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CASE REPORT Table of Contents   
Year : 2009  |  Volume : 52  |  Issue : 2  |  Page : 222-224
Ossifying luteinized thecoma of the ovary with endometrial adenocarcinoma


Department of Pathology, SDMCMSH, Dharwad, India

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   Abstract 

Luteinized thecomas are one of the sex cord stromal tumors of the ovary rarely undergoing ossification. We report a case of a 66-year-old post-menopausal female with the chief complaint of uterine bleeding of 7 months duration. Endometrial curettage performed showed features of endometrial adenocarcinoma. Follow-up total abdominal hysterectomy revealed bilateral luteinized thecomas of the ovary, one of which had undergone massive ossification converting the ovary into a bone. True bone formation in ovarian tumors is rare. This case is the second in the literature of osseous metaplasia in an ovarian luteinized thecoma, with the association of endometrial adenocarcinoma suggesting its functional status.

Keywords: Endometrial adenocarcinoma, luteinized thecoma, ossification

How to cite this article:
Pervatikar S K, Rao R, Dinesh U S. Ossifying luteinized thecoma of the ovary with endometrial adenocarcinoma. Indian J Pathol Microbiol 2009;52:222-4

How to cite this URL:
Pervatikar S K, Rao R, Dinesh U S. Ossifying luteinized thecoma of the ovary with endometrial adenocarcinoma. Indian J Pathol Microbiol [serial online] 2009 [cited 2020 Feb 24];52:222-4. Available from: http://www.ijpmonline.org/text.asp?2009/52/2/222/48925



   Introduction Top


Luteinized thecomas are sex cord stromal tumors of the ovary having features of fibroma or thecoma along with luteinized stromal cell. [1] Heterotopic bone formation is rare in these tumors. Only one such case has been documented in the literature. [2]

We report a case of bilateral luteinized thecomas, one of which had undergone massive ossification, converting the ovary into a bone.


   Case Report Top


A 66-year-old female presented with a complaint of uterine bleeding of 7 months duration. She was post-menopausal since 8 years. Ultrasound revealed an endometrium of 14mm thickness, normal myometrium and a left-sided solid mass with an acoustic shadow.

Computed tomography showed a left oval adnexal mass of bony density measuring approximately 10cm × 7cm × 4cm. The left ovary could not be distinguished separately.

No significant past or family history was present.

Hematological and other laboratory parameters were normal.

Pathological findings

Endometrial curettage performed showed features of endometrial adenocarcinoma.

The patient underwent total abdominal hysterectomy.

Gross

Hysterectomy of the specimen showed a thickened and irregular endometrium. The right ovary measured 4cm × 3cm × 1cm. 3 A cut section showed fibrous white as well as focal yellowish and calcified areas. The left ovarian tumor weighed 2.5kg and measured 10cm × 8cm × 4cm. 3 It was very hard and had to be cut with a bone saw. The cut section showed dense brown well-formed compact bone separated by fibrous septa [Figure 1].

Microscopy

The endometrium showed tumor composed of cells in solid sheets, papillae and clusters with areas of central necrosis [Figure 2a]. Focal glandular pattern was seen [Figure 2b]. Tumor cells were large and pleomorphic with scant eosinophilic cytoplasm and centrally placed vesicular nuclei with coarse chromatin and prominent eosinophilic nucleoli [Figure 2c]. In certain areas, a well-formed rosette pattern was seen. Tumor cells were seen invading the myometrium in the form of solid nests. Tumor spread was seen up to the cervical stroma. Endocervical glands were uninvolved. It was diagnosed as endometrioid carcinoma (high grade). No vascular or lymphatic invasion was seen. Parametrium, lymph nodes and peritoneal fluid were free of tumor cells.

The right ovary showed clusters of large polygonal luteinized stromal cells having abundant clear to eosinophilic cytoplasm surrounded by fibromatous stroma arranged in a storiform pattern. Areas of hyalinization and calcification were seen. In view of the abundance of luteinized cells, it was diagnosed as luteinized thecoma.

Decalcified sections from the left ovary showed most of the ovarian architecture replaced by mature lamellar bone. Haversian canals were seen at places. Areas of fibrothecoma with luteinized cells in small clusters and singles were seen surrounding the bone [Figure 3] and [Figure 4]. Focal areas of hyalinization and calcification were present. Teratomatous elements were not present. No evidence of endometriosis was seen. It was diagnosed as luteinized thecoma with massive ossification.

Both the ovaries did not show any evidence of metastatic spread of endometrioid carcinoma.

The patient is fine and is doing well. She has not required any active management and has had no complications after 1 year of follow-up.


