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      Müllerian Adenosarcoma Arising From Rectal Endometriosis

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          Abstract

          A Müllerian adenosarcoma is an extremely rare tumor characterized by a stromal component of usually low-grade malignancy and by a benign glandular epithelial component. A Müllerian adenosarcoma occurs mainly in the uterus, but also in extrauterine locations. Extrauterine Müllerian adenosarcomas are thought to arise from endometriotic deposits. A 36-year-old female presented to Daegu Catholic University Medical Center with a symptom of loose stool for several months. The imaging studies revealed a rectal mass, so she underwent a laparoscopic low anterior resection. Although extemporary pathology revealed an inflammatory myofibroblastic tumor, the final histologic diagnosis was a Müllerian adenosarcoma arising from rectal endometriosis. To our knowledge, except a concomitant rectal villotubular adenoma, cases of Müllerian adenosarcomas arising the rectal wall are rare. An adenosarcoma arising from endometriosis should be considered in the differential diagnosis of a pelvic mass, even one appearing in rectal wall, because ectopic endometrial tissue exists everywhere.

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          Most cited references15

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          ENDOMETRIAL CARCINOMA OF THE OVARY, ARISING IN ENDOMETRIAL TISSUE IN THAT ORGAN

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            Mullerian adenosarcoma of the uterus: a clinicopathologic analysis of 100 cases with a review of the literature.

            One hundred cases of mullerian adenosarcoma of the uterus were encountered in patients 14 to 89 years of age (median, 58 years), who usually had the symptom of abnormal vaginal bleeding. An enlarged uterus and tissue protruding from the external os were the most common findings on pelvic examination. Five patients presented on multiple occasions with "recurrent polyps" that were interpreted retrospectively as adenosarcomas. Primary treatment, known in 97 cases, included some form of hysterectomy in 93 of them, and conservative resection in four cases. Gross examination of the excised uteri disclosed polypoid masses, some of which had spongy cut surfaces, usually filling the endometrial cavity; less commonly, the tumors were confined to the endocervix or the myometrium or involved more than one site. Histologic examination revealed benign or atypical neoplastic glands within a sarcomatous stroma, which typically formed periglandular cuffs of increased cellularity, intraglandular polypoid projections, or both. The sarcomatous stroma was homologous in 78% of the cases and contained heterologous elements in the remainder. The stromal mitotic rate varied from 1 to 40 mitotic figures (MFs) (mean 9) per 10 high-power fields (HPFs). Extensive areas of stromal fibrosis that focally imparted a deceptively benign appearance to the tumor were common. Myometrial invasion was present in 15 cases, but was deep in only four. Recurrent tumor developed in 23 cases at postoperative intervals of 0.5 to 9.5 years (mean 3.4); in one third of such cases, the interval was 5 years or longer. Recurrent tumor was almost always confined to the vagina, pelvis, or abdomen; hematogenous spread occurred in only two cases. The only feature associated with an increased risk for recurrence was the presence of myometrial invasion. Criteria found useful in separating mullerian adenosarcomas from mullerian adenofibromas included, alone or in combination: two or more stromal MFs per 10 HPFs, marked stromal cellularity, and significant stromal cell atypia.
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              Malignancy in endometriosis: frequency and comparison of ovarian and extraovarian types.

              One thousand consecutive cases of surgically proven endometriosis were reviewed to evaluate the frequency and types of pelvic cancers that were associated with ovarian and extraovarian endometriosis. The frequency and types of histologic abnormalities present in the eutopic endometrium when cancers were noted in endometriosis were also evaluated. In the large subset of cases for which the authors were the primary pathologists and all foci of endometriosis were recorded, the frequency of malignancy was 10.8%. In contrast, the frequency was only 3.2% in cases diagnosed by others previously in our institution. Cancers were more commonly found in ovaries when endometriosis was present in that ovary (5%) compared to when endometriosis was present at other sites (1%). Clear cell and endometrioid carcinomas were the malignancies most commonly seen in ovaries containing endometriosis, while clear cell adenocarcinoma and adenosarcoma were most commonly seen in conjunction with extraovarian endometriosis. The association of endometriosis with endometrioid and clear cell carcinoma was much stronger than that of serous and mucinous tumors (p < .01). Concurrent endometrial pathology was commonly seen in cases of malignant transformation of endometriosis (32% of cases).
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                Author and article information

                Journal
                Ann Coloproctol
                Ann Coloproctol
                AC
                Annals of Coloproctology
                The Korean Society of Coloproctology
                2287-9714
                2287-9722
                October 2014
                28 October 2014
                : 30
                : 5
                : 232-236
                Affiliations
                Department of Surgery, Catholic University of School of Medicine, Daegu, Korea.
                [1 ]Department of Surgery, Daegu Catholic University Medical Center, Daegu, Korea.
                Author notes
                Correspondence to: Daedong Kim, M.D. Department of Surgery, Daegu Catholic University Medical Center, Catholic University of Daegu School of Medicine, 33 Duryugongwon-ro 17-gil, Nam-gu, Daegu 705-718, Korea. Tel: +82-53-650-4061, Fax: +82-53-624-7185, ddkim@ 123456cu.ac.kr
                Article
                10.3393/ac.2014.30.5.232
                4213940
                25360431
                1a72c259-9df5-4c3d-9af5-c81e8821b3c3
                © 2014 The Korean Society of Coloproctology

                This is an open-access article distributed under the terms of the Creative Commons Attribution Non-Commercial License ( http://creativecommons.org/licenses/by-nc/3.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                : 25 May 2014
                : 23 July 2014
                Categories
                Case Report

                mullerian adenosarcoma,endometriosis,rectal neoplasms,adenosarcoma

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