Malignant Struma Ovarii (Papillary Carcinoma) with Hyperthyroidism: A Case Report and Literature Review.
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SUBMITTER: Yang B
PROVIDER: S-EPMC10294126 | biostudies-literature | 2023 Jan-Dec
REPOSITORIES: biostudies-literature
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Malignant Struma Ovarii (Papillary Carcinoma) with Hyperthyroidism: A Case Report and Literature Review.
Yang Bo B Zhong Lijuan L Peng Lilin L Huang Ting T Zhu Di D Lu Yuanzhi Y
Case reports in oncology 20230101 1
Malignant struma ovarii (MSO) is an extremely rare monodermal ovarian teratoma. Preoperative diagnosis and intraoperative freezing diagnosis are exceedingly difficult due to the rarity of the disease and its clinically noncharacteristic manifestations with less than 200 reports in the current literature. In this paper, a case of MSO (papillary carcinoma) with hyperthyroidism was discussed in terms of its epidemiology, clinicopathology, molecular features, treatment, and prognosis. ...[more]
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Project description:Background: SO (Struma ovarii) is a rare form of ovarian teratoma which originates from ovarian dermoid cysts. Due to the rarity of this disease, relevant studies might not be sufficiently documented, especially cases with hyperthyroidism and multiple metastases. Case Presentation: A 40-year-old female patient was admitted to our hospital due to management of early pregnancy along with a recurrent abdominal and pelvic mass. Contrast-enhanced CT images showed an irregular mass (10.7 × 8.6 × 12.8 cm) located in the right side from the hypogastrium to the pelvic cavity and another mass (3.8 × 3.7 cm) in the liver. Laboratory examination showed that CA125 (Carbohydrate Antigen-125) was 118.10 U/mL, Tg (thyroglobulin) was >300 ng/ml, FT4 (free thyroxine) was 22.11 pmol/L, and TSH (thyroid-stimulating hormone) was <0.004 mIU/L. She subsequently underwent liver mass dissection, omentectomy, tumor dissection, peritoneal nodule resection, as well as rectal anterior wall nodule resection. The patient was diagnosed with malignant SO (papillary type) along with multiple metastases. Also, we conducted a literature review based on 290 SO cases from 257 articles. Conclusion: This study showed that malignant SO might be prone to relapse and metastasize (a metastatic rate of 52.94%) and therefore aggressive management might need to be recommended for malignant SO. Also, laparotomy might need to be recommended for large tumors that cannot be resected by laparoscopic surgery since these tumors might be prone to rupture and thus produce peritoneal implants. Furthermore, Graves' disease might need to be considered in the differential diagnosis.
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Project description:Malignant struma ovarii (MSO) with synchronous primary thyroid cancer in the neck is extremely rare and lacks a treatment consensus. A 44-year-old woman presenting with a left ovarian cyst was admitted to Peking Union Medical College Hospital (Beijing, China) Ultrasonography showed a 6 cm solid-cystic left ovarian mass with plentiful blood signals. Other notable findings were an elevated CA125 level and a suspected malignant thyroid nodule. A unilateral salpingo-oophorectomy (USO) was conducted, and the surgical pathology was papillary thyroid cancer (PTC) arising in a struma ovarii. The patient underwent a total thyroidectomy and cervical lymph node dissection, and the pathology of the right lobe nodule was follicular-variant PTC without capsule invasion or lymph node metastasis (5 mm; pT1aN0M0). No further adjuvant therapy was administered. The serum thyroglobulin value was normal before surgery and was undetectable after thyroidectomy. During regular follow-up examinations over 4 years, the patient remained well with no evidence of disease (NED). In a literature review, another 13 cases of MSO coexisting with cervical thyroid cancer that had reported outcomes were found. The MSO was confined to the ovary in all cases. A total of nine patients received radioiodine therapy (RAI) treatment after total thyroidectomy. Two patients relapsed and were successfully cured with RAI after the initial surgery Only one patient died due to another disease, while 11 patients showed NED and the remaining patient was alive with the disease after a median follow-up time of 2 years. This data suggests that USO with personalized RAI may be a preferred option for MSO confined to the ovary plus synchronous primary thyroid cancer due to the conferred satisfactory prognosis.
