Management of malignant struma ovarii: is aggressive therapy justified? Case report and literature review

Thyroid Research - Tập 15 Số 1
Letiţia Leuştean1, Maria-Christina Ungureanu1, Cristina Preda1, Ştefana Bîlha1, Florin Obrocea2, Radu Dănilă3, Laura Stătescu4, Delia Ciobanu5
1Department of Endocrinology, “Grigore T. Popa” University of Medicine and Pharmacy Iasi, Iasi, Romania
2Green Onco-Medical Bucuresti, București, Romania
3Department of Surgery, “Grigore T. Popa” University of Medicine and Pharmacy Iasi, Iasi, Romania
4Department of Dermatology, “Grigore T. Popa” University of Medicine and Pharmacy Iasi, Iasi, Romania
5Department of Pathology, “Grigore T. Popa” University of Medicine and Pharmacy Iasi, Iasi, Romania

Tóm tắt

Abstract Background Struma ovarii (SO) is a rare ovarian teratoma containing predominantly thyroid tissue. In rare situations SO may develop malignancy. Most cases of malignant struma ovarii (MSO) are diagnosed after surgical removal, based on histopathological examination. There are still controversies regarding the extent of surgery and postoperative management in MSO, due to its unpredictable behavior, possible risk of metastasis and relatively high rate of recurrence. Case Presentation We present the case of a patient diagnosed with a right ovarian cyst discovered incidentally during routine ultrasound examination. Its rapid growth and pelvic MRI raised the suspicion of a neoplastic process. She underwent total hysterectomy and bilateral adnexectomy. The anatomopathological diagnosis was MSO with follicular variant of papillary thyroid carcinoma. Prophylactic total thyroidectomy was performed, followed by radioactive iodine ablation (RAI), and suppressive therapy with levothyroxine. At 1 year follow-up, the patient was disease free. Conclusions Even if latest literature reports consider that completion of local surgery with total thyroidectomy and RAI might be too aggressive in cases of MSO without extraovarian extension, in our case it was decided to follow the protocol for primary thyroid carcinoma, in order to reduce the recurrence risk.

Từ khóa


Tài liệu tham khảo

Li S, Yang T, Xiang Y, Li X, Zhang L, Deng S. Clinical characteristics and survival outcomes of malignant struma ovarii confined to the ovary. BMC Cancer. 2021;21(1):383. https://doi.org/10.1186/S12885-021-08118-7.

Fujiwara S, Tsuyoshi H, Nishimura T, Takahashi N, Yoshida Y. Precise preoperative diagnosis of struma ovarii with pseudo-Meigs’ syndrome mimicking ovarian cancer with the combination of 131 I scintigraphy and 18 F-FDG PET: case report and review of the literature. J Ovarian Res. 2018;11(1):11. https://doi.org/10.1186/S13048-018-0383-2.

Aguilera BG, Vazquez RG, Herguido NG, Gallego FS, Gonzalez EN. The lack of consensus in management of malignant struma ovarii. Gynecol Endocrinol. 2015;31(4):258–9.

Selvaggi F, Risio D, Waku M, Simo D, Angelucci D, D’Aulerio A, et al. Struma ovarii with follicular thyroid-type carcinoma and neuroendocrine component: case report. World J Surg Oncol. 2012;10:93. https://doi.org/10.1186/1477-7819-10-93.

Salman WD, Singh M, Twaij Z. A case of papillary thyroid carcinoma in struma ovarii and review of the literature. Patholog Res Int. 2010;2010:1–5.

Rockson O, Kora C, Ramdani A, Basma A, Bouhout T, Serji B, et al. Struma ovarii: two case reports of a rare teratoma of the ovary. J Surg case reports. 2020;2020(12):rjaa493. https://doi.org/10.1093/JSCR/RJAA493.

Al Hassan MS, Saafan T, El Ansari W, Al Ansari AA, Zirie MA, Farghaly H, et al. The largest reported papillary thyroid carcinoma arising in struma ovarii and metastasis to opposite ovary: case report and review of literature. Thyroid Res. 2018;11(1). https://doi.org/10.1186/S13044-018-0054-9

Koehler VF, Keller P, Waldmann E, Schwenk N, Kitzberger C, Schmohl KA, et al. An unusual case of struma ovarii. Endocrinol diabetes Metab case reports. 2021;2021(1). https://doi.org/10.1530/EDM-20-0142

Yoo S-C, Chang K-H, Lyu M-O, Chang S-J, Ryu H-S, Kim H-S. Clinical characteristics of struma ovarii. J Gynecol Oncol. 2008;19(2):135.

Dujardin MI, Sekhri P, Turnbull LW. Struma ovarii: role of imaging? Insights Imaging. 2014;5(1):41–51.

Cui Y, Yao J, Wang S, Zhao J, Dong J, Liao L. The Clinical and Pathological Characteristics of Malignant Struma Ovarii: An Analysis of 144 Published Patients. Front Oncol. 2021;11:645156. https://doi.org/10.3389/FONC.2021.645156.

Wee JYS, Li X, Chern BSM, Chua ISY. Struma ovarii: management and follow-up of a rare ovarian tumour. Singapore Med J. 2015;56(1):35–9.

Dardik RB, Dardik M, Westra W, Montz FJ. Malignant struma ovarii: two case reports and a review of the literature. Gynecol Oncol. 1999;73(3):447–51.

