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CORRESPONDENCE
Year : 2013  |  Volume : 9  |  Issue : 3  |  Page : 529-531

Osteoid differentiation in mesodermal (mullerian) adenosarcoma of ovary


Department of Pathology, Gujarat Cancer and Research Institute, M.P. Shah Cancer Hospital, Ahmedabad, Gujarat, India

Date of Web Publication8-Oct-2013

Correspondence Address:
Trupti Patel
Room # 412, Department of Pathology, The Gujarat Cancer and Research Institute, NCH Campus, Asarwa, Ahmedabad, Gujarat State
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0973-1482.119371

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 > Abstract 

A 55-year-old female presented with abdominal pain and 10 cm mass per abdominal examination. Computerized tomography scan of abdomen and pelvis revealed a heterogeneously enhancing solid cystic mass right ovarian mass and mild ascites. Surgery was performed. Specimens were sent for examination. Microscopic examination revealed an admixture of benign but occasionally atypical appearing mullerian type glands with sarcomatous stroma. Solid area showed undifferentiated tumour cells. Atypical mitoses and necrosis were also seen. Areas with extensive benign osteoid surrounded by fibroblastic stroma were also present. Glandular component showed positivity for CK-7, AE-1 and EMA while sarcomatous areas showed positivity for vimentin only. Mullerian adenosarcoma of ovary with sarcomatous overgrowth (SO) having heterologous component was confirmed. Postoperative 3 cycles of chemotherapy was given and the patient was well till date (three months after surgery).

Keywords: Adenosarcoma, osteoid, ovary


How to cite this article:
Patel T, Gupta A, Trivedi P, Shah M. Osteoid differentiation in mesodermal (mullerian) adenosarcoma of ovary. J Can Res Ther 2013;9:529-31

How to cite this URL:
Patel T, Gupta A, Trivedi P, Shah M. Osteoid differentiation in mesodermal (mullerian) adenosarcoma of ovary. J Can Res Ther [serial online] 2013 [cited 2020 May 30];9:529-31. Available from: http://www.cancerjournal.net/text.asp?2013/9/3/529/119371


 > Introduction Top


Mullerian adenosarcoma, an uncommon mixed tumor of the uterus first described by Clement and Scully in 1974 has distinctive clinico-pathological features, characterized by a malignant low grade stromal component and a generally benign, but occasionally atypical glandular component. [1] Though rare, histologically similar extra uterine tumours originating in ovary, on the peritoneum, in intestinal endometriosis, at the vaginal apex, in hepatic endometriosis and in the fallopian tube have been reported in literature. [2],[3] Commonly described heterologous sarcomatous components in Mullerian adenosarcoma are embryonal rhabdomyosarcoma, islands of fetal type cartilage, lipomatous differentiation and skeletal muscles within an endometrioid sarcomatous component. [2] In ovarian adenosarcoma, however, osteoid as a heterologous mesenchymal component has not been described yet. This report documents a rare case of ovarian adenosarcoma having osteoid in mesenchymal areas.


 > Case Report Top


A 55-year-old postmenopausal Indian female presented with history of lower abdominal pain for past two weeks. A firm to hard mobile mass was felt on per abdominal and per vaginal examination. Serum CA-125 levels were 54.4 U/ml (normal range 0-35 U/ml). Computerized tomography scan of abdomen and pelvis revealed a heterogeneously enhancing solid cystic mass arising from pelvis with internal calcification, suggestive of right ovarian mass. Mild ascites was present, which was cytologically negative for malignant cells. Ovarian mass was excised and sent for frozen section, which was reported as malignant undifferentiated tumour. Total abdominal hysterectomy, bilateral salpingo-oophorectomy, infracolic omentectomy along with excision of tissue from pouch of Douglas was done and sent for routine histopathological examination. Microscopic examination revealed an admixture of benign but occasionally atypical appearing mullerian type glands with sarcomatous stroma [Figure 1]. Most of the glands were endometrial type with some glands showing atrophy and pseudostratification. Single focus of squamous metaplasia was also seen. Few glands were lined by serous type epithelium. Few foci showed tumor cells arranged in small tubules with elongated nuclei - Sex cord - like elements (SCLEs). Sections from solid area showed undifferentiated tumor cells arranged in nests and trabeculae separated by thick fibroblastic septa. Tumour cells were medium to large with hyperchromatic nuclei, oval to spindly with 15-18 mitoses/10 high power fields (hpf). Atypical mitoses and necrosis were also seen [Figure 2]. At places cells were arranged in palisaded pattern and also forming pseudo-rosettes around myxoid and chondroid matrix. Areas with extensive benign osteoid surrounded by fibroblastic stroma were also present. Foci of adipose tissue were also seen along with osteoid [Figure 3].
Figure 1: Microphotograph showing mullerian component with sarcomatous area (H and E stain, 10 ×)

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Figure 2: Microphotograph showing sarcomatous component with mitosis (H and E stain, 40 ×)

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Figure 3: Microphotograph showing osteoid with lipomatous differentiation (H and E stain, 40 ×)

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A panel of IHC was given which included cytokeratin-7, AE-1, vimentin, epithelial membrane antigen (EMA), actin, desmin, S-100, calretinin, alpha-inhibin, CD 10 and CD 99. For CK7 clone OV-TL 12l30 (Diagnostic Biosystem), AE1 clone AE1/AE3 (Diagnostic Biosystem), EMA clone GPl.4(Diagnostic Biosystem), Vimentin clone V9 (Biogenex ), Actin clone 1A4 (Diagnostic Biosystem), Desmin clone 33 (Biogenex), S-100 clone 15E2E2 (Biogenex), Calretinin clone CRTOl (Diagnostic Biosystem), a-inhibin clone Rl (Dako,), CD10 clone 56C6 (Neo marker), CD99 clone HO36-1.1(Neo marker). Glandular component showed positivity for CK-7, AE-1 and EMA while sarcomatous areas showed positivity for vimentin and remaining markers were negative. Final diagnosis was given as mullerian adenosarcoma of ovary with sarcomatous overgrowth (SO) having heterologous component. Tissue from Pouch of Douglas showed presence of metastatic tumor (FIGO stage IIB). [ . [4] Postoperative 3 cycles of chemotherapy was given and the patient was well till date (three months after surgery).


