{"paper_id":"6ac95b47-9da1-44c5-83ae-55864354f589","body_text":"COMMUNICA TION\nA rare case of endometrioma and metastases of previously\noperated struma ovarii\nVincenzo Campo & Sebastiano Campo\nReceived: 20 July 2008 / Accepted: 25 September 2008 / Published online: 7 November 2008\n# Springer-V erlag 2008\nAbstract We present the first case of association between\nan ovarian endometrioma and peritoneal metastases of\nstruma ovarii. In a 20-year-old woman, previously sub-\njected to right adnexectomy at 12 years old for struma\novarii, during operative videolaparoscopy, we observed on\nleft ovary the presence of endometrioma of 6 cm in\ndiameter and on pelvic peritoneum and right lateral\nabdominal wall, red colored nodules of multiple sizes; one\nof these was visible on the diaphragm and one on the right\nanterior lobe of the liver. Definitive histological examina-\ntion revealed endometriotic cyst, and the immunohisto-\nchemical analysis of peritoneal nodules revealed the\npresence of thyroid tissue.\nKeywords Struma ovarii . Metastases . Endometrioma .\nLaparoscopy\nIntroduction\nMature cystic teratomas account approximately 20% of all\novarian tumors [ 1]. These tumors are composed by\nepithelial tissue and can include hair, skin, teeth, bone [ 2],\nand other tissues, and approximately 15% contain thyroid\ntissue [ 3]. To classify a teratoma as struma ovarii, it must\ncontain thyroid tissue greater than 50% [ 4]. Struma ovarii\naccount for about 2.7% of all ovarian teratomas [ 5]; it\noccurs at all ages, but most commonly affects women in the\nfifth and sixth decades of life [ 6]. It is often diagnosed\nduring an exploratory laparotomy for pelvic mass, which is\nthe most frequent presenting symptom. It is usually a\nbenign condition even if occasionally a malignant transfor-\nmation is observed in less than 5% of cases [ 6].\nWe present the first case of association between an\novarian endometrioma and peritoneal metastases of struma\novarii (Figs. 1 and 2) previously operated.\nCase report\nWe report the case of a 22-year-old Caucasian woman who,\nat the age of 12 years, was admitted to her local hospital\ncomplaining of right lower quadrant pain. She underwent\nexploratory laparotomy and right salpingo-oophorectomy\nfor an ovarian cyst. Uterus and left ovary seemed to be\nnormal. The pathologic examination showed “struma\novarii” associated with mature cystic teratoma. A review\nof the pathology revealed no cellular atypia or malignant\nfeatures. The postoperative course was free of complica-\ntions, and the patient did not receive any further investiga-\ntion during the following years. After 8 years, the patient\nwas admitted with signs and symptoms of a left pelvic\ntumor. A pelvic ultrasound scan revealed a left ovarian\nlesion approximately 6 cm in diameter with a cystic\ncomponent, suggestive of endometrioma. The levels of\nalfa-fetoprotein was 3.56 ng/ml, CA-125 was 6.5 UI/ml,\nCEA 0.1 ng/ml, Ca19 –9 was 5 UI/ml, and she had normal\nthyroid parameters. The patient was taken to the operating\nroom for an operative laparoscopy. Surgical exploration\ncould find neither pelvic nor Douglas implants suggestive\nof peritoneal endometriosis or ascites. However, multiple\npink nodules were observed (diameter between 1.0 and\nGynecol Surg (2010) 7:27 –29\nDOI 10.1007/s10397-008-0444-4\nV . Campo (*) : S. Campo\nDepartment of Obstetrics and Gynecology,\nCatholic University of Sacred Heart,\nL.go Gemelli, 8,\n00168 Rome, Italy\ne-mail: vincecampo76@gmail.com\n\n3.0 cm) on the pelvic peritoneum, right lateral abdominal\nwall, diaphragm, vesical plica, and liver. Cystectomy with\nspillage of like chocolate liquid during ovarian mobilization\nand multiple