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      An unusual case of struma ovarii

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          Abstract

          Introduction

          Struma ovarii is a teratoma of the ovaries predominantly composed of thyroid tissue. Hyperthyroidism associated with struma ovarii is rare, occurring in approximately 8% of cases. Due to the rarity of struma ovarii, available data are limited to case reports and small case series.

          Methods and results

          We report on a 61-year-old female patient with known Hashimoto’s thyroiditis on levothyroxine replacement therapy for years with transition to clinical and biochemical hyperthyroidism despite antithyroid medication with carbimazole (10 mg/day), new diagnosis of urothelial carcinoma and an adnexal mass suspicious of ovarian cancer. The patient underwent resection of the adnexal mass and histopathology revealed a mature teratoma predominantly composed of thyroid tissue showing high levels of sodium iodide symporter protein expression. Following struma ovarii resection and disappearance of autonomous production of thyroid hormones, the patient developed hypothyroidism with severely decreased thyroid hormone levels fT4 and fT3 (fT4 0.4 ng/dL, reference interval 0.9–1.7 and fT3 < 1.0 pg/mL, reference interval 2.0–4.4). This has previously been masked by continued thyroid-stimulating hormone suppression due to long-term hyperthyroidism pre-surgery indicating secondary hypothyroidism, in addition to primary hypothyroidism based on the known co-existing chronic lymphocytic thyroiditis of the orthotopic thyroid gland. Levothyroxine administration was started immediately restoring euthyroidism.

          Conclusion

          This case illustrates possible diagnostic pitfalls in a patient with two concurrent causes of abnormal thyroid function.

          Learning points
          • Struma ovarii is an ovarian tumor containing either entirely or predominantly thyroid tissue and accounts for approximately 5% of all ovarian teratomas.

          • In rare cases, both benign and malignant struma ovarii can secrete thyroid hormones, causing clinical and biochemical features of hyperthyroidism.

          • Biochemical features of patients with struma ovarii and hyperthyroidism are similar to those of patients with primary hyperthyroidism. In such cases, thyroid scintigraphy should reveal low or absent radioiodine uptake in the thyroid gland, but the presence of radioiodine uptake in the pelvis in a whole body radioiodine scintigraphy.

          • We give advice on possible diagnostic pitfalls in a case with two simultaneous causes of abnormal thyroid function due to the co-existence of struma ovarii.

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          Most cited references14

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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: two case reports and a review of the literature.

            Struma ovarii consists of thyroid tissue derived from germ cells in a mature teratoma. Malignant transformation is very rare, with clinically evident metastatic disease reported in approximately 20 cases. The rarity of this disease renders evaluation of treatment modalities difficult. There is evidence that these tumors behave like their thyroid counterparts, and cytoreductive surgery followed by ablation with radioactive iodine has been advocated. We report the diagnosis and treatment of 2 patients with metastatic malignant struma ovarii treated with a combination of surgery and radiation therapy. Copyright 1999 Academic Press.
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              • Article: not found

              Expression of benign and malignant thyroid tissue in ovarian teratomas and the importance of multimodal management as illustrated by a BRAF-positive follicular variant of papillary thyroid cancer.

              The most common type of ovarian germ cell tumor is the teratoma. Thyroid tissue, both benign and malignant, may be a component of an ovarian teratoma. Here we review this topic and illustrate major features by presenting multimodal management of a patient with BRAF-positive disseminated follicular thyroid cancer arising in an ovarian teratoma.
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                Author and article information

                Journal
                Endocrinol Diabetes Metab Case Rep
                Endocrinol Diabetes Metab Case Rep
                EDM
                Endocrinology, Diabetes & Metabolism Case Reports
                Bioscientifica Ltd (Bristol )
                2052-0573
                05 March 2021
                2021
                : 2021
                : 20-0142
                Affiliations
                [1 ]Department of Internal Medicine IV University Hospital of Munich, LMU Munich, Munich, Germany
                [2 ]Department of Urology University Hospital of Munich, LMU Munich, Munich, Germany
                [3 ]Department of Pathology University Hospital of Munich, LMU Munich, Munich, Germany
                [4 ]Division of Endocrinology Diabetes, Metabolism and Nutrition, Mayo Clinic Rochester, Minnesota, USA
                Author notes
                Correspondence should be addressed to C Spitzweg; Email: christine.spitzweg@ 123456med.uni-muenchen.de
                Article
                EDM200142
                10.1530/EDM-20-0142
                7983514
                33682680
                9eda61dd-83b2-4344-8990-a858dd8f56c5
                © 2021 The authors

                This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License..

                History
                : 31 December 2020
                : 09 February 2021
                Categories
                Adult
                Female
                White
                Germany
                Thyroid
                Thyroid
                Levothyroxine
                Gynaecology
                Error in Diagnosis/Pitfalls and Caveats
                Error in Diagnosis/Pitfalls and Caveats

                adult,female,white,germany,thyroid,levothyroxine,gynaecology,error in diagnosis/pitfalls and caveats,march,2021

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