Metastatic Malignant Struma Ovarii Research Articles

RET/PTC rearrangement in papillary thyroid carcinoma arising in malignant struma ovarii with abdominal wall metastasis and cervical thyroid gland: a case report and review of the literature

BackgroundStruma ovarii refers to rare mature cystic teratomas containing at least 50% of thyroid tissue, and malignant transformation is known to be even rarer. The synchronous development of malignant struma ovarii and cervical thyroid carcinoma are also scarce and poorly understood due to limited data about molecular features. Here, we present the first report of RET/PTC 1 rearrangement in synchronous metastatic malignant struma ovarii to the abdominal wall and cervical thyroid cancer.Case presentationWe described a 47-year-old multigravida woman with bilateral adnexal and lower abdominal wall masses detected during the evaluation of abnormal uterine bleeding. The patient underwent a hysterectomy, bilateral salpingo-oophorectomy, and surgical removal of abdominal wall mass. Then, the pathological evaluation revealed papillary thyroid carcinoma (PTC) within struma ovarii and metastatic PTC in the abdominal wall fibro adipose tissue. Further, cervical thyroid gland physical examination and ultrasound illustrated a nodule within the left lobe. Subsequently, a total thyroidectomy was performed, and a histological examination revealed PTC. Furthermore, all affected tissue, i.e., struma ovarii, abdominal wall metastasis, and cervical thyroid gland tested for BRAF and RAS mutations and RET/PTC 1 rearrangement. RET/PTC 1 rearrangement was identified among all three different sites. Finally, after six years of follow-up, the patient had no evidence of recurrence or distant metastasis.ConclusionsIn light of these findings, malignant struma ovarii might yield a clue to cervical thyroid carcinoma, and the molecular analysis could provide valuable information for understanding the underlying mechanism, tumor clinicopathological behaviors, and prognosis.

High dose radioiodine therapy preceded by fertility preservation surgery in metastatic malignant struma ovarii: A tale of endurance and prudent management

Struma ovarii is a type of mature ovarian teratoma which accounts for roughly 0.5%–1% (1) of all ovarian tumours and approximately 3% of all ovarian teratomas (2). To be classified as struma ovarii, more than 50% of the tumour must be comprised of thyroid tissue (3). Malignant struma ovarii being rare, no proper guidelines exists regarding its surgical approach or postoperative management. Metastatic malignant struma ovarii, in addition to radical surgery for ovarian mass will require total thyroidectomy to facilitate high dose radioiodine therapy. Here we present the case of a newly married, nulliparous, young lady in her third decade who was diagnosed with malignant struma ovarii with metastatic deposits in fallopian tube and extensive deposits in mesentery and peritoneum., She underwent cryopreservation of embryos followed by bilateral salphingo-oopherectomy + omentectomy + stripping of peritoneum over bladder, abdominal side walls, pelvic peritoneum + appendectomy with preservation of uterus. Total thyroidectomy was done simultaneously. Subsequently she underwent high dose radioiodine therapy. Complete ablation of the residual metastatic deposits were achieved by one sitting of therapy.

FIGO Stage IV and Age Over 55 Years as Prognostic Predicators in Patients With Metastatic Malignant Struma Ovarii.

Introduction: Metastatic malignant struma ovarii (MSO) is an extremely rare disease that lacks treatment consensus and accurate prognosis. The objective of this study was to present the clinical, pathological, and treatment characteristics of metastatic MSO, while also investigate the overall survival (OS) rate and factors affecting prognosis in this population.Materials and Methods: A total of 79 cases of metastatic MSO were reviewed, including four cases of metastatic MSO from our hospital and 75 cases selected from the literature. Logistic regression was used to identify potential factors affecting disease free survival (DFS). The Kaplan-Meier method and log-rank test were used to determine OS; further Cox regression was used to evaluate factors affecting OS.Results: The mean age of all the patients at diagnosis was 43.8 years. The most common metastatic sites were peritoneum, bone, liver, omentum and lung in descending order. Only two patients (2.6%) coexisted with local primary thyroid cancer. Follicular carcinoma (41.8%) as the most prevalent subtype, followed by papillary carcinoma, follicular variant of papillary carcinoma, and mixed follicular-papillary carcinoma. 36.7% of the patients received conservative surgery, 43.0% of them underwent aggressive surgery, and 15.2% of them did not receive any surgery. 74.7% of patients who received adjuvant therapy underwent radioiodine therapy (RAI). Logistics regression revealed that FIGO stage IV was the only prognostic factor in predicting DFS (P = 0.002; Odds Ratio [OR] 5.333; 95% confidence interval [CI]: 1.839–15.471). Only seven deaths occurred. The OS rates at 5, 10, 15 years were 89.3, 82.4, 65.9%, respectively. Multivariate analysis showed age over 55 years (P = 0.006; OR 9.362; 95%CI: 1.895–46.246) was the only risk factor for OS.Conclusion: Patients with metastatic MSO have an excellent disease-specific OS rate, FIGO stage IV and age over 55 years were two factors affecting disease prognosis. Conservative surgery with residual ablation by RAI after total thyroidectomy should be preferred since the benefits of aggressive surgery are uncertain.

