Benign Struma Research Articles

081. Radiofrequency Ablation for Retrosternal Thyroid Tumour: A Rare Case Report

Background: Retrosternal thyroid tumours, though uncommon, present complex diagnostic and therapeutic challenges due to their location behind the sternum. These tumours can cause significant symptoms, such as difficulty breathing and chest pressure, which can impact patient quality of life. Radiofrequency ablation (RFA) has emerged as a minimally invasive treatment option, offering potential benefits over conventional surgical methods for retrosternal thyroid tumor. Case: A 54-year-old male with a history of right thyroid isthmolobectomy presented with a rapidly growing left-sided neck mass, causing increasing chest pressure and shortness of breath. Imaging studies, including ultrasound and CT scans, revealed a large tumour extending retrosternally and compressing the trachea. The patient opted for RFA over surgery. A core biopsy confirmed a benign struma adenomatosa. RFA was performed using the moving shoot technique, resulting in effective tumor ablation. Follow-up imaging showed a significant reduction in tumor size and symptom relief. Conclusion: This case demonstrates that RFA is a safe and effective alternative to conventional surgery for retrosternal thyroid tumours. It provides substantial symptom relief and tumour reduction with fewer complications. RFA should be considered a valuable treatment option for patients who are not ideal candidates for surgery or who prefer less invasive approaches. Continued research and long-term studies are needed to confirm its broader efficacy.

A Rare Case of Struma Ovarii in Afghanistan

Introduction: Struma ovarii is a rare ovarian tumor characterized by containing either entirely or predominantly thyroid tissue (> 50%). It accounts for less than 1% of all ovarian tumors and approximately 3% of ovarian teratomas. The condition was first described by Von Klden in 1895. Case Presentation: In this report, we described a 50-year-old Afghan menopausal woman who referred to our hospital with a 5-month history of weight loss, anorexia, hyperthyroidism, abdominal distension and sinus tachycardia. Abdominal radiological findings revealed left adnexal large multilocular cystic lesion and the patient’s CA-125 serum level was high. Preoperatively, the clinicians suggested a diagnosis of ovarian cancer based on the symptoms, radiological findings and high level of CA-125 serum. However, postoperatively, histopathological examination revealed the lesion to be a benign struma ovarii. Conclusions: Association of struma ovarii with CA-125 and hyperthyroidism is an extremely rare case and are usually misdiagnosed preoperatively due to nonspecific clinical manifestations, and indistinguishable imaging studies, and should be kept in the differential diagnosis of all ovarian cystic lesions. An exact diagnosis is only made by postoperative histopathological examination, and complete surgical resection of the lesion is considered the optimal treatment.

Two rare cases of benign struma ovarii with malignant recurrence.

Struma ovarii is an ovarian teratoma that comprises 2-5% of all ovarian teratomas. Malignant transformation of struma ovarii occurs in less than 5% of all cases, and metastatic disease is even rarer. 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. Case 1 concerns a 37-year-old female featuring HDFCO of the right ovary with multiple metastatic foci, 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. Both patients were treated with surgery, adjunctive total thyroidectomy, and radioactive iodine (131I) therapy. Malignant transformation of struma ovarii is very rare (<5%). Diagnosis of HDFCO without extra ovarian dissemination is difficult due to the resemblance of its histological appearance with normal thyroid tissue. There is no consensus on the postoperative treatment of malignant struma ovarii (MSO). Clinical and histological features of MSO should be assessed for the postoperative treatment decisions. TSH suppression and thyroglobulin level measurements are necessary for patient follow-up.

Struma Ovarii: Single Center Experience.

