Int J Thyroidol.  2023 Nov;16(2):184-189. 10.11106/ijt.2023.16.2.184.

A Case of Malignant Struma Ovarii with Cervical Papillary Thyroid Carcinoma

Affiliations
  • 1Departments of Otorhinolaryngology-Head and Neck Surgery, Soonchunhyang University Seoul Hospital, Seoul, Korea
  • 2Departments of Pathology, Soonchunhyang University Seoul Hospital, Seoul, Korea
  • 3Departments of Obstetrics and Gynecology, Soonchunhyang University Seoul Hospital, Seoul, Korea

Abstract

Struma ovarii (SO) is a rare ovarian teratoma, which is diagnosed when thyroid tissue accounts for more than 50% of the teratoma. The majority of SO are benign, but malignant tumors have been reported in a small percentage of cases. Five percent of SO cases have been proven to be malignant and, as in the thyroid gland, papillary carcinoma is the most common histotype arising in SO. Because this tumor is rare, there are no guidelines regarding the management of this cancer. Usually, total thyroidectomy followed by radioiodine treatment is the treatment of choice in metastatic malignant struma ovarii, but therapeutic decisions should be made individually based on clinical and pathological data. We recently experienced a case of a 45-year-old woman finally confirmed as malignant struma ovarii with cervical thyroid cancer. Therefore, we present this unique case with a review of the literature.

Keyword

Struma ovarii; Malignant struma ovarii; Papillary thyroid carcinoma; Struma ovarii with malignant transformation

Figure

  • Fig. 1 Pathology of the surgical specimen from the oophorectomy previously done at another hospital showing malignant struma ovarii. (A) Microscopically, the tumor is composed of variably sized thyroid follicles with colloid (arrow) (Hematoxylin & Eosin staining [HE], ×40). (B) The tumor cells show mild nuclear membrane irregularity and nuclear clearing in some follicles (arrowhead) (H&E, ×200).

  • Fig. 2 (A, B) Contrast enhanced abdomen and pelvic CT findings. Metastatic lymph nodes are noted in left paraaortic area (arrow in A, arrowhead in B). (C) Coronal T2-weighted enhanced MRI findings. Metastatic lymph nodes are noted in left paraaortic area (arrowheads).

  • Fig. 3 PET-CT findings. (A) Focally increased FDG uptakes in right infrathyroidal area (arrowhead). (B) Focally increased FDG uptakes in left pelvic cavity (arrow). (C) Right thyroid US. (D) Left thyroid US. Normal in size without abnormal focal lesion.

  • Fig. 4 Pathology of the surgical specimen from para-aortic lymph node showing metastatic carcinoma. (A) The tumor (right area) shows variable sized follicles with papillary fronds (H&E, ×40). (B) Tumor cells show round to ovoid nuclei with mild nuclear membrane irregularity, coarse chromatin, and eosinophilic to clear cytoplasm (H&E, ×400).

  • Fig. 5 Intraoperative photographs. (A) Operative field after total thyroidectomy and central lymph node dissection. Left recurrent laryngeal nerve (arrow) preserved. (B) Right recurrent laryngeal nerve (arrowhead) preserved. (C) Surgical specimen.

  • Fig. 6 Pathology of the surgical specimen from thyroid showing papillary microcarcinoma without lymph node metastasis. (A) The tumor (center) shows variable sized follicles with papillary fronds (H&E, ×40). (B) Tumor cells show round to ovoid nuclei with nuclear enlargement, nuclear membrane irregularity, and chromatin clearing (H&E, ×400).


