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Year : 2014  |  Volume : 57  |  Issue : 4  |  Page : 640-641
Extrauterine adenosarcoma arising in omental endometriosis: Rare site of occurrence of a rare tumor


1 Department of Pathology, Jawaharlal Institute of Postgraduate Medical Education and Research, Puducherry, India
2 Departments of Obstetrics and Gynaecology, Jawaharlal Institute of Postgraduate Medical Education and Research, Puducherry, India

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Date of Web Publication11-Oct-2014
 

How to cite this article:
Kar R, Verma SK, Papa D, Sylvia MT. Extrauterine adenosarcoma arising in omental endometriosis: Rare site of occurrence of a rare tumor . Indian J Pathol Microbiol 2014;57:640-1

How to cite this URL:
Kar R, Verma SK, Papa D, Sylvia MT. Extrauterine adenosarcoma arising in omental endometriosis: Rare site of occurrence of a rare tumor . Indian J Pathol Microbiol [serial online] 2014 [cited 2019 Dec 6];57:640-1. Available from: http://www.ijpmonline.org/text.asp?2014/57/4/640/142720


A 30-year-old P1L1 female presented with complaints of abdominal distension of 1-month duration. Ultrasonogram revealed a complex mass postero-superior to the uterus with presence of free fluid. Contrast enhanced computed tomography abdomen showed a predominantly solid, lobulated? bilateral malignant ovarian mass with ascites, omental deposits and loss of fat planes. The patient underwent ascitic fluid analysis for malignant cytology which was given positive on one occasion. She underwent an ultrasound-guided fine needle aspiration cytology which was reported as showing some atypical cells with spindle-shape morphology. With a presumptive diagnosis of bilateral ovarian malignancy, the patient was taken up for surgery after one cycle of preoperative chemotherapy.

Intraoperatively, ovaries appeared normal. There was hemorrhagic ascites and fleshy to friable necrotic masses in the peritoneal cavity with a large fleshy mass in the omentum and the rectovaginal septum. Uterus, ovaries, tubes were studded with these deposits. No deposits were seen over the diaphragm, liver or other viscera. Intraoperative debulking specimen from the mass was sent for frozen section. Sections from the fleshy areas showed ciliated columnar epithelium lined tissue with subepithelium showing endometrial type of glands with stroma with scattered inflammatory cells and occasional scattered atypical cells. Sections from the friable areas showed extensive necrosis. No conclusive report of malignancy could be given. Subsequently, the patient underwent complete surgery and uterus with bilateral tubes and ovaries were removed along with omentectomy and removal of the remaining friable fleshy mass.

Sections from the friable mass showed a tumor composed of both epithelial and stromal component [Figure 1]. The epithelial component was composed of glands and cleft-like spaces with endometrial lining exhibiting tubal metaplasia. There was focal atypia and stratification. Few areas had normal appearing endometrial glands surrounded by endometrial stroma. The stromal component showed a marked overgrowth compared to epithelial component. Periglandular stromal cellularity was increased with moderate pleomorphism and - occasional mitosis. Secondary changes in the form of stromal decidualization, fibrinous exudate, and hemorrhage were also noted. Immunohistochemical stain showed the epithelial component to be positive for epithelial membrane antigen and cytokeratin (CK)7. Estrogen receptor, progesterone receptor, and vimentin were positive in the glands and stroma. CD10 showed focal positivity in the stroma. CK20, carcinoembryonic antigen, CA-125, CD68, and desmin were negative. Sections from the uterus, tubes and right ovary were normal. Left ovary showed surface invagination by endometriotic focus.
Figure 1: Sections from the omental mass showing (a) endometriosis with presence of endometrial glands and stroma (H and E, ×40); (b) estrogen receptor positivity in the glands and stroma (IHC, ×100); (c) and (d) presence of cleft-like glandular spaces having endometrial lining and periglandular increase in stromal cellularity (cambium layer) (H and E, ×40); (e) and (f) CD10 positivity in the stroma surrounding the endometrial glands (IHC, ×200) and (IHC, ×400); (g) stromal component with moderate pleomorphism and secondary changes of fibrin deposition seen adjacent to the leaf-like glandular spaces (H and E, ×100); (h) occasional atypical mitosis seen in the stroma (H and E, ×400). Section from ovary showing (i) Focus of endometriosis on the surface of the ovary (H and E, ×40) with inset showing areas stromal decidualization (H and E, ×100)

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The differential diagnoses includes adenofibroma, adenosarcoma or carcinosarcoma of the omentum. The final diagnosis was an adenosarcoma of the omentum with the possible origin from omental endometriosis. The stromal component in an adenosarcoma can be low grade or high grade and can be homologous or heterologous. This case had stromal overgrowth with a homologous low-grade stromal component.