Struma ovarii (SO) accounts for approximately 1% of all ovarian tumors. The objective of our study is to contribute to the treatment algorithm by presenting our clinical experience in a comprehensive case series of patients diagnosed with SO, predominantly characterized by thyroid tissue within a monodermal teratoma. Patients aged 17 years and older who underwent surgery due to ovarian masses and were histopathologically diagnosed with SO between January 2012 and January 2022 were included in the study. The patients' files were retrospectively reviewed. Demographic data, presenting complaints, radiological findings, tumor sizes, laboratory data, surgical procedures performed, pathology reports, additional treatments, and follow-up information were recorded. The median age of total 19 patients was 41.7 (17-74) years. Among them, malignant struma ovarii was present in 3 patients. In patients with histopathologically confirmed benign struma ovarii, no additional treatment was administered after tumor enucleation. In malignant cases, in addition to unilateral salpingo-oophorectomy, total thyroidectomy, radioactive iodine (RAI) ablation, and L-Thyroxine suppression were performed. No mortality occurred during the follow-up period. Although conservative treatments are considered acceptable treatment in cases of benign struma ovarii, the management of cases with malignant struma ovarii is controversial. Fertility-sparing surgery followed by postoperative adjuvant thyroidectomy and radioactive iodine ablation may be preferred for young women.

Radiation methods in the diagnosis of primary and recurrent malignant ovarian struma: A case report

We provide a rare clinical and diagnostic observation of primary and recurring malignant ovarian struma.&#x0D; Malignant struma of the right ovary was detected 2 years after surgical treatment of primary benign struma of the left ovary. Six months later, the patient was diagnosed with a disease relapse, visualized exclusively according to radioisotope research methods. In the fourth year of anticancer treatment, ultrasonography revealed recurring foci along the peritoneum. According to the ultrasound data on the pelvic peritoneum and the projection of the removed right ovary, multiple solid nodes with high blood flow were visualized. Peak systolic velocity ranged from 2 to 9 cm/s in minor lesions from 4 to 12 mm, with an RI max of 0.53. For 4 years, the patient underwent radioiodine therapy with 131I with an activity of 6.0 GBq; the patients condition during the treatment was satisfactory.

Struma ovarii with synchronous ascites and elevated CA125 level: a retrospective cohort study

Background Benign struma ovarii (SO) with synchronous ascites and elevated CA125 level is extremely rare that the incidence, clinical characteristics, and risk factors remain unclear. Methods We conducted a retrospective study of patients with SO treated in our hospital between 1980 and 2022. Logistic regression was used to identify potential risk factors for SO patients presenting with ascites and elevated CA125 levels. The receiver operating characteristic (ROC) curve was used to evaluate the predictive performance of the identified risk factors. Results A total of 21 patients with synchronous ascites and elevated CA125 levels were identified in 229 patients with SO, the crude incidence rate was 9.17%, and four patients (1.75%) had pseudo-Meigs’ syndrome. Ascites were completely involuted within 1 month postoperatively and the serum CA125 level decreased to normal between 3 d and 6 weeks after surgery. Multivariate logistic regression showed that age ≥49 years (OR 3.71, 95% CI 1.29 − 10.64, p = 0.015), tumor size ≥10.0 cm (OR 8.79, 95% CI 3.05 − 25.35, p < 0.001), and proliferative SO (OR 11.16, 95% CI 3.01 − 41.47, p < 0.001) were the independent risk factors for patients presenting ascites and elevated CA 125 level. The ROC curve revealed that the predictive performance for age and tumor size was unsatisfactory with an area under the curve (AUC) was 0.646 and 0.682, respectively. Linear regression demonstrated that the serum CA125 level has a moderate positive correlation with the volume of ascites (log2 CA125 = 0.6272*log2 ascites + 2.099, p = 0.0001, R 2 = 0.5576). Conclusions Less than one-tenth of patients with SO would present ascites and elevated CA125 levels, while age ≥49 years, tumor sizes ≥10 cm, and the presence of proliferative SO were the risk factors.

Clinical Characteristics, Treatment, and Outcomes of Peritoneal Strumosis: A Report of Three Cases and Systematic Review.

