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CASE REPORT  
Year : 2014  |  Volume : 57  |  Issue : 4  |  Page : 620-622
Female adnexal tumor of probable Wolffian origin


Department of Cytology and Gynecological Pathology, Postgraduate Institute of Medical Education and Research, Chandigarh, India

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Date of Web Publication11-Oct-2014
 

   Abstract 

Female adnexal tumor of probable Wolffian origin (FATWO) is a rare neoplasm arising from the remnants of the mesonephric duct. We report here a case of FATWO in a 70-year-old woman arising from the broad ligament and confirmed on immunohistochemistry. Most of these tumors behave in a benign fashion but certain histological features like hypercellularity, cellular pleomorphism and nuclear atypia as seen in the index case may increase the rate of recurrence and metastasis. Therefore, close follow-up is recommended for FATWO.

Keywords: Broad ligament tumor, female adnexal tumor of probable Wolffian origin, immunohistochemistry, Wolffian origin

How to cite this article:
Gupta AK, Srinivasan R, Nijhawan R. Female adnexal tumor of probable Wolffian origin . Indian J Pathol Microbiol 2014;57:620-2

How to cite this URL:
Gupta AK, Srinivasan R, Nijhawan R. Female adnexal tumor of probable Wolffian origin . Indian J Pathol Microbiol [serial online] 2014 [cited 2021 Oct 28];57:620-2. Available from: https://www.ijpmonline.org/text.asp?2014/57/4/620/142703



   Introduction Top


Female adnexal tumor of probable Wolffian origin (FATWO) is a rare neoplasm arising from the remnants of the mesonephric duct. The varied morphology and the immunohistochemistry of this unusual neoplasm are highlighted in this case report of FATWO arising in the broad ligament in a 70-year-old postmenopausal woman.


   Case report Top


A 70-year-old postmenopausal woman presented with awareness of an abdominal mass on the right side since 1 year. There were no other complaints such as constipation, urinary frequency, weight loss, tenesmus, bleeding per vaginum, dysuria, or hematuria. Pelvic ultrasound scan demonstrated a 6.3 cm × 5.3 cm, oval, heteroechoic solid-cystic lesion with thick wall and multiple septations seen in right adnexa. Serum CA-125 level was 34 (normal values-0-35 U/mL). Her personal and family history was not contributory. She underwent staging laparotomy with total abdominal hysterectomy with bilateral salpingo-oophorectomy and infracolic omentectomy and peritoneal biopsy.

Pathological findings

The tumor measured 6 cm × 6 cm × 2 cm. The outer surface was smooth and grayish white and the cut surface was predominantly solid (80%) with some cystic (20%) areas as shown in [Figure 1]. The tumor was adherent to the right upper lateral aspect of the uterus. The accompanying fallopian tube with fimbria was free of tumor. The uterus, both ovaries, contralateral tube, peritoneal, and omental biopsies were all macroscopically and microscopically free of tumor.
Figure 1: Gross photograph of the right adnexal tumor, with grayish white outer surface and hemorrhagic cut surface, which shows cystic and solid areas

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Hematoxylin and eosin stained formalin-fixed, paraffin-embedded tissue sections from the right adnexal mass were evaluated. They showed an encapsulated tumor with large areas of hemorrhage and hyalinization. At the periphery, there were a few smooth muscle cells adherent to the tumor indicating its close relationship to the fallopian tube. The tumor showed epithelial areas with tubular, cystic and adenomatoid patterns, and an edematous stroma [Figure 2]a. Tubular glands were closely packed and outlined by well-defined basement membrane. Focally cystic dilation and colloid-like material noted in lumen [Figure 2]b. The tubules were lined by low cuboidal epithelial cells with focal stratification. These cells contained round to oval hyperchromatic nuclei with finely granular chromatin. There was a moderate degree of nuclear atypia, however no mitosis were observed [Figure 2]c. The peritoneal and omental biopsies were free of tumor.
Figure 2: Varied histomorphology of female adnexal tumor of probable Wolffian origin. (a) Low-power view showing an adenomatoid pattern with closely packed tubular glands and large area of hyalinization, hemorrhage and edematous stroma (H and E, ×100); (b) tubules showing focal cystic dilation and colloid like material in lumen (H and E, ×200); (c) lining epithelium showing significant hyperchromasia and nuclear atypia but without mitosis (H and E, ×400). Immunohistochemistry showing (d) strong immunoreactivity for CK-7 and (e) focal positivity for calretinin and (f), focal positivity for inhibin, respectively (immunoperoxidase, ×400)

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Immunohistochemistry

Tumor cells were strongly positive for pancytokeratin and Cytokeratin(CK-7) [Figure 2]d and were weakly positive for calretinin and inhibin [Figure 2]e and f. They were negative for CK-20, chromogranin, epithelial membrane antigen (EMA), estrogen receptor (ER), and progesterone receptor (PR).


   Discussion Top


Kariminejad and Scully first reported nine cases of female adnexal tumor, a distinct and unusual neoplasm. The most common site of origin was the broad ligament as seen in six cases with one tumor attached to the posterior leaf of the broad ligament, and five were within the broad ligament [1] as in the index case. Besides this site, this tumor can originate from other sites with remnants of Wolffian duct such as the paratubal region, mesosalpinx or even the paravaginal tissue. The age of presentation is again variable with a wide range from 18 to 81 years with a mean of 50 years. [2] Our case presented in a 70-year-old woman and the site of origin was the broad ligament.