Adenosarcoma is a rare mixed mullerian tumor composed of benign epithelial and malignant stromal component. Most commonly, adenosarcomas arise from the endometrium, including the lower uterine segment, but rare tumors arise in the endocervix and within the myometrium, probably from adenomyosis. Rarely, adenosarcomas have an extrauterine location and involve the ovary, pelvic tissues, or intestinal serosa. [1] Rekhi et al. (2012) reported the largest case series of 19 cases of adenosarcoma in India. All of them were either from uterus, cervix or ovary. [2] Extrauterine mullerian adenosarcomas have been rarely reported in the omentum [3] and are reported at younger age than uterine counterparts and have more aggressive clinical behavior because of the invasion to adjacent pelvic organs at the time of diagnosis, and recurrence. [4] Our patient was a young lady with an aggressive presentation.

Diagnosing adenosarcoma based on ascitic fluid cytology has been reported very rarely. [5] Our case had malignant cells in the ascitic fluid with some of them having spindle cell morphology. Another scenario encountered is the difficulty in diagnosis of the tumor on frozen section. Few cases in the literature have been reported as low-grade mesenchymal neoplasm or benign condition on frozen sections. [4] We had difficulty in our case since the frozen sections showed endometrial glands with dense inflammation and necrosis with few atypical cells. Hence, a descriptive report was given, and the difficulty conveyed to the operating team who went ahead with a complete surgery.

The tumorigenesis of extrauterine mullerian adenosarcoma is complex. Two mechanisms are considered: It is thought to arise from pluripotent mesothelial and mesenchymal cells of the pelvic cavity or from endometriotic deposits. [3] Since our patient had surface endometriosis of left ovary, and also, evidence of histological transition between normal appearing endometriotic foci and the tumor in the omental deposits, origin of adenosarcoma from omental endometriosis seems more plausible.

A rare site of occurrence of a rare tumor with diagnostic difficulty in ascitic fluid cytology and frozen section has been presented.


   Acknowledgment Top


The authors gratefully acknowledge the help of Dr. Neelaiah Siddaraju, Professor and Dr. Arun Roy, Senior Resident, in manuscript preparation.

 
   References Top

1.
D'Angelo E, Prat J. Uterine sarcomas: A review. Gynecol Oncol 2010;116:131-9.  Back to cited text no. 1
    
2.
Rekhi B, Deodhar KK, Maheshwari A, Menon S, Kerkar R, Bajpai J, et al. Clinicopathological spectrum of 19 adenosarcomas of female genital tract, including uncommon clinical associations and immunohistochemical profile, reviewed at a single institution. Indian J Pathol Microbiol 2012;55:326-32.  Back to cited text no. 2
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3.
Visvalingam S, Jaworski R, Blumenthal N, Chan F. Primary peritoneal mesodermal adenosarcoma: Report of a case and review of the literature. Gynecol Oncol 2001;81:500-5.  Back to cited text no. 3
    
4.
Can B, Kuçukali T, Ayhan A, Ozkaya O, Duruka N. Primary extrauterine adenosarcoma. A report of 2 cases. Turk J Cancer 2008;38:26-9.  Back to cited text no. 4
    
5.
Hirakawa E, Kobayashi S, Miki H, Haba R, Saoo K, Yamakawa K, et al. Ascitic fluid cytology of adenosarcoma of the ovary: A case report. Diagn Cytopathol 2001;24:343-6.  Back to cited text no. 5
    

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Correspondence Address:
Rakhee Kar
Department of Pathology, Jawaharlal Institute of Postgraduate Medical Education and Research, Puducherry - 605 006
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.142720

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