Benign struma ovarii (SO) has a probability of metastasis named "peritoneal strumosis", which is extremely rare, such that the specific clinical characteristics, treatment options, and survival outcomes remain unclear. We screened three cases of peritoneal strumosis among 229 cases of SO treated in our hospital. Case 1 was a 36-year-old woman with extensive peritoneal seedings at initial presentation. The second one was a 49-year-old with trocar site implant 11 years after laparoscopic adnexectomy. Case 3 was a 45-year-old woman who had an isolated lesion at the anterior surface of the rectum after laparoscopic ovarian cystectomy for SO 14 years ago. These three patients underwent surgery without any adjuvant treatment and remained disease-free after 30 to 68 months. A systematic review was then conducted and another 16 cases were identified. More than half (10/19, 52.6%) of the patients had previous SO-related ovarian surgery. The median interval between prior SO-related surgery and the initial presentation of peritoneal strumosis was 10.0 years; both regional and distant metastasis, even in the liver, lung, and heart, could also be affected. Surgery was the mainstay therapy (18/19, 94.7%), in which six patients (6/19, 31.7%) were treated with total thyroidectomy (TT) followed by radioiodine (RAI) therapy. Postoperative chemotherapy was only applied in one patient, and the last one only received a diagnostic biopsy without further treatment. Recurrence was noted in two patients with a median recurrence-free survival of 12 years, where surgical excision and RAI were then performed. No death occurred after a mean follow-up of 53 months, where 12 patients achieved no evidence of disease and five were alive with the disease. Peritoneal strumosis has unpredictable biological behaviors and the crude incidence is approximately 1.3% in SO. Patients with peritoneal strumosis have excellent survival outcomes, irrespective of different treatment strategies employed. Surgery with personalized RAI should be preferred and long-term close monitoring is recommended.

Struma Ovarii Masquerading as a Mucinous Ovarian Cyst

Struma ovarii is an uncommon ovarian teratoma distinguished by the unusual presence of thyroid tissue. The vast majorities are benign, and ~5% are malignant. Clinical symptoms, laboratory investigations, imaging modalities are nonspecific. Even histopathologic features may be misleading in a few cases. It is defined as a teratoma composed exclusively of or containing more than 50% thyroid tissue. Common differentials include serous and mucinous cystadenoma. Awareness of existence of such entities may help in utilization of available histologic and imunohistochemical tools for correct diagnosis. Oophorectomy is the treatment of choice for benign struma. Keywords: Struma ovarii, thyroid, teratoma, cystadenoma

Abstract 769: Genomic and transcriptomic characterization of benign and malignant struma ovarii

Abstract Background: Struma Ovarii (SO) is a rare ovarian teratoma characterized by the presence of thyroid tissue in &amp;gt;50% of the tumor. The majority of SO are benign; however, malignant transformation occurs in up to 5% of the cases. The molecular foundations of benign and malignant SO are grossly unknown. Therefore, the goal of this study was to perform the first comprehensive genomic and transcriptomic analysis of the benign and malignant SO. Material and Method: We performed whole-exome sequencing (WES) and targeted RNA-sequencing (seq) on the DNA and RNA extracted from formalin-fixed paraffin-embedded SO tumor tissue samples. WES library was prepared using Agilent’s SureSelect XT HS2 kit, with 4 samples failing the quality assessment (QA). Variants were called from GATK processed WES data and annotated using VEP (with ClinVar and COSMIC databases). The targeted RNA-Seq library was prepared using the TruSight RNA Pan-Cancer Panel kit covering 1385 cancer genes, with all samples passing QA. The clinical characteristics of the study cohort were summarized by percentages for categorical variables and medians with 25-75% interquartile ranges for continuous variables. Results: The study included 31 tissue samples - 21 benign and 10 malignant, including 6 cases of papillary thyroid cancer (PTC), 3 of follicular variant of PTC, and 1 of follicular thyroid cancer. Patients with benign SO were characterized by the median age at diagnosis of 39 years [33-54], tumor size of 3.1 cm [2.5-5.8], while the patients with malignant SO presented at age of 45 [28-54], tumor size of 6 cm [0.85-14] and metastatic disease in 30% (3/10) - 2 patients with peritoneal metastases and 1 patient with pelvic lymph node metastases. The E1A Binding Protein P300, EP300 (6/27), and Isocitrate dehydrogenase, IDH2 (5/27) were the topmost mutated genes in the SO samples. Malignant SO samples were characterized with the presence of pathogenic variants of KRAS (pQ61L and pG12V), NRAS (pQ61R), TP53 (splice site) mutations, and Nuclear Receptor Binding SET Domain Protein 1 (NSD1) fusion as the most common molecular drivers. Among benign SO samples, the most common driver was Thyroglobulin (TG) fusion with either Guanine Nucleotide binding protein (GNAS) or Rac Family Small GTPase 1 (RAC1). Differential expression analysis showed that the member of tumor suppressor family - tumor protein 63 (TP63) was the most downregulated (Log2FC = -3), while Double-sex and Mab-3 Related Transcription Factor 1 (DMRT1), implicated in the development of germ cell tumors, was the most upregulated gene in malignant SO samples over benign (Log2FC = 2.1; padj&amp;lt;0.05). Conclusions: In contrast to cancer arising from the thyroid gland, characterized by BRAFV600E as the most common mutation, malignant SO belongs to RAS-like tumors. The downregulation of tumor suppressors and upregulation of DMRT1 might be implicated in the malignant transformation of SO. Citation Format: Shilpa Thakur, Darryl Nousome, Kshama Aswath, Stephanie Cardenas, Sonam Kumari, Ruth Adewale, Maria Merino, Esra Dikoglu, Padmasree Veeraraghavan, Sriram Gubbi, Joanna Klubo-Gwiezdzinska. Genomic and transcriptomic characterization of benign and malignant struma ovarii [abstract]. In: Proceedings of the American Association for Cancer Research Annual Meeting 2022; 2022 Apr 8-13. Philadelphia (PA): AACR; Cancer Res 2022;82(12_Suppl):Abstract nr 769.

