement; however, the nodular masses were free and not arising from the mesentery. Radiological impression was of bilateral complex ovarian cyst (malignancy) with ascitis and possible peritoneal deposits.

Based on the clinical and radiological findings, explorative laprotomy was planned. On explorative laprotomy, large round to oval firm whitish, yellow, tan brown-colored solid to cystic nodular bosselated masses were seen involving and encompassing bilateral ovaries and many were present in the peritoneum floating freely. However, these masses were not adherent to any visceral organs including the uterus and the mesentery was also spared. A clinical impression of hydatid cyst disease or malignancy was made. Section was sent for intraoperative frozen section diagnosis which turned out to be benign ovarian tumor. Bilateral salphingo-opherectomy, along with resection and removal of multiple nodular growths found in the peritoneal cavity was also carried out and sent to histopathology for further evaluation and diagnosis.

Grossly, the tumors encompassing both ovaries bilaterally measured to be of size 11 × 7× 8 cm and 9 × 7× 7 cms. Varying sized nodules resected from the peritoneal cavity showed considerable variation in size largest being 6 × 5× 5 cms and smallest being 3 × 2× 2 cms. All the tumor mass were of similar gross appearance appearing as grayish white to tan, tan yellow, and few hemorraghic. The nodules were firm to cystic, cut section in firm nodules showed fibromatous surface with minute cavities scattered throughout the surface of the solid tumor [Figure 1] and [Figure 2]. Cystic nodules had unicolucations with occasional cysts showing multilocualtions. Cysts were filled with serous fluid of watery nature. Microscopically, the solid nodules consisted of predominant stromal fibromatous areas along with presence of glands within the stromal component. Glands are lined by simple cuboidal cells. The cystic nodules also show a simple lining of cuboidal cells, with calcified concretions at places [Figure 3] and [Figure 4].
Figure 1: Gross appearance of multiple tumor nodules resected from bilateral ovarian adenexa as well as removed from the peritoneum

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Figure 2: Gross cut section of the tumor nodules showing predominantly solid fibromatous whitish yellow cut surface in most of the nodules

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Figure 3: H and E stained slide x10 view showing tumor tissue with uniform appearing cuboidal cell lining, at the peripheral rim with extensive fibrocoallgenous solid stroma

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Figure 4: H and E stained slide with x40 view shows tumor tissue with predominant stromal fibrous component with simple cuboidal glandular element embedded in between

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 > Discussion Top


Ovarian serous adenofibromas are uncommon variants of serous surface epithelial tumors of ovary. The usual presentation of an benign ovarian tumor is that of abdominal discomfort or palapable mass in the pelvis or lower abdomen. Further stages of the ovarian tumors can be appreciated with presentation of vaginal bleeding, ascities along with presence of mass even on vagino rectal examination suggestive of spread along bilateral pelvic gutters and cul de sac.

A typical serous adenoma is rather a benign condition with no further consequences, unless left unattended surgically. However, certain variants of benign serous epithelial tumors such as the serous cyst adenofibroma, have clinical presentation mimicking carcinoma like features, at times they can be limited to one ovary or spread to the contralateral ovary. Present case presented with multiple round to oval masses which filled the peritoneal, which showed features of benign cyst adenofibroma on histopathology. The origins of such multiple masses in a benign cyst adenofibroma is perplexing. The serous cyst adenofibromas may present as solid multiple nodules, which may extend into extraovarian sites as in coelomic, peritoneal cavity. Histologically, the tumors have traditionally been thought to derive from the epithelium that normally lines the outer aspect of the ovary, variously referred to as surface, coelomic, or germinal. [3] This epithelium is continuous with the mesothelium that covers the peritoneal cavity, representing a modification of it and sharing with it a common origin and many morphologic features. [4]

However, borderline or low-grade malignant potential ovarian tumors can present rarely with a variety of proliferative epithelial lesions involving the peritoneum with phenotypic features indicative of müllerian differentiation. Theoretically, they could develop through two different mechanisms: (1) Spread from an ovarian (or, less commonly, endometrial or tubal) source; and (2) autochthonous origin from the so-called "secondary mόllerian system," i.e., the pelvic and lower abdominal mesothelium and the subjacent mesenchyme of females. The two most important manifestations of this process are designated, respectively, as implants and endosalpingiosis. Implants can be non invasive (epithelial or desmoplastic) and invasive implants. But due to the extensive fibromatous components in the stroma of these ovarian tumors, some authors have gone as far as suggesting that ovarian tumors arising from this structure should be regarded as mesotheliomas, a proposal that has not met with acceptance, since clearcut immunohistochemical, [5],[6] ultrastructural, and biologic differences between ovarian surface epithelium and extraovarian peritoneal mesothelium exist. [7] Furthermore, ovarian epithelial tumors differ significantly as a group from peritoneal mesotheliomas on morphologic and behavioral grounds, these differences being highlighted by two rare but notable occurrences: The ovarian tumor with a bona fide mesotheliomatous appearance [8] and the existence of intra-abdominal extraovarian malignancies having more resemblance to ovarian carcinomas than to peritoneal mesotheliomas.

