Two rare cases of benign struma ovarii with malignant recurrence

Kanella Kantreva; Stavroula Paschou; Katerina Stefanaki; Kanella Pappa; Paraskevi Kazakou; Dionisios Vrachnis; Evangelia Kavoura; Kitty Pavlakis; Eirini Giovannopoulou; Konstantinos Lathouras; Maria Alevizaki; Katerina Saltiki

doi:10.1530/endoabs.101.PS1-03-02

PS1-03-02

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Endocrine Abstracts (2024) 101 PS1-03-02 | DOI: 10.1530/endoabs.101.PS1-03-02

ETA2024 Poster Presentations Thyoid cancer case reports-1 (9 abstracts)

Two rare cases of benign struma ovarii with malignant recurrence

Kanella Kantreva ¹ , Stavroula Paschou ¹ , Katerina Stefanaki ¹ , Kanella Pappa ¹ , Paraskevi Kazakou ² , Dionisios Vrachnis ³ , Evangelia Kavoura ⁴ , Kitty Pavlakis ⁴ , Eirini Giovannopoulou ⁵ , Konstantinos Lathouras ⁵ , Maria Alevizaki ⁶ & Katerina Saltiki ⁷

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¹Alexandra Hospital, School of Medicine, National and Kapodistrian University of Athens, Endocrine Unit and Diabetes Center, Department of Clinical Therapeutics, Athens, Greece; ²General Hospital of Alexandra, Endocrine Unit and Diabetes Center, Alexandra Hospital, School of Medicine, National and Kapodistrian University of Athens, Athens, Greece, Department of Clinical Therapeutics, Athens, Greece; ³Alexandra Hospital, National and Kapodistrian University of Athens Medical School, Endocrine Unit and Diabetes Center, Department of Clinical Therapeutics, Alexandra Hospital, School of Medicine, National and Kapodistrian University of Athens, Athens, Greece, Department of Therapeutics, Athens, Greece; ⁴Iaso Hospital, Pathology Department, Athens, Greece; ⁵Iaso Hospital, Department of Gynecological Oncology, Athens, Greece; ⁶Kapodistrian University of Athens, Endocrine Unit, Departmemt Clinical Therapeutics, Medical School National Kapodistrian University, Athens, Greece, Endocrine Unit Departmemt Therapeutics, Athens, Greece; ⁷Clinical Therapeutics, Athens, Medical School Athens, University, Private, Athens, Greece

Objective: Struma ovarii is an ovarian teratoma defined by the presence of thyroid tissue, that comprises more than 50% of the overall mass. It constitutes 2-5% of ovarian teratomas and 1% of all ovarian tumors. The majority of struma ovarii cases are benign. Malignant transformation of struma ovarii is very rare (less than 5% of all cases) and metastatic disease occurs even more rarely. Most malignant struma ovarii (MSO) cases account for papillary (70%) or follicular carcinomas. Highly differentiated follicular carcinoma arising from the ovary (HDFCO) is a rare version of struma ovarii that is accompanied by extra ovarian dissemination in the peritoneal cavity (also referred to as peritoneal strumosis), and sometimes by systemic dissemination. We report two cases initially diagnosed with benign struma ovarii that presented malignant transformation, specifically highly differentiated follicular carcinoma of the ovary (HDFCO) some years after the first diagnosis.

Methods: Case 1 concerns a 37-year-old female featuring HDFCO of the right ovary with multiple metastatic focuses who was diagnosed with benign struma ovarii 14 years ago. Case 2 concerns a 26-year-old female diagnosed with HDFCO of the left ovary. This patient was initially diagnosed with benign struma ovarii 6 years ago that recurred 4 years after the diagnosis.

Results: Both patients were treated with surgery, adjunctive total thyroidectomy, and radioactive Iodine (I-131) therapy. After ablation, suppression therapy with levothyroxine was initiated and they stayed closely monitored with abdominal imaging and thyroglobulin levels. Both patients showed biochemical disease persistence and one of our patients showed additional structural persistence. Thus, a second radioiodine treatment was recommended.

Conclusions: Early diagnosis of the rare HDFCO is significant and post-thyroidectomy radioiodine therapy is mandatory in the majority of cases. TSH suppression and thyroglobulin levels measurements are necessary for the patient’s follow-up in adjunction with abdominal imaging. We emphasize the necessary cooperation of medical specialties in terms of multidisciplinary tumor boards of specialized referral centers for these cases.