Thyrotoxic struma ovarii after thyroid ablation: a case report of laparoscopic management

Struma ovarii is a rare type of germ cell tumor and a rare cause of ectopic thyroidal hormonal production. We report a case of laparoscopic management of a thyrotoxic struma ovarii. In a 49-year-old patient, previously treated by ablative surgery and radioactive iodine for Graves' disease, a new onset of hyperthyroidism occurred unexpectedly. A whole-body scan with 131I detected increased pelvic uptake, suggesting struma ovarii. The patient was subsequently treated with laparoscopic bilateral salpingo-oophorectomy. The postoperative course was uneventful. Histopathologic examination revealed the presence of a benign struma ovarii. Three years after treatment the patient is disease free and is receiving thyroid replacement therapy with levothyroxine. &#x0D; In this case the thyrotoxic struma ovarii was diagnosed preoperatively and laparoscopic surgery proved to be a useful and reliable method for ovarian ablation.

Papillary thyroid carcinoma arising in struma ovarii

Abstract Introduction: Struma Ovarii is one type of mature teratoma in which thyroid tissue represents more than 50% of the ovarian tumor. Diagnosis is very difficult due to lack of typical symptoms. In majority of these patients, diagnosis is incidental. Patient concerns: A 65- year-old female presented to our hospital with complaints of uterine bleeding and abdominal pain. Diagnosis: Ultrasonography showed no lesions in the uterus and left ovary, while the right ovary was enlarged, comprising a dense mass of 4 cm in diameter. Computed tomography scan revealed a mass, with fatty density and absence of fluid in the Douglas space. The lymph nodes in the posterior peritoneal space showed no enlargement. Additional Magnetic Resonance Imaging confirmed the presence of the mass with clear margins, high T1 signal and T2 sequence indicating dermal cysts. Blood tumor markers were normal. However, beta-human chorionic gonadotropin levels were slightly high. Interventions: Total hysterectomy was performed along with bilateral salpingo-oophorectomy. Outcomes: The biopsy of the mass revealed features of papillary thyroid carcinoma. The lesion was found to be benign, and the patient showed no recurrence so far. Conclusion: Struma Ovarii is a rare condition, found in only 1% of all ovarian tumors; it is benign in 95% of the cases. The clinical presentation can vary in terms of symptoms and signs. Ultrasonography can reveal the differences between a benign struma ovarii and a malignant one. Complete hysterectomy and bilateral salpingo-oophorectomy are the treatment of choice. Specific guidelines for pre- and postoperative management of the patients are yet to be elucidated.

Struma ovarii and peritoneal strumosis during pregnancy

BackgroundStruma ovarii is a special type of ovarian dermoid cyst and accounts for approximately 2–3 % of all dermoid tumours. Benign struma ovarii may manifest as distant metastasis, called peritoneal strumosis, which makes it biologically similar to malignancy, and has been reported in limited cases but never discovered during pregnancy.Case presentationWe report a patient with a history of right struma ovarii cystectomy. During pregnancy, pelvic masses with non-specific clinical presentation were found again. During the caesarean section, contralateral struma ovarii with dissemination of nodules in the peritoneal cavity was found, and pathology revealed that the masses were thyroid follicle ovarian goitres.Discussion and conclusionsRecurrent benign struma ovarii with extraovarian dissemination is a rare aggressive clinical manifestation different from malignancy. It is emphasized that adequate assessment and complete resection of suspicious masses are of great importance.