Graves’ disease in assocation with metastatic malignant struma ovarii: A descriptive case

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

MON-440 Unusual Case of Metastatic Struma Ovarii Diagnosed at the Time of Hysterectomy

Introduction: Struma ovarii is rare ovarian teratoma composed of more than 50% mature thyroid tissue, which can rarely transform to malignancy. There are fewer than 200 cases reported and no established treatment approach. We present a case of metastatic malignant struma ovarii. Case Presentation: A 41 year old female was diagnosed with metastatic papillary thyroid carcinoma (PTC) after it was found on uterine histopathology. History was notable for remote left ovarian cystectomy of a mature teratoma with prominent thyroid component. She had no personal or family history of craniocervical radiation or thyroid cancer. Eight years later, she had a total hysterectomy for menorrhagia. Pathology showed subcentimeter serosal deposits of follicular variant PTC, staining positive for thyroglobulin, CK19, and HBME1. Endometrial and cervical pathology were normal; the myometrium had many leiomyomata. Post-hysterectomy TSH was 2.3 (0.34 - 5.66 µIU/mL), and thyroglobulin 44.3 (<=33.0 ng/mL). Thyroid ultrasound showed a 7 mm isoechoic nodule without lymphadenopathy. Whole body PET/CT showed multiple hypermetabolic masses in the pelvic peritoneum and liver. The right ovary had many cystic lesions and was enlarged to 5.3 x 4.5 cm. She underwent partial hepatectomy, oophorectomy, salpingectomy, and omental resection with no residual disease. Pathology showed follicular variant PTC in both ovaries, peritoneum, colonic mesentery, and omentum. Thyroglobulin fell to 6.9 ng/mL 3 weeks later. A 0.2 cm focus of follicular variant PTC with capsular invasion was found on pathology after total thyroidectomy. She underwent radioiodine ablation with 150 mCi. Postablative scan showed residual activity in the thyroid bed and right hemipelvis, but no new foci of activity. CT abdomen showed resolution of perihepatic lesions and thyroglobulin declined further to 0.5 ng/mL. Discussion: A low risk of recurrence (7.5%) has been reported in patients with malignant struma ovarii, with survival rates of 96.7% at 5 years and 84.9% at 20 years, despite a variety of surgical and adjuvant management strategies. Unilateral cystectomy, unilateral salpingo-oophorectomy, or total abdominal hysterectomy and bilateral salpingo-oophorectomy may be sufficient for patients with well differentiated thyroid cancer arising in struma ovarii without metastases. Thyroid ultrasound should be performed to exclude primary thyroid malignancy. Patients with distant metastases may benefit from aggressive treatment including resection of gross abdominal and pelvic disease and total thyroidectomy to facilitate radioactive iodine ablation and surveillance for recurrence. Conclusion: Due to its rarity, there is no consensus on optimal treatment of malignant struma ovarii. More research in this field is warranted.

Rare Clinical Entity: Metastatic malignant struma ovarii diagnosed during pregnancy \u2013 Lessons for management