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Project description:Malignant struma ovarii presenting with follicular carcinoma is extremely rare, and its mechanism of tumorigenesis remains unknown. Here, we present a case of malignant struma ovarii with peritoneal dissemination of follicular carcinoma, for which a molecular analysis for major oncogenic gene alterations in follicular thyroid carcinoma was performed. A 39-year-old nulliparous woman was referred with a diagnosis of highly differentiated follicular carcinoma of ovarian origin. Primary thyroid cancer was not diagnosed, and she had a normal thyroid function. 123I scintigraphy revealed multiple peritoneal dissemination that was surgically resected. Histologically, the tumor consisted of numerous follicles without nuclear features of papillary thyroid carcinoma. Tumor samples were investigated for 50 cancer-related genes, including RAS, BRAF, and p53, and PPARg-PAX8 gene fusion by targeted DNA sequencing and fluorescence in situ hybridization, respectively. No major oncogenic gene alterations were detected. These negative findings suggest a different mechanism of tumorigenesis from that of adult-type follicular thyroid carcinoma.
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Project description:The monodermal teratoma struma ovarii is a rare ovarian tumour; however, struma ovarii presenting with pseudo-Meigs' syndrome and raised cancer antigen (CA)-125 levels is even rarer. In elderly patients, this presentation can potentially lead to a misdiagnosis of a malignant ovarian carcinoma, resulting in unnecessary extensive surgery. We report a 55-year-old female who presented to the Lok Nayak Jai Prakash Narayan Hospital, New Delhi, India, in 2016 with progressive abdominal discomfort, fatigue and abdominal distention. Clinical and radiological features were indicative of a malignant ovarian tumour and ascites. Serum CA-125 levels were elevated at 258 U/mL. A left-sided salpingo-oophorectomy was performed, after which the serum CA-125 levels normalised. There was no evidence of recurrence at a six-month follow-up. A frozen section procedure confirmed the diagnosis of a struma ovarii. This rare condition should be considered as a differential diagnosis in patients presenting with ovarian masses, ascites and raised CA-125 levels.
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Project description:IntroductionStruma ovarii (SO) is a rare ovarian mature teratoma which is composed of 50% or more thyroid tissues. Malignant transformation is reported to occur in less than 5% of all cases, and it leads to 5-6% metastatic disease. The most common type is a papillary carcinoma. However, follicular carcinoma of arising from SO is extremely rare.Case presentationA 23-year-old nullipara complained of right lower abdominal pain. CT scan revealed a large complex cystic and solid mass in pelvic cavity with moderate amount ascites with peritoneal thickening and omental infiltration. We performed a right salpingo-oophorectomy and surgical staging.Clinical discussionIt was diagnosed with follicular carcinoma, stage IA. After the multidisciplinary team meeting, we decided not to conduct additional treatment, including total thyroidectomy followed by radioiodine treatment and T4 therapy because thyroid tests were normal findings.ConclusionThere is still no established treatment consensus for follicular carcinoma of SO patients. The choice for a conservative or radical approach depends on the doctor. However, conservative surgery could be considered for low risk, early stage patients who have childbearing potential.
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Project description:BackgroundPulmonary papillary adenoma is a rare benign tumor in the periphery of the lung. We report a 66-year-old female patient with a tumor in the lower lobe of the right lung and present the clinicopathological features and review the literature.Case presentationA tumor in the lower lobe of the right lung was found incidentally on chest X-ray during the physical examination of the patient, and the patient occasionally had a dry cough that was not treated. The tumor was clearly demarcated and lobulated on CT scan. After 2 years of follow-up, the boundary of the tumor was still clear, with more lobulations and the enhanced scan showed uniform enhancement. Grossly, the tumor had a granular cut surface and was easy to fall off, which was helpful for the diagnosis of papillary adenoma during intraoperative frozen examination. Under the microscope, most areas of the tumor had the typical morphological structure of papillary adenoma. However, the tumor locally protruded into the surrounding lung tissue, accompanied by crowded cells and high cell proliferation index. It was suggested that this case of papillary adenoma had malignant potential and needed active intervention and treatment.ConclusionPulmonary papillary adenoma is a rare epithelial tumor with malignant potential. Surgical treatment should be performed as soon as possible after diagnosis to prevent malignant transformation.