From a clinical case to a review of literature: Struma ovarii associated with ascites and high serum CA125

Struma ovarii is a rare dermoid tumor of the ovary (1% of all ovarian tumors and 2,7% of ovarian teratomas), with more than 50% of the overall tissue being thyroid. Approximately one third of the cases present ascites. Pseudo-Meigs syndrome has been reported in fewer than 10 cases and 5-8% present thyroid hyperfunction. However, only 26 cases reported the association of ascites or pseudoMeigs’ syndrome, elevated cancer antigen 125 and benign struma ovarii in the literature. The authors submit a case of benign struma ovarii that presented as a pelvic mass with ascites and highly elevated CA 125 serum levels and an extensive literature survey on this topic.

Struma ovarii-A rare entity

Struma ovarii, also known as goiter of the ovary is a rare disease.1 Considering the rarity of this tumor and constrained literature in Nepal’s context, we hereby present a case of a 46-year-old female, asymptomatic, with normal blood parameters who presented with an abdominal mass. She was suspected of malignant ovarian mass on an ultrasonogram. Intraoperative frozen section examination revealed a benign tumor suggestive of dermal tumor versus struma ovarii. Postoperatively, histopathological examination was performed to assess the percentage of thyroid tissue and a diagnosis of benign struma ovarii was made which was confirmed by thyroglobulin positivity on immunohistochemistry. No features of immature teratoma and malignancy were identified.

SUN-483 A Retrospective Diagnosis of Malignant Struma Ovarii After Discovery of Pulmonary Metastases

Background: Malignant struma ovarii is a rare ovarian tumor that is histologically identical to differentiated thyroid carcinoma.1 We present a case of a struma ovarii that was recognized as being malignant only after the discovery of pulmonary metastases. Clinical Case: A 29 year old female presented to the hospital with acute right lower abdominal pain, suspicious for ovarian torsion. She underwent urgent right salpingoopherectomy and pathology demonstrated a mature cystic teratoma with benign struma ovarii. Two years later, a CT of the abdomen incidentally revealed bilateral pulmonary nodules. Review of the imaging showed that these pulmonary nodules were also present two years prior, and had since become larger. Video-assisted thoracoscopic surgery was performed and lung biopsy was positive for well-differentiated thyroid carcinoma. The patient then underwent total thyroidectomy which revealed a 0.3 x 0.3 cm infiltrative papillary thyroid cancer, follicular variant, without lymphovascular invasion. Thyroglobulin level decreased from 169 ng/mL pre-operatively to 80 ng/mL post-operatively, but then continued to be variable ranging from 56 to 252 ng/mL (1.6-50 ng/mL). Thyroglobulin antibodies remained negative.Pathology from right ovary was re-reviewed at a second institution and found to be consistent with highly differentiated thyroid carcinoma with characteristic nuclear features of papillary thyroid carcinoma.A diagnostic whole body I-131 scan showed uptake within the thyroid bed, bilateral lung nodules, left distal thigh and right mid thigh. These thigh lesions were not visualized on lower extremity ultrasound. After dosimetry was performed, the patient received radioactive iodine-131 200 mCI. Post-therapy scan six days later demonstrated uptake in the thyroid bed, bilateral lungs and bilateral thighs. About five months later, thyroglobulin level had decreased to 0.4 ng/mL with a suppressed TSH. A repeat CT chest demonstrated that the lung nodules had all decreased in size, largest from 0.5 cm to 0.3 cm. Conclusion: Careful examination of struma ovarii pathology should be performed to evaluate for malignant features since benign appearing histology can present diagnostic difficulty.2 In this case, thyroglobulin level was lower than reported in previous cases; however, sites of metastases were responsive to radioactive iodine therapy indicating well differentiated disease and a favorable prognosis.

