|Year : 2014 | Volume
| Issue : 2 | Page : 108-111
Peritoneal and mediastinal highly differentiated follicular carcinoma of ovarian origin
Kathleen Carey1, Manoj Jain1, Murli Krishna2, Joseph Accurso1
1 Department of Radiology, Mayo Clinic, Jacksonville, FL 32224, USA
2 Department of Pathology, Mayo Clinic, Jacksonville, FL 32224, USA
|Date of Web Publication||9-Apr-2014|
Department of Radiology, Mayo Clinic, 4500 San Pablo Road South, Jacksonville, FL 32224
Source of Support: None, Conflict of Interest: None
| Abstract|| |
A 70-year-old female patient presented to her primary care doctor with persistent elevated alkaline phosphatase of suspected metastatic etiology. Computed tomography demonstrated epicardial and peritoneal nodules. Biopsy of one of the peritoneal nodules revealed thyroid tissue and extraovarian struma ovarii was considered. The patient had a history of remote total abdominal hysterectomy and bilateral salpingo-oophorectomy 31 years prior for endometriosis with no available pathology from that surgery. The patient recalls being told that she had a left ovarian cyst. A thyroid ultrasound was performed that demonstrated multiple nodules without concerning features; however, due to high clinical suspicion, a total thyroidectomy was performed. Upon full histological evaluation a 0.5 cm papillary microcarcinoma was found. Given the rarity of metastatic papillary cancer to the peritoneum and the small size and grade of the tumor, a diagnosis of highly differentiated follicular carcinoma of ovarian origin was favored. The patient was subsequently treated with radioiodine therapy.
Keywords: Highly differentiated follicular carcinoma of ovarian origin, metastatic papillary cancer, struma ovarii
|How to cite this article:|
Carey K, Jain M, Krishna M, Accurso J. Peritoneal and mediastinal highly differentiated follicular carcinoma of ovarian origin. Indian J Nucl Med 2014;29:108-11
|How to cite this URL:|
Carey K, Jain M, Krishna M, Accurso J. Peritoneal and mediastinal highly differentiated follicular carcinoma of ovarian origin. Indian J Nucl Med [serial online] 2014 [cited 2019 May 19];29:108-11. Available from: http://www.ijnm.in/text.asp?2014/29/2/108/130301
| Introduction|| |
Struma ovarii is defined as an ovarian teratoma composed primarily of thyroid tissue. ,,,,, The thyroid elements may be benign or malignant in histology, the latter most commonly appearing as papillary and less frequently follicular thyroid-type carcinoma.
Extra-ovarian spread of struma ovarii is rare, but when it does occur, it is usually intra-abdominal and the majority of cases are diagnosed post-operatively. The extra-ovarian tissue can have a histologically benign appearance or can demonstrate features seen in thyroid-type carcinoma. The former has been historically classified as "peritoneal strumosis," although more recent literature , recognizes these cases as neoplastic, terming them highly differentiated follicular carcinoma of ovarian origin (HDFCO).
HDFCO is characterized by "extraovarian dissemination of thyroid elements with histological resemblance to non-neoplastic thyroid tissue." , Our literature search reveals only two documented cases of HDFCO with spread to the peritoneum, making this a rare but important entity to consider in cases with appropriate pathological findings. ,,
| Case Report|| |
Here we report a gravida 3, para 2 Caucasian 70-year-old female patient who was evaluated at an outside institution for persistently elevated alkaline phosphatase levels with no clinical complaints. A bone scan was obtained at that time and demonstrated abnormal uptake in the cranium, long bones and pelvis, raising the possibility of metastatic disease and the patient was referred to our institution. The patient demonstrated an elevated thyroglobulin level of 101.5 ng/ml (normal 0-33.0 ng/ml). In addition, the tumor marker CA 15-3 was also elevated at 34.7 u/ml (normal 10-19.9 u/ml).
Contrast enhanced computed tomography scan of the chest, abdomen and pelvis demonstrated multiple peritoneal nodules, the largest of which measured 3.2 cm × 2.2 cm [Figure 1]. In addition, hyperenhancing epicardial nodules were also noted [Figure 2]. Biopsy of one of the peritoneal lesions revealed thyroid tissue without atypia and we investigated whether struma ovarii was possible. The patient had undergone an elective total abdominal hysterectomy and bilateral salpingo-oophorectomy for endometriosis at age 39 years. The pathology findings were not available. The patient reported being told of a left ovarian cyst without the need for follow-up.
|Figure 1: Computed tomography image of the pelvis demonstrating multiple peritoneal lesions, the largest of which is demonstrated above|
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Given the number of lesions associated with the omentum, the patient underwent a diagnostic laparoscopy and partial omentectomy. Pathology from the excised omental nodules demonstrated histologically benign appearing thyroid tissue with multiple thyroid follicles of various sizes [Figure 3]. Pelvic washings were negative for malignant cells.
|Figure 3: Papillary thyroid carcinoma, with characteristic nuclear grooves (arrowheads) and a nuclear inclusion (arrow) (H and E, ×400)|
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As part of the diagnostic evaluation a bone marrow biopsy was performed and was negative for malignancy. In addition, a thyroid ultrasound was performed that demonstrated multiple small nodules with a dominant nodule in the right upper thyroid lobe. Three of the nodules were biopsied and the cytologic findings were consistent with benign nodules.