   Discussion Top


Luteinized thecomas are tumors having features of thecoma or fibroma or fibrothecoma along with luteinized stromal cells in clusters, singles or groups. They are most frequent in post-menopausal females but a significant number also occur in a younger age group compared with typical thecomas. In a study carried out on 46 patients of these tumors, 50% were estrogenic, 39% were non-functioning and 11% were androgenic. [3] In another series described by Roth and Sternberg, two of the four patients in whom the tumor was producing androgens were virilized. [4]

In the present case, the patient attained menopause at a late age of 58 years. She had post-menopausal uterine bleeding with endometrial adenocarcinoma both of which can be attributed to the estrogen-producing nature of the tumor.

Luteinized thecomas are usually unilateral except a special subtype that is associated with fatal sclerosing peritonitis. [5] In our case, although the tumor was bilateral, no evidence of sclerosing peritonitis was found on ultrasound or at surgery.

True bone formation in ovarian tumors is rare except as a component of mature teratoma of the ovary or in mixed mesodermal tumors with heterologous elements. Osseous metaplasia has been documented in well-differentiated sertoli-Leydig cell tumors, mucinous cystadenomas, fibromas and serous papillary cystadenocarcinoma of the ovary. [6] Four cases of typical thecoma with calcification and psammoma bodies have been reported. [7] In a large number of cases in which ossification was seen in the ovary, an associated endometriosis or chronic inflammation was present, but was absent in our case. [8] Our case is different from the only other documented case of ossified luteinized thecoma in that our case was associated with endometrial adenocarcinoma of the uterus, suggesting its functional status.

The cause for bone formation in the ovary is not known. A previous torsion or infarction can explain the bone formation seen in some non-neoplastic lesions of the ovary like endometriotic cyst and in normal ovaries. In the present case, the most probable cause for the bone formation may be hyalinization, dystrophic calcification and subsequent osseous metaplasia. Another theory postulated by some authors is that the tumor may produce bone-forming factors like transforming growth factor β or bone morphogenetic proteins (BMP), causing metaplastic transformation of the undifferentiated mesenchymal stromal stem cells into osteoblasts. [9] BMP are a family of growth factors regulating a wide variety of biological processes like bone formation and psammoma body formation in ovarian tumors.

In conclusion, bone formation in ovarian tumors is probably due to interplay of various factors that are not completely understood at present.

These may be responsible for poorly differentiated adenocarcinoma instead of the well-differentiated adenocarcinoma of the endometrium, which occurs in thecoma or thecosis, and has to be ascertained.

 
   References Top

1.Zaloudek CF. Tumors of the female genital tract. In: Fletcher CM, editor. Diagnostic histopathology of tumors. 3 rd ed. Churchill Livingstone; 2007. p. 593-4.  Back to cited text no. 1    
2.Morizane M, Ohara N, Mori T, Murao S. Ossifying luteinized thecoma of the ovary. Arch Gynecol Obstet 2003;267:167-9.  Back to cited text no. 2  [PUBMED]  [FULLTEXT]
3.Zhang J, Young RH, Arseneau J, Scully RE. Ovarian stromal tumors containing lutein or leydig cells: A clinopathological analysis of fifty cases. Int J Gynecol Pathol 1982;1:270-85.  Back to cited text no. 3  [PUBMED]  
4.Roth LM, Sternberg WH. Partly Luteinized theca cell tumors of the ovary. Cancer 1983;51:1697-704.  Back to cited text no. 4  [PUBMED]  
5.Young RH, Scully RE.Sex cord-stromal, steroid cell and other ovarian tumors with endocrine, paraendocrine, and paraneoplastic manifestations. In: Kurman RJ, editor. Blaustein's pathology of the female genital tract. 5 th ed. Springer; 2002. p. 921-3.  Back to cited text no. 5    
6.Bosscher J, Barnhill D, O'Connor D, Doering D, Nash J, Park R Osseous metaplasia in ovarian papillary serous cystadenocarcinoma. Gynecol Oncol 1990;39:228-31.  Back to cited text no. 6    
7.Young RH, Clement PB, Scully RE. Calcified thecomas in young women: A report of four cases. Int J Gynecol Pathol 1988;7:343-50.  Back to cited text no. 7  [PUBMED]  
8.Zahn CM, Kendall BS. Heterotopic bone in the ovary associated with a mucinous cystadenoma. Mil Med 2001;166:915-7  Back to cited text no. 8  [PUBMED]  
9.Mukonoweshuro P, Oriowolo A. Stromal osseous metaplasia in a ­low-grade ovarian adenocarcinoma. Gynecol Oncol 2005;99:222-4.  Back to cited text no. 9    

Top
Correspondence Address:
S K Pervatikar
SDM College of Medical Sciences and Hospital, Manjushree Nagar, Dharwad - 580 009
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.48925

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    Figures

  [Figure 1], [Figure 2a], [Figure 2b], [Figure 2c], [Figure 3], [Figure 4]

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