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      Papillary Thyroid Cancer in a Struma Ovarii in a 17-Year-Old Nulliparous Patient: A Case Report

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          Abstract

          Introduction: Struma ovarii accounts for 2% of mature teratomas. Struma ovarii is diagnosed when thyroid tissue accounts for >50% of the teratoma. Malignant transformation is rare, occurring in <5% of struma ovarii cases. Case presentation: A 17-year-old patient was diagnosed with papillary thyroid cancer in struma ovarii. The patient exhibited menstrual disorders. Abdominal and pelvic CT revealed a 17 cm mass in the left adnexa. Laparoscopic removal of the left adnexa with enucleation of right ovarian cysts was performed. Histopathological diagnosis was a follicular variant papillary carcinoma measuring 23 mm in diameter. Immunohistochemical positive expression of CK19, TTF-1, and thyroglobulin (Tg) confirmed the diagnosis. Molecular analysis detected the BRAF K601E mutation in ovarian tumor tissues. Preoperative serum Tg concentration was >300 ng/mL, which decreased to 38.2 ng/mL after gynecological surgery with undetectable anti-Tg antibodies. The patient underwent total thyroidectomy with no cancer detected on histopathological examination. The patient was treated with I-131 and showed no recurrence 4 years after the diagnosis. Conclusions: Malignant struma ovarii is diagnosed by surgery. Because papillary carcinoma in struma ovarii is rare and there are no guidelines regarding the management of this type of cancer, therapeutic decisions should be made individually based on clinical and pathological data.

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          Most cited references39

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          BRAF mutations are associated with some histological types of papillary thyroid carcinoma.

          Mutations in the BRAF gene have recently been detected in a wide range of neoplastic lesions with a particularly high prevalence in melanoma and papillary thyroid carcinoma (PTC). The hot-spot mutation BRAF(V599E) is frequently detected in PTC (36-69%), in contrast to its absence in other benign or malignant thyroid lesions. In order to unravel whether there is any association between the occurrence of the BRAF mutation and the histological pattern of PTC, in this study a previous series of 50 PTCs was extended to 134 cases, including ten cases of PTC-related entities-hyalinizing trabecular tumour (HTT) and mucoepidermoid carcinoma (MEC). Using PCR/SSCP and sequencing, the BRAF(V599E) mutation was detected in 45 of the 124 PTCs (36%). No mutations were detected in any case of HTT and MEC. BRAF(V599E) was present in 75% of Warthin-like PTCs and 53% of conventional PTCs, whereas no BRAF(V599E) mutations were detected in any of the 32 cases of the follicular variant of PTC. BRAF(V599E) was also detected in 6 of 11 cases of the oncocytic variant of PTC that displayed a papillary or mixed follicular-papillary growth pattern and in none of the four oncocytic PTCs with a follicular growth pattern. A distinct mutation in BRAF (codon K600E) was detected in three cases of the follicular variant of PTC. This study has confirmed the high prevalence of BRAF(V599E) in PTC and has shown that the mutation is almost exclusively seen in PTC with a papillary or mixed follicular-papillary growth pattern, regardless of the cytological features of the neoplastic cells. The results support the existence of an oncocytic variant of PTC that should be separated from the oncocytic variant of follicular carcinoma and suggest that the follicular variant of PTC may be genetically different from conventional PTC. Copyright 2004 John Wiley & Sons, Ltd.
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            Clinical characteristics of struma ovarii.

            To evaluate the clinical characteristics of struma ovarii. Twenty-five cases of struma ovarii were reviewed retrospectively from June 1994 to April 2007. The presenting clinical, radiologic, and pathologic features of the patients were reviewed. The mean age of the patients in this study was 45.3 years. The majority was of premenopausal status. Sixteen patients had clinical symptoms such as low abdominal pain, palpable abdominal mass and vaginal bleeding. Although one patient had an abnormal thyroid function test, the laboratory findings normalized after operative treatment. CA-125 levels were elevated in 6 cases. Diagnosis by preoperative imaging studies were 8 dermoid cysts, while only 3 cases were diagnosed as struma ovarii. There were 4 cases of malignant struma ovarii, and no patients with recurrent disease. Struma ovarii is a rare tumor. The presented clinical, laboratory and radiological findings of patients are very diverse. The diagnosis was confirmed by pathologic findings. The treatment of benign struma ovarii is surgical resection only. The cases of malignant struma ovarii may need adjuvant treatment, but recurrence is uncommon.
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              Malignant struma ovarii: a case report and analysis of cases reported in the literature with focus on survival and I131 therapy.

              Malignant struma ovarii is a rare type of germ cell tumor that is most often diagnosed postoperatively. The natural history and optimal treatment regimen for the disease are essentially unknown due to the small numbers of published cases. A 32-year-old woman presented with pelvic pain and an ovarian mass that was ultimately treated by total abdominal hysterectomy/bilateral salpingo-oophorectomy. Postoperatively, she was diagnosed with a malignant struma ovarii. The patient was subsequently treated with thyroidectomy and I(131) ablation and is currently disease free. A Medline literature search was performed and clinical data from 23 additional cases were compiled. In this review of 24 cases, 16 patients were followed conservatively postoperatively while 8 received varied additional therapy (4 with I(131)). There were 8 recurrences and all occurred in the conservatively managed patients. I(131) for recurrent disease provided an initial complete response in 7 women. Treatment with thyroidectomy and I(131) should be considered in the first line of management for malignant struma ovarii.
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                Author and article information

                Journal
                Diagnostics (Basel)
                Diagnostics (Basel)
                diagnostics
                Diagnostics
                MDPI
                2075-4418
                15 January 2020
                January 2020
                : 10
                : 1
                : 45
                Affiliations
                [1 ]Collegium Medicum, Jan Kochanowski University, IX Wieków Kielc Av.19, 25-319 Kielce, Poland; agonet@ 123456poczta.onet.pl (A.G.); kazzerr@ 123456gmail.com (R.Ś.); aldonako@ 123456onkol.kielce.pl (A.K.)
                [2 ]Endocrinology Clinic, Holycross Cancer Center, S. Artwińskiego St. 3, 25-734 Kielce, Poland
                [3 ]Molecular Diagnostics, Holycross Cancer Center S. Artwińskiego St. 3, 25-734 Kielce, Poland; artur_kowalik@ 123456yahoo.com
                [4 ]Surgical Pathology, Holycross Cancer Center, S. Artwińskiego St. 3, 25-734 Kielce, Poland; janusz_kopczynski@ 123456yahoo.com
                Author notes
                [* ]Correspondence: danutagp@ 123456o2.pl
                [†]

                These authors contributed equally to this work.

                Author information
                https://orcid.org/0000-0003-0936-4890
                https://orcid.org/0000-0002-3718-999X
                https://orcid.org/0000-0001-5647-5898
                Article
                diagnostics-10-00045
                10.3390/diagnostics10010045
                7168171
                31952290
                e332e5f2-58f7-4fe5-8819-634b1eee6ac5
                © 2020 by the authors.

                Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license ( http://creativecommons.org/licenses/by/4.0/).

                History
                : 23 December 2019
                : 13 January 2020
                Categories
                Case Report

                malignant struma ovarii,papillary thyroid cancer,teratoma,thyroidectomy,immunohistochemistry

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