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      Primary malignant melanoma arising from ruptured ovarian mature cystic teratoma with elevated serum CA 19–9: a case report and review of literature

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          Abstract

          Background

          Ovarian mature cystic teratomas comprise tissues derived from all three germ layers. In rare incidences, malignant tumors may arise from ovarian mature cystic teratoma, which occurs in 0.2–1.8% of cases. A variety of tumors can arise within mature cystic teratoma, among which malignant melanoma is exceedingly rare.

          Case presentation

          A 42-year-old woman presented with abdominal pain. Transvaginal ultrasonography showed mixed echogenic cystic masses in both ovaries. Her serum cancer antigen (CA19–9) level was elevated at 29,770 U/ml. Surgical excision was performed. Histologic examination showed infiltrating nests of pleomorphic cells with prominent nucleoli and black pigments in the background of a mature cystic teratoma. These pleomorphic cells showed strong immunoreactivity for Melan-A and HMB-45. The patient was re-evaluated and the possibility of a melanoma at any other site was ruled out. Based on these findings, we concluded that the malignant melanoma originated from the ovarian mature cystic teratoma.

          Conclusion

          We report a rare case of primary malignant melanoma derived from an ovarian mature cystic teratoma.

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          Most cited references14

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          CA19-9 elevation in ovarian mature cystic teratoma: Discrimination from ovarian cancer – CA19-9 level in teratoma

          Background We aimed to identify clinical characteristics of ovarian mature cystic teratoma (MCT) in association with CA19-9 elevation, and to determine if CA19-9 is a useful marker in discrimination of MCT from ovarian cancer (OC). Material/Methods Medical records of 322 women with pathologically-confirmed MCT or OC (stage 1 or 2) were reviewed retrospectively. The relationships between the characteristics of MCT (mean diameter, bilaterality, and pathologic components) and elevated CA19-9 were evaluated. Tumor markers in MCT were compared to those in OC. Results MCTs with CA19-9 elevation were correlated with a larger diameter (8.53±3.84 cm vs. 6.95±3.97 cm, p=0.002) and presence of fat component (67.1% vs. 32.9%, p<0.001), compared to those with normal CA 19-9. Although the incidence of CA19-9 elevation was not different between patients with MCT and OC (p=0.700), the mean value of CA19-9 was higher in those with OC (114.66±20.66 U/mL vs. 508.58±261.63 U/mL, p=0.013). In addition, simultaneous elevation of CA125 and CA19-9 was associated with a higher probability of malignant neoplasm (p<0.001; odds ratio: 23.7; 95% confidence interval: 8.863–63.576) than single elevation of CA 19-9. Conclusions CA19-9 could be an important tool in the diagnosis of ovarian mature cystic teratoma. CA19-9, in combination with CA125, might be a useful marker in discrimination of MCT from cancer.
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            Primary malignant melanoma arising in a cystic teratoma

            The malignant transformation of a cystic teratoma is a rare event, occurring in about 0.2% to 1.8%. Primary malignant melanoma arising from the ovary is extremely rare. A primary melanoma in an ovarian cystic teratoma originates most frequently at the dermoepidermal junction, similar to a cutaneous melanoma. Though there are no standard adjuvant regimens for the treatment of primary malignant melanoma of the ovary. We present another case report of malignant melanoma developing in cystic teratoma.
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              Malignant melanoma metastatic to the ovary. A clinicopathologic analysis of 20 cases.

              Twenty cases of malignant melanoma metastatic to the ovary are reported. The patients, whose ages ranged from 21 to 60 (average 37.5) years, typically presented because of abdominal swelling or pain. Approximately 50% of the patients also had metastatic tumor outside the ovary, usually within the pelvis and upper abdomen, at the time of presentation. Twelve patients were known to have had a cutaneous malignant melanoma 1 month to 13 years before their ovarian tumors were discovered, and pigmented lesions had been removed previously from three other patients. Most patients are known to have died within a few years of discovery of their ovarian tumors but two were alive without evidence of disease 5 and 8 years later. The ovarian tumors, which were bilateral in nine cases, ranged up to 20 (average 10.5 cm) in greatest dimension. Six of them were either entirely black or had discernible black or brown foci. The most common microscopic appearance was that of large cells with abundant eosinophilic cytoplasm growing in nodular aggregates or diffusely. Occasional tumors were characterized by small cells with scanty cytoplasm, and in five tumors spindle cells were present. Another pattern was growth in the form of discrete rounded aggregates having a nevoid appearance. Eight tumors contained folliclelike spaces. Major cytologic features of the tumors included prominent nucleoli in 13, cytoplasmic pseudoinclusions in many nuclei in five, and intracytoplasmic melanin pigment in nine cases. In the 10 cases studied immunohistochemically, most of the tumor cells were strongly positive for S-100 protein and fewer cells were positive for HMB-45 in the seven tumors that were stained for this antigen. Melanosomes were identified in the three tumors examined ultrastructurally. These neoplasms often were difficult to differentiate from many other types of tumors, including juvenile granulosa cell tumor and small cell carcinoma, because of the presence of folliclelike spaces.
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                Author and article information

                Contributors
                cwk-ksk@gmail.com
                atrai83@naver.com
                obgyn2001@jbnu.ac.kr
                kyjang@jbnu.ac.kr
                +82-63-270-4691 , kmkim@jbnu.ac.kr
                Journal
                BMC Womens Health
                BMC Womens Health
                BMC Women's Health
                BioMed Central (London )
                1472-6874
                27 November 2019
                27 November 2019
                2019
                : 19
                : 149
                Affiliations
                [1 ]ISNI 0000 0004 0470 4320, GRID grid.411545.0, Department of Obstetrics and Gynecology, , Chonbuk National University Medical School, Research Institute of Clinical Medicine of Chonbuk National University-Biomedical Research Institute of Chonbuk National University Hospital, ; Jeonju, Republic of Korea
                [2 ]ISNI 0000 0004 0470 4320, GRID grid.411545.0, Department of Pathology, , Chonbuk National University Medical School, Research Institute of Clinical Medicine of Chonbuk National University-Biomedical Research Institute of Chonbuk National University Hospital, ; 567 Baekje-daero, Dukjin-gu, Jeonju, 54896 Republic of Korea
                [3 ]ISNI 0000 0004 0470 4320, GRID grid.411545.0, Research Institute for Endocrine Sciences, , Chonbuk National University Medical School, ; Jeonju, Republic of Korea
                Author information
                http://orcid.org/0000-0001-7074-7183
                Article
                853
                10.1186/s12905-019-0853-8
                6882322
                31775731
                ce103f7a-929e-4b7c-b645-a1aa264e44cb
                © The Author(s). 2019

                Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License ( http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver ( http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

                History
                : 14 December 2018
                : 15 November 2019
                Funding
                Funded by: FundRef http://dx.doi.org/10.13039/501100002864, Chonbuk National University Hospital;
                Categories
                Case Report
                Custom metadata
                © The Author(s) 2019

                Obstetrics & Gynecology
                ovary,mature cystic teratoma,melanoma,primary
                Obstetrics & Gynecology
                ovary, mature cystic teratoma, melanoma, primary

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