Masked Tumor: A Rare Presentation of Struma Ovarii Disguised under Features Imitating Malignancy

ABSTRACT Aim To present a rare case of struma ovarii in a 46-year-old lady presenting with features pointing toward malignancy. Background Struma ovarii is a specialized or monodermal teratoma, which is predominantly composed of mature thyroid tissue. It accounts for less than 5% of all ovarian teratomas. It is mostly benign, and malignant changes are extremely rare. Most patients are asymptomatic or may present with mild signs and symptoms. Although the vast majority are benign, they may rarely present mimicking malignancy. Case description A 46-year-old lady presented with a large complex abdominopelvic mass with ascites and elevated CA-125 levels, thus arising suspicion of malignancy. Postoperatively, histopathology revealed a benign struma ovarii, which was further confirmed by immunohistochemistry. Conclusion Struma ovarii can imitate ovarian malignancy clinically, especially if it is complex and found in combination with ascites and an elevated CA-125 level. There is paucity of literature about such cases, and hence, it makes an accurate and timely diagnosis difficult. Clinical significance This case is one of the very few that provide a description of an atypical presentation of a rare tumor, struma ovarii. A high level of clinical suspicion may help with better preparedness before surgery and prevent extensive laparotomies in such patients. How to cite this article Chandra S, Shetty S, Mallya V, et al. Masked Tumor: A Rare Presentation of Struma Ovarii Disguised under Features Imitating Malignancy. J South Asian Feder Obst Gynae 2020;12(6):436–437.

Hyperfunctioning benign struma ovarii in a young patient with Graves’ disease: Challenges in fertility-sparing management

Introduction: Concomitant Graves’ disease with struma ovarii is a rare condition, with 18 cases reported to date. Preoperative diagnosis of struma ovarii is challenging due to nonspecific imaging features. Many patients are taken for extensive staging laparotomy in view of concerns of ovarian malignancy, however, benign struma ovarii can be treated satisfactorily by cystectomy or oophorectomy.

Pleomorphic giant cell anaplastic carcinoma arising in struma ovarii

Monodermal teratoma consist of predominantly (&gt;50%) or all component are thyroid tissue called struma ovarii. Malignant transformation of struma ovarii is very rare. Anaplastic carcinoma is the most rare type. A 52 years female with chief complaint of malaise and mass on lower abdomen. CT scan showed solid mass in ovarium that extend to upper abdominal cavity. CA-125 level is elevated. The frozen section was conducted, macroscopically showed tumor size is 15 cm, mostly composed of necrotic area, others are multicystic and solid area. Microscopic examination showed most are necrotic tissue with anaplastic cells in solid form, infiltrative between stromal with scattered pleomorphic giant cells. Benign struma ovarii component are found. The diagnosis was confirmed by examination of paraffin blocks from remain frozen section tissue, showed the same result. One area showed well differentiated carcinoma of papillary carcinoma follicular variant. Tumor was diagnosed as pleomorphic giant cell anaplastic carcinoma arising in struma ovarii based on clinical data, laboratory, radiological, macroscopic, and histopathology findings.Â

Struma ovarii: A report of three cases and literature review

Struma ovarii is a variant of mature cystic teratoma, with predominant thyroid element. Confirmatory diagnosis is by histopathology. It may mimic as ovarian malignancy in some. It may be associated with ascites in minority; even CA-125 has been found to be raised in some cases. We here report three cases – two operated for suspected ovarian malignancy in a postmenopausal female and one case operated for persistent benign ovarian cyst with raised CA-125 in a young female. In all histopathological diagnosis was a benign struma ovarii. It is difficult to diagnose these cases preoperatively as there are no specific clinical, radiological or serum markers for these rare tumors in the absence of thyroid biology abnormality. Most cases are diagnosed on histopathology. These benign tumors many times require extensive staging laparotomy for suspected ovarian malignancy in the absence of preoperative diagnosis. Prognosis is good for these tumors. Subtle radiological signs and a clinical suspicion can avoid extensive staging laprotomies in these patients.

Coexistence of Graves' disease and unilateral benign struma ovarii: A case report

Searchable abstracts of presentations at key conferences in endocrinology ISSN 1470-3947 (print) | ISSN 1479-6848 (online)