Malignant Struma Ovarii: a Case Report of Laparoscopic Management E

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Gynecologic Oncology 90 (2003) 191Ð194 www.elsevier.com/locate/ygyno Case Report

Malignant struma ovarii: a case report of laparoscopic management E. Volpi,b,* A. Ferrero,b P.G. Nasi,a and P. Sismondib

a Operative Unit of Thyroid Surgery, Umberto I Hospital, Largo Turati 62, Turin, Italy b Department of Gynecologic Oncology, University of Turin, Turin, Italy Received 2 January 2002

Abstract Background. Struma ovarii is a rare disease. Malignant transformation is even rarer. Data about its management are lacking. We describe the first reported case of a malignant struma ovarii treated and staged by laparoscopy. Case. A 49-year-old patient was operated by laparoscopy for a right ovarian teratoma. The patient did not show symptoms of hyperthyroidism. The ovarian teratoma was removed in a plastic bag and definitive hystology showed foci of papillary adenocarcinoma in a struma ovarii. The patient was then staged by laparoscopic sugery undergoing left adnexectomy, multiple peritoneal and omental biopsies, and common iliac and paracaval lymph node sampling. Hysterectomy was not performed. The postoperative course was uneventful and the patient was released on the second day. Thyroglobulin level was monitored and the patient is free of disease after more than 1 year. Conclusion. The preoperative diagnosis of malignant struma ovarii is difficult. Even with cautious evaluation of the patient, some risk of wrong diagnosis is possible. This is why a meticulous technique of laparoscopic surgery in removing the ovary is important. Laparoscopic staging may also intervene in very limited cases; the expertise to perform open staging of the patient is necessary but the postoperative course is fast. © 2003 Elsevier Science (USA). All rights reserved.

Introduction mass. She was still menstruating, was 1.67 m tall, and weighted 85 kg for a body mass index of 30.47. Struma ovarii was first described by Von Kalden in 1895 and The patient did not show any sign of hyperthyroidism Gottschalk in 1899 [1] and is diagnosed when thyroid tissue is and had already had an appendectomy. predominant in a teratoma [2]. Even if malignant transformation is Preoperatively the patient was evaluated with transvag- rarely reported, metastatic disease is possible [3Ð6]. inal ultrasound repeated twice. Sonography revealed a Due to the rarity of malignant transformation which dishomogeneous mass diagnosed as an ovarian teratoma 7 occurs in about 5% of all struma ovarii [7], data about the ϫ 6 ϫ 6 cm. In the cyst a solid area was seen. Ca-125 was management of the tumor are lacking and based on the within the limit range. The patient refused the first sched- experience of ovarian tumor and of thyroid carcinoma. uled procedure in July 2001 and underwent surgery in late We report on a case of thyroid papillary adenocarcinoma October 2001. In the meanwhile the ultrasound scan showed in a struma ovarii which was treated and staged by laparo- no modification. The patient gave consent only to monolat- scopic surgery. eral salpingo-oophorectomy, refusing bilateral adnexectomy. Case report At laparoscopy, right salpingo-oophorectomy was performed. At entry, the right ovary was white and smooth. The M.C., 49 years old, Caucasian, was scheduled for lapa- left ovary and all the abdomen appeared free of disease. roscopic salpingooophorectomy because of a right ovarian Even if peritoneal washing was obtained it was not deliv- ered for cytological examination. Laparoscopic surgery was performed isolating the infundibulopelvic ligament about 4 * Corresponding author. Via Rattazzi 3, 10124, Turin, Italy. Fax: ϩ00390115082683. cm cranial to the ovary and visualizing the ureter. Bipolar E-mail address: [email protected] (E. Volpi). coagulation was used for hemostasis. After coagulation of

Fig. 1. (a, b) Struma ovarii with foci of thyroid papillary carcinoma. the utero-ovarian ligament and of the tube, the mass was lymph node sampling. Hysterectomy was discussed but we extracted by means of an Endo-catch (Auto Suture). A 3-cm decided not to perform it. Four trocars were used for laparos- minilaparotomy was performed and morcellement of the copy; they were positioned in the usual diamond position. No mass was accomplished in the plastic bag. Sebum and hair left upper quadrant trocar was used. The right ancillary trocar were seen and so a frozen section was not asked for. was passed in a different site than the previous extraction port. Pathologic findings were consistent with a diagnosis of The latter was excised and sent for histology. The duration of foci of thyroid papillary carcinoma in a struma ovarii. The the surgery was about 2.5 h (Fig. 2). Definitive histology was right ovarian mass, 6 cm in size, showed a smooth outer negative. Eight lymph nodes were retrieved. surface. Microscopic examination revealed a mature cystic The postoperative course was uneventful and the patient teratoma containing predominantly thyroid tissue. Arising was released after 2 days. No postoperative treatments were within the thyroid tissue a multifocal papillary adenocarci- performed. noma of the thyroid was found. The fallopian tube was Serum thyroglobulin level evaluation and clinical examination unremarkable (Fig. 1). were performed every 4 months for the first year. Thyroglobulin We proposed to the patient a staging procedure and a levels were in the normal range. The patient is free of disease after laparoscopic approach was planned. more than 1 year. Thyroid function is at present normal. Before staging the patient had a complete thyroid function evaluation and thyroid sonography, which were all in the normal range. Thyroglobulin level was 1.7 ng/ml (nor- Discussion mal value between 1.0 and 43.0). The patient underwent a second laparoscopic procedure To the best of our knowledge, this is the first report of with peritoneal washing, left adnexectomy, multiple perito- complete management of a papillary adenocarcinoma in a neal and omental biopsies, and common iliac and paracaval struma ovarii by means of laparoscopy. E. Volpi et al. / Gynecologic Oncology 90 (2003) 191–194 193