biopsies of the nodules on the anterior\nabdominal wall and vesical plica were performed. We\nremoved cystic capsule by endobag. The patient was\ndischarged the day after surgery. On histopathological\nexamination, the ovarian cyst was found to be an\nendometriotic lesion, and the nodules were made up of\nfibrous tissue with follicular spaces lined by thyroid cells\nwith grooves and clear nuclei. Immunohistochemical study\nof the abdominal nodules showed strong expression of\nthyroglobulin (Tg), suggesting a diagnosis of recurrent\nstruma ovarii with features consistent with the follicular\nvariant of papillary thyroid carcinoma.\nDiscussion\nOnly three cases of simultaneous presence of endometriosis,\nand mature cystic teratoma of the ovary are described in\nliterature. Ferrario [7], in a 23-year-old woman submitted to\nlaparotomy for bilateral salpingo-oophorectomy because of\npelvic mass, described the simultaneous presence in the right\novary of an endometriotic cyst and a dermoid cyst. Caruso\nand Pirrelli [ 8], in a 28-year-old women with bilateral\novarian dermoid cysts, show in the left ovary an endome-\ntrioma. Frederick et al. [9] reported a case of a young woman\nwith bilateral dermoid cysts and endometriotic deposits in\nthe pelvis.\nTo our knowledge, we report the first case of association\nbetween ovarian endometrioma and peritoneal metastasis of\na recurrent struma ovarii 8 years after annessectomy and\nclassified as benign at histopathology and with no sensible\nexplanation to justify the malignant transformation of the\novarian tissue and the resultant metastatic spread.\nOur case is quite similar to V admal’s[ 10], who described a\ncase of recurrent struma ovarii with malignant transformation\n6 years after abdominal hysterectomy and bilateral salpingo-\noophorectomy in a 51-year-old woman operated for a left\novarian mass diagnosed at histopathology as a pure struma\novarii without cellular atypia or malignant features.\nThe interest of our case is due to the occasional finding,\nduring operative laparoscopy for other disease, of a\nrecurrent metastatic struma ovarii after 8 years.\nThe real incidence of malignancy in struma ovarii is\ndifficult to assess due to the rare nature of this condition.\nAn incidence of 0.1 –0.3% has been quoted in the literature\n[11]. Even if many authors [ 12] consider as benign the\npresence of peritoneal metastasis of thyroid tissue, others\nsuggest that any struma ovarii exhibiting metastatic\nbehavior should be regarded as malignant [ 4]. Metastasis\nof malignant struma ovarii is seen in approximately 5% of\ncases [ 13], even if a recent review [ 14] demonstrated a\nhigher metastasis rate of 23%, which was mainly intra-\nabdominal, although blood-borne metastasis can occur in\nthe liver, brain, lung, bone, and the contralateral ovary. In\nour patient, we found metastatic implants on pelvic\nperitoneum, right lateral abdomen wall, diaphragm, and\non the right anterior lobe of the liver. It is doubtful if these\nlesions are due to spillage during the first operation, even if\na laparotomic adnexectomy, which presents a lower risk of\nspillage, was performed.\nMost of the cases of malignant struma ovarii are\nsubclinical. Approximately 5 –8% of patients have clinical\nhyperthyroidism [ 6]. In our experience, after surgery, the\npatient had normal thyroid parameters (TSH 1.65 mcg/ml,\nFT3 2.1 pg/ml, and FT4 2.8 pg/ml) but presented higher\nlevels of thyroglobulin (305 ng/ml); at ultrasound exami-\nnation, no thyroid nodules were found.