Female adnexal tumor of probable Wolffian origin has been described to have a varied morphology which was highlighted in the series reported by Kariminejad and Scully. [1] They described different morphological patterns including tubular, trabecular, solid, and microcystic (sieve-like) growth patterns. [3] Cellular atypia and an increased number of mitosis favors malignancy, but cases with minimal nuclear atypia and a very low mitotic figure may also occur. [2] Due to the varied morphology the differential diagnosis includes benign neoplasm such as adenomatoid tumor, and malignant neoplasms like mesothelioma and metastatic carcinoma. Immunohistochemistry should, therefore, be performed for confirmation of the diagnosis as well as its distinction from other entities. The largest series of the immunohistochemistry of FATWO has reported tumor cells positive in 100% cases for vimentin, pancytokeratin (AE1+AE3), and CAM 5.2, mostly positive for inhibin and calretinin, focally positive for CK-7 in 88% cases, variably staining for ER and PR in about 24% tumors, EMA positivity in 12% and negative for monoclonal carcinoembryonic antigen and CK-20. [4] Immunoreactivity for c-Kit in recurrent FATWO has also been reported by two authors in separate case reports; in such cases treatment with appropriate tyrosine kinase inhibitors proved beneficial. [5],[6] In the case under report, the tumor was strongly immunoreactive for CK-7 and weakly positive for calretinin and inhibin. Adenomatoid tumor was unlikely as these are strongly positive for calretinin and inhibin. Metastatic carcinoma, endometrial carcinoma, and fallopian tubal adenocarcinoma were all considered in the differential diagnosis especially in view of the significant nuclear atypia and strong diffuse CK-7 positivity. However, the tumor cells were negative for EMA, ER, PR as well as CK-20 and chromogranin. Considering the morphology (nuclear atypia but no mitosis) and this immunohistochemical profile, this tumor was consistent with FATWO albeit with strong CK-7 positivity.

Female adnexal tumor of probable Wolffian origin is considered a tumor of low malignant potential, and although most tumors behave in a benign fashion, multiple recurrences have been reported. [7] Therefore, the most common treatment is total abdominal hysterectomy with bilateral salpingo-oophorectomy. Malignant FATWO has been reported previously metastases to liver, [8] and a case of disseminated malignant FATWO is also on record. [9] These metastases can occur a few years after initial presentation [8] or at presentation. [9] Certain histological features like hypercellularity, cellular pleomorphism, nuclear atypia and increased mitotic activity may increase the rate of recurrence and metastasis. [10] However, the usual outcome is benign in patient treated surgically. The current case had only nuclear atypia but no mitosis and had complete surgery performed. Hence, no adjuvant therapy was considered necessary although this patient has been advised a close follow-up.



 
   References Top

1.
Kariminejad MH, Scully RE. Female adnexal tumor of probable Wolffian origin. A distinctive pathologic entity. Cancer 1973;31:671-7.  Back to cited text no. 1
[PUBMED]    
2.
Taxy JB, Battifora H. Female adnexal tumor of probable Wolffian origin: Evidence of a low grade malignancy. Cancer 1976;37:2349-54.  Back to cited text no. 2
    
3.
Young RH, Scully RE. Ovarian tumors of probable Wolffian origin. A report of 11 cases. Am J Surg Pathol 1983;7:125-35.  Back to cited text no. 3
[PUBMED]    
4.
Devouassoux-Shisheboran M, Silver SA, Tavassoli FA. Wolffian adnexal tumor, so-called female adnexal tumor of probable Wolffian origin (FATWO): Immunohistochemical evidence in support of a Wolffian origin. Hum Pathol 1999;30:856-63.  Back to cited text no. 4
    
5.
Syriac S, Durie N, Kesterson J, Lele S, Mhawech-Fauceglia P. Female adnexal tumor of probable Wolffian origin (FATWO) with recurrence 3 years postsurgery. Int J Gynecol Pathol 2011;30:231-5.  Back to cited text no. 5
    
6.
Steed H, Oza A, Chapman WB, Yaron M, De Petrillo D. Female adnexal tumor of probable Wolffian origin: A clinicopathological case report and a possible new treatment. Int J Gynecol Cancer 2004;14:546-50.  Back to cited text no. 6
    
7.
Brescia RJ, Cardoso de Almeida PC, Fuller AF Jr, Dickersin GR, Robboy SJ. Female adnexal tumor of probable Wolffian origin with multiple recurrences over 16 years. Cancer 1985;56:1456-61.  Back to cited text no. 7
[PUBMED]    
8.
Sheyn I, Mira JL, Bejarano PA, Husseinzadeh N. Metastatic female adnexal tumor of probable Wolffian origin: A case report and review of the literature. Arch Pathol Lab Med 2000;124:431-4.  Back to cited text no. 8
    
9.
Heller DS, Kadire B, Cracchiolo B. Malignant female adnexal tumor of probable Wolffian origin: A case report. J Reprod Med 2011;56:175-7.  Back to cited text no. 9
    
10.
Deen S, Duncan TJ, Hammond RH. Malignant female adnexal tumors of probable Wolffian origin. Int J Gynecol Pathol 2007;26:383-6.  Back to cited text no. 10
    

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Correspondence Address:
Radhika Srinivasan
Department of Cytology and Gynecological Pathology, Postgraduate Institute of Medical Education and Research, Chandigarh -160 012
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.142703

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    Figures

  [Figure 1], [Figure 2]

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