Abstract
Introduction
Struma ovarii are quite unusual lesions that represent less than 3% of all teratomas and its malignant transformation is very uncommon. The clinical manifestations are characteristic of pelvic tumor and the hormonal metabolism is not usually modified. Radiography, employing ultrasound procedures, is the most commonly used pre-surgical detection method but only histological examination makes the diagnosis. The malignance recognition by pathological study not always is easy; in this sense, it requires an exhaustive sampling of the lesion, being specially carefully in some aspects related with malignant transformation such as extending beyond the capsula and involving peripheral tissues. A thyroidal differentiation must be confirmed by immunohistochemical study and other local processes with similar histology should be ruled out. Given the exceptional character of malignant forms, there does not appear to be unanimous agreement on a standard therapy with a somewhat uncertain prognosis.
Case report
We show a case of a 22-year-old patient with an ovarian tumor that was discovered by ultrasound examination and surgically removed. The histologic study revealed struma ovarii with malignant transformation towards follicular carcinoma and unlike previously published cases, had a prevalence of clear cells. The patient was submitted to a second surgical staging intervention, with conservative surgery and follow-up controls being considered given that was a young woman with a desire to have children. Laparoscopy was employed as the best method capable to facilitate shorter convalescence.
Outcome
Clinical and analytical controls, measuring thyroglobulin levels, has been satisfactory up to the present.
Similar content being viewed by others
References
Ashton MA (1995) Strumal carcinoid of the ovary associated with hyperinsulinaemic hypoglycemia and cutaneous melanosis. Histopathology 27:463–467
Basolo F, Molinaro E, Agate L, Pinchera A, Pollina L, Chiapetta G, Monaco C, Santoro M, Fusco A, Miccoli P, Elisei R, Capezzone M, Pacini F (2001) RET protein expression has no prognostic impact on the long-term outcome of papillary thyroid carcinoma. Eur J Endocrinol 145:599–604
Bayot MR, Chopra IJ (1995) Coexistence of struma ovarii and Graves’s disease. Thyroid 5:469–471
Berghella V, Ngadiman S, Rosenberg H, Hoda S, Zuna RE (1997) Malignant struma ovarii. A case report and review of the literature. Gynecol Obstet Invest 43:68–72
Bhansali A, Jain V, Rajwanshi A, Lodha S, Dash RJ (1999) Follicular carcinoma in a functioning struma ovarii. Postgrad Med J 75:617–618
Bonadio AG (2002) Hashimoto’s thyroiditis in “struma ovarii”. Case report an review of the literature. Minerva Endocrinol 27:49–52
Burg J, Kommos E, Bittinger F, Moll R, Kirkpatrick CJ (2002) Mature cystic teratoma of the ovary with struma and benign Brenner tumor: a case report with immunohistochemical characterization. Int J Gynecol Pathol 21:74–77
Carvalho JP, Carvalho FM, Oliveira FF, Camargo RY (2002) Hypothyroidism following struma ovarii resection: a case report. Rev Hosp Clin Fac Med Sao Paulo 57:112–114
Chan SW, Farrell KE (2001) Metastatic thyroid carcinoma in the presence of struma ovarii. Med J Aust 175:373–374
Checrallah A, Medlej R, Saade C, Khayat G, Halaby G (2001) Malignant struma ovarii: an unusual presentation. Thyroid 11:889–892
Cheung CC, Ezzat S, Freeman JL, Rosen IB, Asa SL (2001) Immunohistochemical diagnosis of papillary thyroid carcinoma. Mod Pathol 14:338–342
Dalgaard JB, Wetheland P (1956) Struma ovarii. A follow-up study of 20 cases. Acta Clin Scand 112:1
Dardik RB, Dardik M, Westra W, Montz FJ (1999) Malignant struma ovarii: two case reports and a review of the literature. Gynecol Oncol 73:447–451
DeSimone CP, Lele SM, Modesitt SC (2003) Malignant struma ovarii: a case report and analysis of cases reported in the literature with focus on survival and I131 therapy. Gynecol Oncol 89:543–548