Given the patient's extensive extra-ovarian struma ovarii, a total thyroidectomy was recommended prior to radioiodine therapy. Pathological evaluation of the thyroid revealed a 0.5 cm focus of well-differentiated papillary carcinoma (a microcarcinoma) without lymphovascular invasion, stage PT1 [Figure 4].
|Figure 4: Abdominal thyroid tissue characterized by histologically bland thyroid tissue including follicles and colloid (H and E, ×400)|
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Total body I-123 uptake scan was performed for treatment planning purposes and demonstrated innumerable areas of uptake within the chest, abdomen and pelvis [Figure 5]. A discussion was held regarding the appropriate I-131 treatment dose. Given concerns about bowel radiation injury, the patient was treated with 30.5 mCi of radioiodine 131.
|Figure 5: Total body radioactive iodine scan prior to therapy shows uptake in the abdomen as well as small foci in the mediastinum. At 24 h I-123 uptake in the cervical thyroid bed was measured at 1.4% (normal = 8%-29%)|
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A follow-up I-123 uptake scan 4 months later showed resolution of the chest uptake and significant decline in the number of lesions throughout the abdomen and pelvis that were consistent with a short-term positive response [Figure 6]. Thyroglobulin tumor marker at the follow-up scan was elevated at 91 ng/ml. Plans were made for continued follow-up.
|Figure 6: Follow-up total body radioactive iodine scan demonstrates resolution mediastinal uptake and significantly decreased uptake in multiple abdominal and pelvic nodules|
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| Discussion|| |
Struma ovarii occurs when thyroid tissue is the predominant element in an ovarian teratoma. In a small percentage of these cases, the thyroid tissue has a malignant histology and is capable of spreading to the peritoneum, bones, cranium and multiple organs. , Struma ovarii has been stated to metastasize in 5-23% of cases.  The majority of metastases are diagnosed upon excision of the primary ovarian mass. There are less than thirty reports of struma ovarii metastasizing to distant sites  and even fewer that demonstrate the significantly delayed presentation that was seen in this case. We could find only one reported case in which peritoneal dissemination was discovered 26 years after initial operation; However, several cases of delayed dissemination to other organ systems exist. , For example McDougall et al. reported the discovery of vertebral and liver metastases in a patient 41 years after oophorectomy for ovarian tumor. 
Roth and Karseladze  have recently proposed a new entity termed "HDFCO". This term has been applied to patients with metastases of ovarian struma that have a bland, nonmalignant histologic appearance. This diagnosis characteristically cannot be made until distant metastasis has occurred. The case presented here is consistent with the diagnosis of HDFCO.
The current case is complicated by the lack of any significant ovarian pathology from the patient's hysterectomy and bilateral oophorectomy 31 years ago. After pathology review of the omentectomy specimens confirmed thyroid tissue without atypia, the location of the primary tumor remained unclear-thyroid versus ovarian.
The histologic features of the papillary thyroid microcarcinoma in this case were distinctly different from the histology of the omental nodules. The possibility of distant metastases from the papillary thyroid microcarcinoma was felt to be highly unlikely due to its small size and lack of cervical lymph node metastases. ,,
Although we were unable to review the original ovarian pathology from 31 years ago, which was reported as benign, our investigation supports this case as a likely additional example of HDFCO and illustrates the insidious nature of struma ovarii and the importance of maintaining a high degree of suspicion, even in patients who have had remote oophorectomy.
| References|| |
|1.||Makani S, Kim W, Gaba AR. Struma Ovarii with a focus of papillary thyroid cancer: A case report and review of the literature. Gynecol Oncol 2004;94:835-9. |
|2.||Roth LM, Karseladze AI. Highly differentiated follicular carcinoma arising from struma ovarii: A report of 3 cases, a review of the literature, and a reassessment of so-called peritoneal strumosis. Int J Gynecol Pathol 2008;27:213-22. |
|3.||Roth LM, Miller AW 3 rd , Talerman A. Typical thyroid-type carcinoma arising in struma ovarii: A report of 4 cases and review of the literature. Int J Gynecol Pathol 2008;27:496-506. |
|4.||Salman WD, Singh M, Twaij Z. A case of papillary thyroid carcinoma in struma ovarii and review of the literature. Patholog Res Int 2010;2010:352476. |
|5.||Song HJ, Xue YL, Xu YH, Qiu ZL, Luo QY. Rare metastases of differentiated thyroid carcinoma: Pictorial review. Endocr Relat Cancer 2011;18:R165-74. |
|6.||Džepina D, Zurak K, Petric V, Cupiæ H. Pathological characteristics and clinical perspectives of papillary thyroid cancer: Study of 714 patients. Eur Arch Otorhinolaryngol 2013 [Epub ahead of print]. |
|7.||McDougall IR. Metastatic struma ovarii: The burden of truth. Clin Nucl Med 2006;31:321-4. |
|8.||Willemse PH, Oosterhuis JW, Aalders JG, Piers DA, Sleijfer DT, Vermey A, et al. Malignant struma ovarii treated by ovariectomy, thyroidectomy, and 131I administration. Cancer 1987;60:178-82. |
|9.||Roth LM, Talerman A. The enigma of struma ovarii. Pathology 2007;39:139-46. |
|10.||McDougall IR, Krasne D, Hanbery JW, Collins JA. Metastatic malignant struma ovarii presenting as paraparesis from a spinal metastasis. J Nucl Med 1989;30:407-11. |
|11.||Dinneen SF, Valimaki MJ, Bergstralh EJ, Goellner JR, Gorman CA, Hay ID. Distant metastases in papillary thyroid carcinoma: 100 cases observed at one institution during 5 decades. J Clin Endocrinol Metab 1995;80:2041-5. |
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