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      Uterine tumor resembling ovarian sex cord tumor: A rare case report

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          There is no author summary for this article yet. Authors can add summaries to their articles on ScienceOpen to make them more accessible to a non-specialist audience.

          Rationale:

          A uterine tumor resembling an ovarian sex cord tumor (UTROSCT) is a clinically rare disease with an unclear origin and biological behavior.

          Patient concerns:

          We present a case of UTROSCT in a 42-year-old woman who presented with abnormally increased menstrual volume for 2 years.

          Diagnoses:

          Initially, only ultrasound examination was performed to diagnose uterine fibroids, and then the tumor was surgically removed and sent for pathological examination. The patient was ultimately diagnosed with UTROSCT mainly based on pathological immunohistochemical examination and was further diagnosed with low malignant potential for recurrence based on genetic testing.

          Interventions and Outcomes:

          The patient underwent hysterectomy and bilateral adnexectomy, and no adjuvant radiotherapy or chemotherapy was performed after the surgery. Follow-up to date has indicated that she is in good condition.

          Lessons:

          UTROSCT is a rare disease that requires pathological immunohistochemical examination to confirm the diagnosis and genetic testing when necessary so that a clear diagnosis can inform better decision-making regarding treatment measures.

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          Uterine tumors resembling ovarian sex-cord tumors. A clinicopathologic analysis of fourteen cases.

          P Clement, R E Scully (1976)
          Uterine tumors with histologic resemblances to ovarian sex-cord tumors were encountered in 14 women in the fourth to sixth decades of life, most of whom had abnormal vaginal bleeding and enlarged uteri suggesting leiomyomas. The tumors in six cases (Group I) were identical to one or another form of well-recognized endometrial stromal tumor except for focal epithelial-like differentiation resembling that seen in sex-cord tumors. These tumors were mostly poorly circumscribed large mural masses. The focal sex-cord-like differentiation was manifested by cords, trabeculae, nests and tubules of epithelial-like cells; eosinophilic bodies resembling Call-Exner bodies were seen in one case. Five of the six patients with tumors of this type have been followed for postoperative intervals of 22 months to 15 years, and pelvic recurrences have developed in three. The tumors in eight cases (Group II) differed from those in Group I by having a predominant or exclusive pattern reminiscent of an ovarian sex-cord tumor. These tumors, most of which were single, well-circumscribed, yellow to tan, mural masses 4 to 10 cm in diameter, appeared benign, with no recurrence during postoperative periods of four months to seven years. The relations of the two groups of tumors to one another and to previously described similar tumors. including so-called granulosa-cell tumors and "plexiform tumors" of the uterus, are discussed, and an origin from endometrial stromal cells is postulated.
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            Ovarian steroids, stem cells and uterine leiomyoma: therapeutic implications.

            Molly B Moravek, Ping Yin, Masanori Ono … (2015)
            Uterine leiomyoma is the most common benign tumor in women and is thought to arise from the clonal expansion of a single myometrial smooth muscle cell transformed by a cellular insult. Leiomyomas cause a variety of symptoms, including abnormal uterine bleeding, pelvic pain, bladder or bowel dysfunction, and recurrent pregnancy loss, and are the most common indication for hysterectomy in the USA. A slow rate of cell proliferation, combined with the production of copious amounts of extracellular matrix, accounts for tumor expansion. A common salient feature of leiomyomas is their responsiveness to steroid hormones, thus providing an opportunity for intervention. A comprehensive search of PUBMED was conducted to identify peer-reviewed literature published since 1980 pertinent to the roles of steroid hormones and somatic stem cells in leiomyoma, including literature on therapeutics that target steroid hormone action in leiomyoma. Reviewed articles were restricted to English language only. Studies in both animals and humans were reviewed for the manuscript. Estrogen stimulates the growth of leiomyomas, which are exposed to this hormone not only through ovarian steroidogenesis, but also through local conversion of androgens by aromatase within the tumors themselves. The primary action of estrogen, together with its receptor estrogen receptor α (ERα), is likely mediated via induction of progesterone receptor (PR) expression, thereby allowing leiomyoma responsiveness to progesterone. Progesterone has been shown to stimulate the growth of leiomyoma through a set of key genes that regulate both apoptosis and proliferation. Given these findings, aromatase inhibitors and antiprogestins have been developed for the treatment of leiomyoma, but neither treatment results in complete regression of leiomyoma, and tumors recur after treatment is stopped. Recently, distinct cell populations were discovered in leiomyomas; a small population showed stem-progenitor cell properties, and was found to be essential for ovarian steroid-dependent growth of leiomyomas. Interestingly, these stem-progenitor cells were deficient in ERα and PR and instead relied on the strikingly higher levels of these receptors in surrounding differentiated cells to mediate estrogen and progesterone action via paracrine signaling. It has been well established that estrogen and progesterone are involved in the proliferation and maintenance of uterine leiomyoma, and the majority of medical treatments currently available for leiomyoma work by inhibiting steroid hormone production or action. A pitfall of these therapeutics is that they decrease leiomyoma size, but do not completely eradicate them, and tumors tend to regrow once treatment is stopped. The recent discovery of stem cells and their paracrine interactions with more differentiated cell populations within leiomyoma has the potential to provide the missing link between developing therapeutics that temper leiomyoma growth and those that eradicate them. © The Author 2014. Published by Oxford University Press on behalf of the European Society of Human Reproduction and Embryology. All rights reserved. For Permissions, please email: journals.permissions@oup.com.
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              Uterine tumors resembling ovarian sex cord tumors are polyphenotypic neoplasms with true sex cord differentiation.