\nFig. 2 Left ovarian endometrioma\nFig. 1 Peritoneal metastases of struma ovarii and endometrioma on\nleft ovary\n28 Gynecol Surg (2010) 7:27 –29\n\nThe treatment of malignant struma ovarii remains\ncontroversial. Since metastatic struma ovarii is similar to\nmetastatic thyroid carcinoma, 131I therapy represents an\nideal way to treat both diseases [ 14]. However, as in\npatients with differentiated thyroid carcinoma, before 131I\ntreatment, it is necessary to remove the thyroid gland\ncompletely to exclude a primary thyroid carcinoma with\nsubsequent ovarian metastasis.\nAccording to others [ 10], and considering the presence\nof peritoneal metastasis, to increase the uptake of 131I by\nthe metastatic nodules, we suggest to the patient to have a\ntotal thyroidectomy followe d by radioiodine ablation.\nFurthermore, we propose to the patient an ovarian tissue\ncryopreservation and GnRHa administration before 131I\ntreatment in order to preserve fertility. We believe that these\nmeans should be offered to all young women diagnosed\nwith cancer.\nIn conclusion, our case shows that even if malignant\ntransformation is uncommon, close follow-up is advised\nafter surgery, to identify precociously the progression of the\ndisease and its potential transformation to a higher grade\nneoplasm; at least a follow-up period of 10 years is\nrecommended [ 15], consisting of sequential thyroglobulin\nmeasurements and total-body scintiscanning with 131I if\nrecurrence is suspected.\nReferences\n1. Weldon-Linne CM, Rushovich AM (1983) Mature cystic teratomas\nwith homunculi. Obstet Gynecol 61:588–594\n2. Campo S, Marone M, Gambadauro P , Garcea N (2000) Laparoscopic\nconservative excision of a rare asymptomatic 11-cm ovarian dermoid\ncyst containing a mandible with seven teeth. Gynaecol Endosc 9:\n65–68\n3. Kabukcuoglu F, Baksu A, Yilmaz B, Aktumen A, Evren I (2002)\nMalignant struma ovarii. Pathol Oncol Res 8:145 –147\n4. Devaney K, Snyder R, Norris H, Tavassoli F (1993) Proliferative\nand histologically malignant Struma Ovarii: a clinicopathologic\nstudy of 54 cases. Int J Gynecol Pathol 12:333 –343\n5. Talerman A (1982) Germ cell tumors of the ovary. In: Kurman RJ\n(ed) Pathology of the female genital tract. Springer, New Y ork,\npp 694 –696\n6. Rosenblum N, LiV olsi V , Edmonds P , Mikuta J (1989) Malignant\nstruma ovarii. Gynecol Oncol 32:224 –227\n7. Ferrario E (1960) Association of ovarian endometriosis and\ndermoid cyst. Minerva Ginecol 12:570 –572\n8. Caruso ML, Pirrelli M (1997) A rare association between ovarian\nendometriosis and bilateral ovarian teratoma. Case report. Minerva\nGinecol 49:341–343\n9. Frederick J, DaCosta V , Winter S, Tenant I, Mckenzie C,\nMcDonald Y (2003) Endometriosis co-existing with bilateral\ndermoid cysts of the ovaries treated by laparoscopy. West Indian\nMed J 52:179 –181\n10. V admal MS, Smilari TF, Lovecchio JL, Klein IL, Hajdu SI (1997)\nDiagnosis and treatment of disseminated struma ovarii with\nmalignant transformation. Gynecol Oncol 64:541 –546\n11. Matsuda K, Maehama T, Kanazawa K (2001) Malignant struma\novarii with thyrotoxicosis. Gynecol Oncol 82:575 –577\n12. Thomas RD, Batty VB (1992) Metastatic malignant struma ovarii.\nTwo case reports. Clin Nuclear Med 17:577 –578\n13. McDougall I, Krasne D, Hanbey J, Collins J (1989) Metastatic\nmalignant struma ovarii presenting as paraparesis from a spinal\nmetastasis. J Nucl Med 30:407 –411\n14. Rotman-Pikielny P , Reynolds JC, Barker WC, Y en PM, Skarulis\nMC, Sarlis NJ (2000) Recombinant human thyrotropin for the\ndiagnosis and treatment of a highly functional metastatic struma\novarii. J Clin Endocrinol Metab 85:237 –244\n15. Makani S, Kim W, Gaba AR (2004) Struma ovarii with a focus of\npapillary thyroid cancer: a case report and review of the literature.\nGynecol Oncol 94:835 –839\nGynecol Surg (2010) 7:27 –29 29","source_license":"CC0","license_restricted":false}