Dohke M, Watanabe Y, Takahashi A, Katayama T, Amoh Y, Ishimori T, Okumura A, Oda K, Mitsudo K, Minami K, Ukita M, Dodo Y (1997) Struma ovarii: MR findings. J Comput Assist Tomogr 21:265–267
Doldi N, Taccagni GL, Bassan M, Frigerio L, Mangili G (1998) Hashimoto’s disease in a papillary carcinoma of the thyroid originating in a teratoma of the ovary (malignant struma ovarii). Gynecol Endocrinol 68:210–213
Dunzendorfer T, de las Morenas A, Ka lir T, Levin RM (1999) Struma ovarii and hyperthyroidism. Thyroid 9:499–502
Emoto M, Obama H, Horiuchi S, Miyakawa T, Kawarabayashi T (2000) Transvaginal color Doppler ultrasonic characterization of benign and malignant ovarian cystic teratomas and comparison with serum squamous carcinoma antigen. Cancer 15:2298–2304
Erez SE, Richard RM, Shettles LB (1965) Hashimoto’s disease in a benign cystic teratoma of the ovary. Am J Obstet Gynecol 92:273
Grandet PJ, Remi MH (2000) Struma ovary with hyperthyroidism. Clin Nucl Med 25:763–765
Gusberg SB, Danforth DN (1944) Clinical significance of struma ovarii. Am. J Obstet Gynecol 48:537
Hamazaki S, Okino T, Tsukayama C, Okada S (2002) Expression of thyroid transcription factor-1 in strumal carcinoid and struma ovarii: an immunohistochemical study. Pathol Int 52:458–462
Hamel P, Crouet H, Balout P, Herlicoviez M (1996) Ovarian goiter with hyperthyroidism. A case report. J Gyneocol Obstet Biol Reprod 25:797–798
Hemli JM, Barakate MS, Appleberg M, Delbridge LW (2001) Papillary carcinoma of the thyroid arising in struma ovarii: report of a case and review of management guidelines. Gynecol Endocrinol 15:243–247
Huh JJ, Montz FJ, Bristow RE (2002) Struma ovarii associated with pseudo-Meigs’ syndrome and elevated serum CA 125. Gynecol Oncol 86:231–234
Jhiang SM, Mazzaferri EL (1994) The ret/PTC oncogen in papillary thyroid carcinoma. J Lab Clin Med 123:331–337
Jhiang SM, Cho JY, Furminger TL, Sagartz JE, Tong Q, Capen CC, Mazzaferri EL (1998) Thyroid carcinomas in RET/PTC transgenic mice. Recent Results Cancer Res 154:265–270
Joja L, Asakawa T, Mitsumori A, Nakagawa T, Hiraki Y, Kudo T, Ando M, Akamatsu M (1998) Struma ovarii: appearance on MR images. Abdom Imaging 23:652–656
Kabuckuoglu F, Baksu A, Yilmaz B, Aktumen A, Evren I (2002) Malignant struma ovarii. Pathol Oncol Res 8:145–147
Kim JC, Kim SS, Park JY (2000) MR findings of struma ovarii. Clin Imaging 24:28–33
Konez O, Hanelin LG, Jenison EL, Goyal M, Randolph W (2000) Functioning liver metastasis on an I-131 whole body scan: a case of malignant struma ovarii. Clin Nucl Med 25:465–496
Kostoglou-Athanassiou I, Lekka-katsuoli I, Gogou L, Papagrigoriou L, Chatonides I, Kaldrymides P (2002) Malignant struma ovarii: report of case and review of the literature. Horm Res 58:34–38
Lieberman G, Buscombe JR, Hilson AJ, Thakrar D, MacLean A (1998) The detection of struma ovarii in two patients by radioimmunoscintigraphy. Am J Obstet Gynecol 179:262–263
Logani S, Baloch ZW, Snyder PJ, Weinstein R, LiVolsi VA (2001) Cystic ovarian metastasis from papillary thyroid carcinoma: a case report. Thyroid 11:1073–1075
Loy TS, Darkow GV, Spollen LE, Diaz-Arias AA (1994) Immunostaining for Leu-7 in the diagnosis of thyroid carcinoma. Arch Pathol Lab Med 118:172–174
Mancuso A, Triolo O, Leonardi L, De Vivo A (2001) Struma ovarii: a rare benign pathology which may erroneously suggest malignance. Acta Obstet Gynecol Scand 80:1075–1076
Matsuda K, Maehama T, Kanazawa K (2001) Malignant struma ovarii with thyrotoxicosis. Gynecol Oncol 82:575–577
Matsuki M, Kaji Y, Matsuo M, Kobashi Y (2000) Struma ovarii: MR findings. Br J Radiol 73:87–90
Nieminen I, Von Numers C, Widholm C (1964) Struma ovarii. Acta Obstet Gynecol Scand 42:399
Okada S, Ohaki Y, Kawamura T, Hayashi T, Kumazaki T (2000) Cystic struma ovarii: imaging findings. J Comput Assist Tomogr 24:413–415