There have been two interesting case reports on serous adenofibromas of ovary, both presenting in different clinical spectrums. The first case has been reported by P. E. Hughesdon was mentioned in the Proceedings of the Royal Society of Medicine, [9] where the patient had bilateral ovarian tumor, but because of the pinpoint hemorraghic appearance of both the ovaries along with collection of blood in the pouch of douglasa clinical impression of associated endometriosis was suspected eventually on histopathological examination of the ovaries, adenofibroma was confirmed though in between the stroma there were areas of cleft formation and presence of acini (glandular appearance as well) surrounded with loose stroma, this histopathological feature confirmed a coexisting diagnosis of endometriosis as well.

A second case of ovarian adenofibroma presented with signs mimicking malignancy, on radiological ultrasound as well as CT scans. As the ovarian tumor consisted of complex cystic as well as solid areas the heterogenous radiological scans made the case as suspected to be malignant which post resection was submitted for histopathological studies was diagnosed as Cyst adenofibroma. [10] A CT scan also is of limited value in evaluating this tumor. In a study by Cho et al., all 16 cases of ovarian cystadenofibromas, presenting as complex cystic masses with solid components, were preoperatively misdiagnosed as malignant ovarian neoplasms on CT scan or MRI. [11] Akin to the above two clinical case reports, the present case report of bilateral ovarian mass with ascites on clinical examination and radiological findings was found to be highly suspicious of an infective process or malignancy, and turned out to be benign serous adenofibroma on histopathological examination.

It can be thus concluded that such benign tumors with excellent prognosis, can be at times overlapped clinically and radiologically with a misleading grave diagnosis. It is imperative to utilize frozen section based histopathological intraoperative diagnosis to help aid in immediate management of such tumors, which definitely help as an adjunct to facilitate complete operative management and prevent recurrence.

 
 > References Top

1.
Compton HL, Finck FM. Serous adenofibroma and cystadenofibroma of the ovary. Obstet Gynecol 1970;36:636-45.  Back to cited text no. 1
[PUBMED]    
2.
Czernobilsky B, Bornstein R, Lancet M. Cystadenofibroma of the ovary. A clinicopathologic study of 34 cases and comparison with serous cystadenoma. Cancer 1974;34:1971-81.  Back to cited text no. 2
    
3.
Bell DA. Ovarian surface epithelial-stromal tumors. Hum Pathol 1991;22:750-62.  Back to cited text no. 3
    
4.
Blaustein A. Peritoneal mesothelium and ovarian surface cells--shared characteristics. Int J Gynecol Pathol 1984;3:361-75.  Back to cited text no. 4
[PUBMED]    
5.
Barnetson RJ, Burnett RA, Downie I, Harper CM, Roberts F. Immunohistochemical analysis of peritoneal mesothelioma and primary and secondary serous carcinoma of the peritoneum: Antibodies to estrogen and progesterone receptors are useful. Am J Clin Pathol 2006;125:67-76.  Back to cited text no. 5
    
6.
Comin CE, Saieva C, Messerini L. H-caldesmon, calretinin, estrogen receptor, and Ber-EP4: A useful combination of immunohistochemical markers for differentiating epithelioid peritoneal mesothelioma from serous papillary carcinoma of the ovary. Am J Surg Pathol 2007;31:1139-48.  Back to cited text no. 6
    
7.
Davidson B, Risberg B, Berner A, Bedrossian CW, Reich R. The biological differences between ovarian serous carcinoma and diffuse peritoneal malignant mesothelioma. Semin Diagn Pathol 2006;23:35-43.  Back to cited text no. 7
    
8.
Addis BJ, Fox H. Papillary mesothelioma of ovary. Histopathology 1983;7:287-98.  Back to cited text no. 8
[PUBMED]    
9.
Hughesdon PE. Adenofibroma of ovary with partial differentiation to endometriosis. Proc R Soc Med 1949;42:62.  Back to cited text no. 9
[PUBMED]    
10.
Wasnik A, Elsayes K. Ovarian cystadenofibroma: A masquerader of malignancy. Indian J Radiol Imaging 2010;20:297-9.  Back to cited text no. 10
[PUBMED]  Medknow Journal  
11.
Cho SM, Byun JY, Rha SE, Jung SE, Park GS, Kim BK, et al. CT and MRI findings of cystadenofibromas of the ovary. Eur Radiol 2004;14:798-804.  Back to cited text no. 11
    


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  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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