Goffredo P, Sawka AM, Pura J, Adam MA, Roman SA, Sosa JA. Malignant struma ovarii: a population-level analysis of a large series of 68 patients. Thyroid. 2015;25(2):211–5.

Siegel MR, Wolsky RJ, Alvarez EA, Mengesha BM. Struma ovarii with atypical features and synchronous primary thyroid cancer: a case report and review of the literature. Arch Gynecol Obstet. 2019;300(6):1693–707.

Wolff EF, Hughes M, Merino MJ, Reynolds JC, Davis JL, Cochran CS, et al. Expression of benign and malignant thyroid tissue in ovarian teratomas and the importance of multimodal management as illustrated by a BRAF-positive follicular variant of papillary thyroid cancer. Thyroid. 2010;20(9):981–7.

Robboy SJ, Shaco-Levy R, Peng RY, Snyder MJ, Donahue J, Bentley RC, et al. Malignant struma ovarii: An analysis of 88 cases, including 27 with extraovarian spread. Int J Gynecol Pathol. 2009;28(5):405–22.

Shanbhogue AKP, Shanbhogue DKP, Prasad SR, Surabhi VR, Fasih N, Menias CO. Clinical syndromes associated with ovarian neoplasms: a comprehensive review. Radiographics. 2010;30(4):903–19.

Park SB, Kim JK, Kim KR, Cho KS. Imaging findings of complications and unusual manifestations of ovarian teratomas. Radiographics. 2008;28(4):969–83.

Shen J, Xia X, Lin Y, Zhu W, Yuan J. Diagnosis of Struma ovarii with medical imaging. Abdom Imaging. 2011;36(5):627–31.

Matsuki M, Kaji Y, Matsuo M, Kobashi Y. Struma ovarii: MRI findings. Br J Radiol. 2000;73(865):87–90.

Matsumoto F, Yoshioka H, Hamada T, Ishida O, Noda K. Struma ovarii: CT and MR findings. J Comput Assist Tomogr. 1990;14(2):310–2.

Yamashita Y, Hatanaka Y, Takahashi M, Miyazaki K, Okamura H. Struma ovarii: MR appearances. Abdom Imaging. 1997;22(1):100–2.

Joja I, Asakawa T, Mitsumori A, Nakagawa T, Hiraki Y, Kudo T, et al. Struma ovarii: appearance on MR images. Abdom Imaging. 1998;23(6):652–6.

Loizzi V, Cormio G, Resta L, Fattizzi N, Vicino M, Selvaggi L. Pseudo-Meigs syndrome and elevated CA125 associated with struma ovarii. Gynecol Oncol. 2005;97(1):282–4.

Jin C, Dong R, Bu H, Yuan M, Zhang Y, Kong B. Coexistence of benign struma ovarii, pseudo-Meigs’ syndrome and elevated serum CA 125: Case report and review of the literature. Oncol Lett. 2015;9(4):1739–42.

Muthusamy S, Azhar Shah S, Abdullah Suhaimi SN, Kassim N, Mahasin M, Mohd Saleh MF, et al. CD56 expression in benign and malignant thyroid lesions. Malays J Pathol. 2018;40(2):111–9.

Pyo JS, Kim DH, Yang J. Diagnostic value of CD56 immunohistochemistry in thyroid lesions. Int J Biol Markers. 2018;33(2):161–7.

Wang J, Luo H, Yang T, Yang F, Tian T. Imaging features of struma ovarii in conventional ultrasound and CEUS. Chinese J Med Imaging Technol. 2017;33(10):1531–4.

Leong A, Roche PJR, Paliouras M, Rochon L, Trifiro M, Tamilia M. Coexistence of malignant struma ovarii and cervical papillary thyroid carcinoma. J Clin Endocrinol Metab. 2013;98(12):4599–605.

Kumar SS, Rema P, R AK, Varghese BT. Thyroid type papillary carcinoma arising in a mature teratoma. Indian J Surg Oncol. 2014;5(3):168–70.

Yassa L, Sadow P, Marqusee E. Malignant struma ovarii. Nat Clin Pract Endocrinol Metab. 2008;4(8):469–72.

Janszen EWM, Van Doorn HC, Ewing PC, De Krijger RR, De Wilt JHW, Kam BLR, et al. [Malignant struma ovarii]. Ned Tijdschr Geneeskd. 2008;152(29):1647 (author reply 1647-8).

Marti JL, Clark VE, Harper H, Chhieng DC, Sosa JA, Roman SA. 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. 2012;22(4):400–6.

McGill JF, Sturgeon C, Angelos P. Metastatic struma ovarii treated with total thyroidectomy and radioiodine ablation. Endocr Pract. 2009;15(2):167–73.

Poli R, Scatolini M, Grosso E, Maletta F, Gallo M, Liscia D, et al. Malignant struma ovarii: next-generation sequencing of six cases revealed Nras, Braf, and Jak3 mutations. Endocrine. 2021;71(1):216–24.

Tan A, Stewart CJR, Garrett KL, Rye M, Cohen PA. Novel BRAF and KRAS Mutations in Papillary Thyroid Carcinoma Arising in Struma Ovarii. Endocr Pathol. 2015;26(4):296–301.

Filetti S, Durante C, Hartl D, Leboulleux S, Locati LD, Newbold K, et al. Thyroid cancer: ESMO Clinical Practice Guidelines for diagnosis, treatment and follow-up†. Ann Oncol Off J Eur Soc Med Oncol. 2019;30(12):1856–83.