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Project description:Background: Malignant struma ovarii (MSO) is an extremely rare ovarian malignant tumor and there is limited data on the survival outcomes and prognostic predictors of MSO. The objectives of this study were to investigate the disease-free survival (DFS), overall survival (OS), and disease-specific survival (DSS) rates of patients with MSO, and also evaluate the prognostic factors in this population. Methods: A retrospective study was conducted and 194 cases of MSO were selected. DFS was assessed by the logistic regression, OS by the Kaplan-Meier method, and DSS was evaluated by the Cox regression. Results: The median age of these patients was 46.0 years; 142 cases (73.2%) were confined to the ovary and 52 cases (26.8%) had extraovarian metastasis at the initial diagnosis of MSO. During the follow-up, 75.3% of these patients showed no evidence of disease and 18.0% were alive with disease. Only 13 deaths occurred, with 10 attributed to MSO. The 5, 10, and 15-year OS rates were 91.4, 87.7, and 83.5%, respectively. The 5, 10, and 15-year DSS rates were 93.8, 90.0, and 85.7%, respectively. Logistic regression revealed that International Federation of Gynecology and Obstetrics (FIGO) stage IV was the only risk factor for DFS [p < 0.001; odds ratio (OR) 7.328; 95% CI 3.103-16.885, FIGO stage IV vs. stage I; p = 0.021; OR 4.750, 95% CI 1.264-17.856, FIGO stage IV vs. stage II-III]. The multivariate Cox regression analysis showed that poor differentiation was the only risk factor for both OS (p = 0.005, OR 6.406; 95% CI 1.730-23.717) and DSS (p = 0.001, OR 9.664; 95% CI 2.409-38.760), while age ≥45 years was the prognostic predictor for OS (p = 0.038, OR 4.959; 95% CI 1.093-22.508). Conclusion: Survival outcomes were excellent in patients with MSO, irrespective of the treatment strategy, FIGO stage IV, age ≥45 years, and poor differentiation of tumors were the independent risk factors.
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2024-11-13 | GSE278926 | GEO
Project description:BackgroundMalignant struma ovarii (MSO) is a unique type of ovarian malignancy that data on the survival outcome is limited and management strategy remains controversial due to its extreme rarity.MethodsTo investigate the clinical characteristics and treatment options in patients with MSO confined to the ovary, while also evaluating the recurrent-free survival (RFS) and overall survival (OS) rate in this population, a retrospective study was conducted. One hundred twenty-five cases of MSO confined to the ovary were enrolled and their clinical characteristics, treatment strategies, and results of follow-up were analyzed. OS and RFS were assessed by Kaplan-Meier analyses and Cox regression models.ResultsThe most common pathological subtype in this cohort was papillary carcinoma (44.8%). Other reported subtypes, in order of prevalence, were follicular variant of papillary carcinoma, follicular carcinoma, and mixed follicular-papillary carcinoma. Surgical treatment options varied in this cohort that 8.0% of the patients received ovarian cystectomy, 33.6% underwent unilateral salpingo-oophorectomy (USO), 5.6% received bilateral salpingo-oophorectomy (BSO), 21.6% received total abdominal hysterectomy with BSO (TAH/BSO), and 17.6% were treated with debulking surgery; 20.0% of them received radioiodine therapy (RAI). Twenty-seven patients experienced recurrence with a median RFS of 14.0 years (95% confidence interval [CI], 9.5-18.5). The 5-year and 10-year recurrent rate were 27.1, 35.2%, respectively. Eight patients died during follow-up, with five attributed to MSO; the 5-year, 10-year, and 20-year OS rate was 95.3, 88.7 and 88.7%, respectively. However, the univariate and multivariate Cox regression showed no potential risk factor for RFS and OS.ConclusionPatients with MSO confined to the ovary had an excellent survival outcome, despite varied treatment strategies, and the recurrent rate was relatively high. We recommend USO as the preferred surgical option in this population since more aggressive surgery does not improve outcomes and the benefits of RAI are uncertain.
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