Reference

References

1. Peterson WF, Prevost EC, Edmunds FT, Hundley JM Jr, Morris FK. 1955; Benign cystic teratomas of the ovary; a clinico-statistical study of 1,007 cases with a review of the literature. Am J Obstet Gynecol. 70(2):368–82. DOI: 10.1016/S0002-9378(16)37681-5. PMID: 13238472.
2. Devaney K, Snyder R, Norris HJ, Tavassoli FA. 1993; Proliferative and histologically malignant struma ovarii: a clinicopathologic study of 54 cases. Int J Gynecol Pathol. 12(4):333–43. DOI: 10.1097/00004347-199310000-00008. PMID: 8253550.
3. Rosenblum NG, LiVolsi VA, Edmonds PR, Mikuta JJ. 1989; Malignant struma ovarii. Gynecol Oncol. 32(2):224–7. DOI: 10.1016/S0090-8258(89)80037-X. PMID: 2910784. PMCID: PMC10147963.
Article
4. Makani S, Kim W, Gaba AR. 2004; Struma ovarii with a focus of papillary thyroid cancer: a case report and review of the literature. Gynecol Oncol. 94(3):835–9. DOI: 10.1016/j.ygyno.2004.06.003. PMID: 15350384.
Article
5. Grandet PJ, Remi MH. 2000; Struma ovarii with hyperthyroidism. Clin Nucl Med. 25(10):763–5. DOI: 10.1097/00003072-200010000-00001. PMID: 11043711.
Article
6. Kim S-J, Pak K, Lim H-J, Yun K-H, Seong S-J, Kim T-J, et al. 2002; Clinical diversity of struma ovarii. Korean J Obstet Gynecol. 748–52.
7. Zhang X, Axiotis C. 2010; Thyroid-type carcinoma of struma ovarii. Arch Pathol Lab Med. 134(5):786–91. DOI: 10.5858/134.5.786. PMID: 20441513.
Article
8. Hatami M, Breining D, Owers RL, Del Priore G, Goldberg GL. 2008; Malignant struma ovarii--a case report and review of the literature. Gynecol Obstet Invest. 65(2):104–7. DOI: 10.1159/000108654. PMID: 17890867.
Article
9. Soto Moreno A, Venegas EM, Rodriguez JR, Sanchez F, Robles MJ, Martinez MA, et al. 2002; Thyroid carcinoma on an ovarian teratoma: a case report and review of the literature. Gynecol Endocrinol. 16(3):207–11. DOI: 10.1080/gye.16.3.207.211. PMID: 12192892.
Article
10. Janszen EW, van Doorn HC, Ewing PC, de Krijger RR, de Wilt JH, Kam BL, et al. 2008; Malignant struma ovarii: good response after thyroidectomy and I ablation therapy. Clin Med Oncol. 2:147–52. DOI: 10.4137/CMO.S410. PMID: 21892277. PMCID: PMC3161682.
11. Marti JL, Clark VE, Harper H, Chhieng DC, Sosa JA, Roman SA. 2012; Optimal surgical management of well-differentiated thyroid cancer arising in struma ovarii: a series of 4 patients and a review of 53 reported cases. Thyroid. 22(4):400–6. DOI: 10.1089/thy.2011.0162. PMID: 22181336.
Article
12. Krishnamurthy A, Ramshankar V, Vaidyalingam V, Majhi U. 2013; Synchronous papillary carcinoma thyroid with malignant struma ovarii: a management dilemma. Indian J Nucl Med. 28(4):243–5. DOI: 10.4103/0972-3919.121975. PMID: 24379539. PMCID: PMC3866674.
13. Leite I, Cunha TM, Figueiredo JP, Felix A. 2013; Papillary carcinoma arising in struma ovarii versus ovarian metastasis from primary thyroid carcinoma: a case report and review of the literature. J Radiol Case Rep. 7(10):24–33. DOI: 10.3941/jrcr.v7i10.1593. PMID: 24421920. PMCID: PMC3888341.
Article
14. Leong A, Roche PJ, Paliouras M, Rochon L, Trifiro M, Tamilia M. 2013; Coexistence of malignant struma ovarii and cervical papillary thyroid carcinoma. J Clin Endocrinol Metab. 98(12):4599–605. DOI: 10.1210/jc.2013-1782. PMID: 24217901.
Article
15. González Aguilera B, Guerrero Vazquez R, Gros Herguido N, Sánchez Gallego F, Navarro Gonzalez E. 2015; The lack of consensus in management of malignant struma ovarii. Gynecol Endocrinol. 31(4):258–9. DOI: 10.3109/09513590.2014.995616. PMID: 25539066.
16. Brusca N, Del Duca SC, Salvatori R, D'Agostini A, Cannas P, Santaguida MG, et al. 2015; A case report of thyroid carcinoma confined to ovary and concurrently occult in the thyroid: is conservative treatment always advised? Int J Endocrinol Metab. 13(1):e18220. DOI: 10.5812/ijem.18220. PMID: 25745492. PMCID: PMC4338667.
17. Boyd JC, Williams BA, Rigby MH, Kieser K, Offman S, Shirsat H, et al. 2017; Malignant struma ovarii in a 30-year old nulliparous patient. Thyroid Res. 10:3. DOI: 10.1186/s13044-017-0038-1. PMID: 28572843. PMCID: PMC5450076.
18. Middelbeek RJW, O'Neill BT, Nishino M, Pallotta JA. 2017; Concurrent intrathyroidal thyroid cancer and thyroid cancer in struma ovarii: a case report and literature review. J Endocr Soc. 1(5):396–400. DOI: 10.1210/js.2017-00052. PMID: 29264493. PMCID: PMC5686688.
19. Gomes-Lima CJ, Nikiforov YE, Lee W, Burman KD. 2018; Synchronous independent papillary thyroid carcinomas in struma ovarii and the thyroid gland with different RAS mutations. J Endocr Soc. 2(8):944–8. DOI: 10.1210/js.2018-00132. PMID: 30087949. PMCID: PMC6065485.
Article
20. Siegel MR, Wolsky RJ, Alvarez EA, Mengesha BM. 2019; Struma ovarii with atypical features and synchronous primary thyroid cancer: a case report and review of the literature. Arch Gynecol Obstet. 300(6):1693–707. DOI: 10.1007/s00404-019-05329-z. PMID: 31679038.
Article
21. McGill JF, Sturgeon C, Angelos P. 2009; Metastatic struma ovarii treated with total thyroidectomy and radioiodine ablation. Endocr Pract. 15(2):167–73. DOI: 10.4158/EP.15.2.167. PMID: 19289330.
Article
22. Egan C, Stefanova D, Thiesmeyer JW, Lee YJ, Greenberg J, Beninato T, et al. 2022; Proposed risk stratification and patterns of radioactive iodine therapy in malignant struma ovarii. Thyroid. 32(9):1101–8. DOI: 10.1089/thy.2022.0145. PMID: 35765923.
Article
Full Text Links
  • IJT
Actions
Cited
CITED
export Copy
Close
Share
  • Twitter
  • Facebook
Similar articles
Copyright © 2024 by Korean Association of Medical Journal Editors. All rights reserved.     E-mail: koreamed@kamje.or.kr