Ordi J, Romagosa C, Tavassoli FA, Nogales F, Palacin A, Condom E, Torne A, Cardesa A (2003) CD10 expression in epithelial tissues and tumors of the gynecologic tract: a useful marker in the diagnosis of mesonephric, trophoblastic, and clear cell tumors. Am J Surg Pathol 27:178–186
Rabczynski JK, Zabel M, Prudlak L, Kochman AT, Lewandowski M (1999) Strumal carcinoid of ovary: a case report with immunohistochemical investigations. Clin Exp Obstet Gynecol 26:112–114
Ramondetta LM, Carlson JA Jr, Schwarting R (1997) Atypical Meigs’ syndrome and bilateral ovarian stromal hyperplasia. A case report. J Reprod Med 42:603–605
Ross DS (1998) Syndromes of thyrotoxicosis with low radioactive iodine uptake. Endocrinol Metab Clin North Am 27:169–185
Rotman-Pikielny P, Reynolds JC, Barker WC, Yen PM, Skarulis MC, Sarlis NJ (2000) Recombinant human thyrotropin for the diagnosis and treatment of highly functional metastatic struma ovarii. J Clin Endocrinol Metab 85:237–244
Scully RE (1970) Recent purposes in ovarian cancer. Hum Pathol 1:73
Shigeta H, Taga M, Kurogi K, Kitamura H, Motoyama T, Gorai I (1999) Ovarian strumal carcinoid with severe constipation: immunohistochemical and mRNA analyses of peptide YY. Hum Pathol 30:242–246
Simkin PH, Ramirez LA, Zwizig SL, Alfonso SA, Fraire AE, Khan A, Dunn AD, Dunn JT, Braverman LE (1999) Monomorphic teratoma of the ovary: a rare cause of triiodothyronine toxicosis. Thyroid 9:949–954
Sugg S, Ezzat S, Rosen IB, Freeman JL, Asa SL (1998) Distinct multiple ret/PTC gene rearrangements in multifocal papillary thyroid neoplasia. J Clin Endocrinol Metab 83:4116–4122
Takemori M, Nishimura R, Sugimura K, Obayashi C, Yasuda D (1995) Ovarian strumal carcinoid with markedly high serum levels of tumour markers. Gynecol Oncol 58:266–269
Takeuchi K, Murata K, Fujita I (2000) Malignant struma ovarii with peritoneal metastasis: report of two cases. Eur J Gynecol Oncol 21:260–261
Tallini G, Santoro M, Helie M, Carlomagno F, Salvatore G, Chiapetta G, Carcangiu ML, Fusco A (1998) RET/PTC oncogen activation defines a subset of papillary thyroid carcinomas lacking evidence of progression to poorly differentiated or undifferentiated tumor phenotypes. Clin Cancer Res 4:287–294
Tennvall J, Ljungberg O, Hogberg T (1997) Malignant struma ovarii with peritoneal dissemination. Histopathology 31:289–290
Vadmal MS, Smilari TF, Lovecchio JL, Klein IL, Hajdu DI (1997) Diagnosis and treatment of disseminated struma ovarii with malignant transformation. Gynecol Oncol 64:541–546
Volpi E, Ferrero A, Nasi PG, Sismondi P (2003) Malignant struma ovarii: a case report of laparoscopic management. Gynecol Oncol 90:191–194
Woodruff JD, Rauh JT, Markley RL (1966) Ovarian struma. Obstet Gynecol 27:194
Yamashita Y, Hatanaka Y, Takahashi M, Miyazaki A (1997) Struma ovarii: MR appearances. Abdom Imaging 22:100–102
Young RH, Scully RE (2002) Differential diagnosis of ovarian tumors based primarily on their patterns and cell types. Semin Diagn Pathol 18:161–235
Zakhem A, Aftimos G, Kreidi R (1990) Malignant struma ovarii: report of case and review of the literature. J Surg Oncol 43:61–65
Zalel Y, Caspi B, Tepper R (1997) Doppler flow characteristics of dermoid cysts: unique appearance of struma ovarii. J Ultrasound Med 16:355–358
Zalel Y, Seidman DS, Oren M, Archiron R, Gotlieb W, Mashiach S, Goldenberg M (2000) Sonographic and clinical characteristics of struma ovarii. J Ultrasound Med 19:857–861
Acknowledgement
The authors wish to thank Martin Hadley-Adams for the translation and assistance with the English language.
Author information
Authors and Affiliations
Corresponding author
Rights and permissions
About this article
Cite this article
García, A., Castellví, J., López, M. et al. Malignant struma ovarii mimic clear cell carcinoma. Arch Gynecol Obstet 271, 251–256 (2005). https://doi.org/10.1007/s00404-004-0605-2
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s00404-004-0605-2