BackgroundMalignant struma ovarii is an ovarian teratoma containing at least 50% thyroid tissue which has the potential to metastasize and produce thyroid hormone. Given its rarity, management strategies are not well-established. We report a case of metastatic malignant struma ovarii discovered during pregnancy with lessons for evaluation and management.Case presentationA 30-year-old woman who was two months pregnant was discovered to have struma ovarii with over half of the struma comprised of papillary thyroid cancer. Following tumor resection, delivery, and thyroidectomy, she underwent evaluation with stimulated thyroglobulin testing and diagnostic staging sodium iodide-131 scan (I-131), which revealed the presence of skeletal metastases. Following administration of 320 mCi I-131, post-therapy scan also showed miliary pulmonary metastases with improved ability to localize the bony and pulmonary metastases with concurrent SPECT/CT imaging. A second dosimetry-guided I-131 therapy resulted in complete resolution of pulmonary metastases; however, small foci of residual bone disease persisted. Post-therapy scans demonstrated additional findings not shown on diagnostic I-131 scans obtained prior to both her initial and second I-131 therapy.ConclusionsSPECT/CT provides accurate anatomic correlation and localization of metastatic foci and can serve as a baseline study to assess interval response to treatment. Post-therapy scans should always be obtained when I-131 treatment is administered, as additional findings may be revealed versus low dose I-131 activity diagnostic scans. This patient had a high metastatic burden that would not have been discovered in a timely fashion with the conservative approach advocated by others. Thyroidectomy followed by a diagnostic staging radioiodine scan and a stimulated thyroglobulin level should be considered in patients with malignant struma ovarii for guiding therapeutic I-131 administration as metastatic risk is difficult to predict based on histopathologic examination.

Long-term survival in metastatic malignant struma ovarii treated with oral chemotherapy: A case report.

Malignant struma ovarii is a rare type of ovarian tumor. Metastasis from malignant struma ovarii is rare and has only been documented in 5–6% of cases. The natural history and optimal treatment strategy for malignant struma ovarii remains controversial due to its rarity. The current report presents the case of a 45-year-old female who presented with a tumor of the rib bone. Following resection, the postoperative diagnosis was a metastasizing thyroid carcinoma. No abnormality was detected in the thyroid gland, however, computed tomography revealed a tumor in the left ovary. The patient underwent a left salpingo-oophorectomy and a wedge resection of the right ovary. The postoperative diagnosis was determined as a mature cystic teratoma with malignant struma ovarii (thyroid type, follicular carcinoma) of the left ovary and mature cystic teratoma of the right ovary. Four years subsequent to the initial diagnosis, multiple lung metastases were detected. The following chemotherapies were administered sequentially and intermittently: Tegafur-uracil, paclitaxel/carboplatin and oral etoposide. During this period, the metastatic lesions extended into the bone and progressed slowly. The patient continues to survive with the disease and 24 years have passed since the initial diagnosis, 20 years following the diagnosis of multiple lung metastates. The present report describes a rare case of malignant struma ovarii in which surgical resection and pathological examination of a metastatic rib tumor resulted in the identification of the primary ovarian lesion. The clinical behavior of malignant struma ovarii does not necessarily indicate a histological malignancy, therefore, prediction of future metastasis is difficult and the optimal treatment strategy for malignant struma ovarii is controversial. The present case indicates that the long-term use of oral anticancer agents may facilitate the maintenance of tumor dormancy.

131I Therapy Using rhTSH Stimulation and Dosimetry in Metastatic Malignant Struma Ovarii

A 38-year-old woman with metastatic malignant struma ovarii, including massive liver metastases and retroperitoneal lymphadenopathy, underwent ovarian resection and retroperitoneal lymph nodes excision, partial hepatectomy, and radiofrequency ablation for liver metastases. She underwent thyroidectomy and received three I treatments using recombinant human thyrotropin stimulation and radioiodine dosimetry. posttherapy I imaging, anatomic images, and thyroglobulin levels showed significant diminution in the tumor burdens and remarkable decline in thyroglobulin levels. This case provided valuable information on recombinant human thyrotropin-assisted I ablation in conjunction with dosimetry in an unusual presentation of iodine-avid malignant struma ovarii with bulky metastases.

Metastatic Struma Ovarii: Late Presentation, Unusual Features and Multiple Radioactive Iodine Treatments

Metastatic malignant struma ovarii is rare and there is a lack of agreement on the criteria of diagnosis and the lines of management. Here we describe a patient with struma ovarii that was initially diagnosed as benign and presented 10 years later with distant metastases. At this time, a pathological review of the initial lesion found that it contained invasive well-differentiated follicular carcinoma. The case was associated with a number of unusual features and challenging management issues, such as a delayed diagnosis of recurrence, functioning metastases with treatment consequences, tumour lysis-induced thyrotoxicosis and cerebrospinal fluid rhinorrhea. The diagnosis and management of struma ovarii should be led by an expert multidisciplinary team. Radioactive iodine should be considered in the management of metastatic disease.

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.