 > Discussion Top


After recognition of mullerian adenosarcoma of ovary its numbers are increasing in the literature; some of which differ from their more common uterine counterpart including a more aggressive behaviour. As compared to its uterine counterpart, mullerian adenosarcoma of ovary occur around 50 years and presents with palpable pelvic mass as in our case, while for the former the peak age incidence is eighth decades and presented with abnormal vaginal bleeding. Mullerian adenosarcoma of ovary are generally similar microscopically to their uterine counterpart except few areas like SCLEs were seen in 15% of cases in contrast to 7% of uterine adenosarcoma and SO was seen in 30% of the ovarian tumor as compared to 8% in the uterine counterpart. [2],[5] Our case also had minor foci of SCLEs and SO. The histogenesis of ovarian adenosarcoma is unknown, but an origin from endometriosis is suggested [3] Another possible source of origin is surface epithelium and ovarian stroma. [2] Heterologous element of mullerian adenosarcoma of ovary most commonly contains embryonal rhabdomyosarcoma, islands of fetal type cartilage, lipomatous differentiation and skeletal muscles within an endometrioid sarcomatous component. [2] Our case is unique in such a way that along with foci of lipomatous differentiation tumor also showed many areas of benign osteoid. It is described in its uterine counterpart and in malignant mixed mullerian tumour of ovary but not in ovarian adenosarcoma till date. [6],[7],[8] The presence of heterologous components like striated muscle fibers, lipoblast, cartilage and osteoid are indicative of malignant behavior. [7],[9] The differential diagnosis of mullerian adenosarcoma with SO, with heterologous component and having SCLEs areas of ovary includes endometrial stromal sarcoma (ESS), immature teratoma and sex-cord-stromal tumour respectively. ESS occurs in the same age group and is usually unilateral. Thorough sampling is required to exclude minor foci of adenosarcoma as it has poor prognosis compared to former. Immature teratomas most commonly occur in first three decades of life and are almost non-existent after menopause and contain embryonal neuroectodermal elements and endodermal derivatives in almost all cases. [8] When ovarian adenosarcoma contain predominant SCLEs, sex cord tumour may be entertained. Typical diagnostic areas of adenosarcoma need to be found out after thorough sampling. The presence of a rare gland and endometrioid appearance of the stromal component are diagnostic clues. Lastly the presence of marked epithelial atypia even in absence of invasive epithelial component suggest malignant mixed mullerian tumour.


 > Conclusion Top


Benign osteoid can be a component of mullerian adenosarcoma of ovary. Mullerian adenosarcoma of ovary has poorer prognosis than its uterine counterpart, with recurrence in almost two third of cases.

 
 > References Top

1.Clement PB, Scully RE. Mullerian adenosarcoma of the uterus: A clinicopathologic analysis of 10 cases. Cancer 1974;34;1138-49.  Back to cited text no. 1
    
2.Eichhorn JH, Young RH, Clement PB, Scully R. Mesodermal (Mullerian) adenosarcoma of the ovary. A clinicopathologic analysis of 40 cases and a review of literature. Am J Surg Pathol 2002;26:1243-58.  Back to cited text no. 2
    
3.Clement PB, Scully RE. Extrauterine mesodermal (mullerian) adenosarcoma. A clinicopathologic analysis of 5 cases. Am J Clin Pathol 1978;69:276-83.  Back to cited text no. 3
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4.Heintz AP, Odicino F, Maisonneuve P, Beller U, Benedet JL, Creasman WT, et al. Carcinoma of the ovary. J Epidemiol Biostat 2001;6:107-38.  Back to cited text no. 4
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5.Clement PB, Scully RE. Mullerian adenosarcoma of the uterus: A clinicopathologic analysis of 100 cases with a review of literature. Hum Pathol 1990;21:363-81.  Back to cited text no. 5
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6.Orenstein HH, Richart RM, Fenoglio CM. Mullerian adenosarcoma of the uterus: Literature review, case report and ultrastructural observations. Ultrastruct Pathol 1980;1:189-200.  Back to cited text no. 6
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7.Farhat MH, Hobeika EM, Moumneh G, Nassar AH. Uterine mullerian adenosarcoma with sarcomatous overgrowth fatal recurrence within two weeks of diagnosis: A case report. J Med Case Reports 2007;1:103.  Back to cited text no. 7
    
8.Scully RE, Young RH, Clement PB. Tumors of the ovary, maldeveloped gonads, fallopian tube and broad ligament. In: Rosai J, editor. Atlas of tumor pathology. 3 rd series, fascicle 23. Bethasda, MD: Armed Forces Institute of Pathology; 1998. p. 107, 126, 128, 141, 260-70.  Back to cited text no. 8
    
9.Kerner H, Lichlig C. Mullerian adenosarcoma presenting as cervical polyp: A clinicopathologic analysis of 7 cases with a review of literature. Obstet Gynecol 1993;81:655-9.  Back to cited text no. 9
    


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