              Jaime Prat, S Carinelli, Julie Irving (2005)
              In this study, we present the clinicopathologic features and immunophenotypic characteristics of five cases of uterine tumors resembling ovarian sex cord tumors and three cases of endometrial stromal tumors with sex cord-like elements, with emphasis on immunohistochemical markers of sex cord differentiation. The mean patient age was 42 years (range 19-69 years), and vaginal bleeding was the most common clinical presentation. The tumors were usually polypoid masses arising in the uterine fundus, with a mean tumor size of 6.7 cm. Sex cord patterns in uterine tumors resembling ovarian sex cord tumors, including anastomosing cords, trabeculae, small nests, tubules, and in one case, a striking retiform architecture with Leydig-like cells, comprised from 70 to 100% of the tumor volume. All uterine tumors resembling ovarian sex cord tumors were positive for two or more markers of sex cord differentiation; all five cases showed strong immunoreactivity for calretinin, with coexpression of CD99 (four cases), Melan-A (two cases), and inhibin (two cases). Endometrial stromal tumors with sex cord-like elements were less frequently positive for markers of sex cord differentiation, with each case positive for one marker (calretinin, two cases; CD99, one case). In addition, all eight cases were frequently positive for cytokeratin, CD10, vimentin, estrogen receptor, and progesterone receptor; desmin immunoreactivity, when present, was limited to minor foci of smooth muscle. Overall, the morphologic and immunohistochemical findings in uterine tumors resembling ovarian sex cord tumors strongly support that these unusual uterine tumors are polyphenotypic neoplasms with true sex cord differentiation.
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                Author and article information

                Contributors
                Xue-Ying Wang:
                ORCID: https://orcid.org/0000-0001-8288-8805
                Jiao Chen
                Jiang-Hua Huang
                Journal
                Journal ID (nlm-ta): Medicine (Baltimore)
                Journal ID (iso-abbrev): Medicine (Baltimore)
                Journal ID (publisher-id): MD
                Title: Medicine
                Publisher: Lippincott Williams & Wilkins (Hagerstown, MD )
                ISSN (Print): 0025-7974
                ISSN (Electronic): 1536-5964
                Publication date Collection: 02 September 2022
                Publication date (Electronic): 02 September 2022
                Volume: 101
                Issue: 35
                Electronic Location Identifier: e30414
                Affiliations
                [a ] Department of Ultrasound, Affiliated Hospital of North Sichuan Medical College, Nanchong, Sichuan ProvinceChina
                [b ] Department of Obstetrics and Gynecology, Affiliated Hospital of North Sichuan Medical College, Nanchong, Sichuan ProvinceChina
                [c ] Department of Ultrasound, the Second Clinical Medical College of North Sichuan Medical College, Nanchong, Sichuan ProvinceChina.
                Author notes
                *Correspondence: Maochun Zhang, Department of Obstetrics and Gynecology, Affiliated Hospital of North Sichuan Medical College, Nanchong, Sichuan Province 637000, China (e-mail: Zmc15984818929@ 123456163.com ).
                Author information
                https://orcid.org/0000-0001-8288-8805
                Article
                Accession ID: 00104
                DOI: 10.1097/MD.0000000000030414
                PMC ID: 9439761
                PubMed ID: 36107540
                SO-VID: 22506184-d710-4023-8e50-d3c03d0d1d6a
                Copyright © Copyright © 2022 the Author(s). Published by Wolters Kluwer Health, Inc.
                License:

                This is an open access article distributed under the Creative Commons Attribution License 4.0 (CCBY), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                Date received : 22 July 2022
                Date accepted : 26 July 2022
                Categories
                Subject: Research Article
                Subject: Clinical Case Report
                Custom metadata
                OPEN-ACCESS TRUE

                Keywords: case report,immunohistochemical,treatment,uterine tumor resembling ovarian sex cord tumor
                Data availability:
                Keywords: case report, immunohistochemical, treatment, uterine tumor resembling ovarian sex cord tumor

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