Fig. 2. The vena cava, the psoas muscle, and the ureter at the end of the sampling lymphadenectomy.

Even if ultrasound can frequently diagnose dermoid performing a biopsy of the peritoneum underlying the port cysts, it is not useful in showing struma ovarii. However, and also of the scar and subcutaneous tissue. Histology struma ovarii should be remembered, mostly in “solid- showed no disease in the port site. looking” teratomas. Anyhow, malignancy in the struma Difficulties with clinical diagnostic criteria combined ovarii is so rare that by no means can it be ruled out. The with the rarity of this tumor make evaluation of various preoperative diagnosis of struma ovarii is possible in pa- treatments controversial [4]. tients having hyperthyroidism by thyroglobulin measure- After the pathologic findings of a malignant struma ment [8] or by scanning [9], but our patient was totally ovarii, we decided to stage our patients according to ovarian asymptomatic. Furthermore, the reported rate of hyperthy- cancer and thyroid cancer guidelines. Thyroid sonography roidism in patients having struma ovarii is only 7Ð8% [3,5]. was performed, thyroid function was evaluated, and thyro- It is our policy to ask for frozen section analysis in the globulin level was measured as a future marker of disease. diagnosis of suspicious masses during laparoscopic surgery. Even if it has been reported in few cases [12Ð14], we In this case, it was not asked for because the ultrasound of believe that laparoscopic ovarian staging can be performed the patient had remained unchanged for about 4 months and safely in some particular patients. Hysterectomy was an part of the content of the cyst presented as sebum and hair. important source of discussion, since complete staging This may also demonstrate a slow pattern of growth of this would require also this procedure which some authors claim papillary tumor in this patient. to be necessary in these cases [3]. However, we eventually We usually perform bilateral salpingo-oophorectomy in decided to remove the other ovary, to evaluate the perito- a 49-year-old patient. In this case the patient wanted to have neum and the lymph nodes, avoiding hysterectomy. We feel the healthy ovary left. that laparoscopic assisted vaginal hysterectomy would only Some authors [10,11] claim that immediate visual exam- increase the operative time, but not the complexity of sur- ination of the sample by means of the laparoscope can rule gery. out malignancy. We are used, in suspicious cases, to remove In the staging procedure proposed for stage I papillary the ovary with a long pedicle and to avoide spillage as much thyroid carcinoma recognized nodal involvement should be as possible. Removal through a plastic bag in these cases is removed, but selective node removal can be performed and necessary. Since we wanted to be completely sure of the radical neck dissection is not required [15]. According to port site of extraction, even if morcellement was performed this staging procedure and after consultation of our hospi- in the bag, at the second laparoscopy we changed access, tal’s endocrine surgeon, we decided to perform a sampling 194 E. Volpi et al. / Gynecologic Oncology 90 (2003) 191–194 of lymph nodes. In this case, common iliac and paracaval [4] Kouraklis G, Safioleas M, Glinavou A, Xipolitas N, Papachristodou- lymphadenectomy would be difficult since the patient had a lou A, Karatzas G. Struma ovarii: report of two cases and clinical BMI of over 30. The lymph node sampling was performed review. Eur J Surg 2001;167:470Ð1. [5] Devaney K, Snyder R, Tavassoli FA. Proliferative and histologically up to about the origin of the inferior mesenteric artery. malignant struma ovarii: a clinicopathologic study of 54 cases. Int J In these cases, laparoscopic surgery has shown great Gynecol Pathol 1993;12:333Ð43. advantages with respect to open staging since the patient [6] Vadmal MS, Smilari TF, Lovecchio JL, Klein IL, Hajdu SI. Diagno- was released within 2 days. sis and treatment of disseminated struma ovarii with malignant trans- After evaluation of definitive histology and according to formation. Gynecol Oncol 1997;64(3):541Ð6. [7] Rosenblum NG, LiVolsi VA, Edmonds PR, Mikuta JJ. Malignant the advice of the endocrine surgeon, the patient did not struma ovarii. Gynecol Oncol 1989;32(2):224Ð7. 131 receive I therapy. [8] Matsuda K, Maheama T, Kanazawa K. Malignant struma ovarii with Thyroglobulin levels have been shown to be more sen- thyrotoxicosis. Gynecol Oncol 2001;82(3):575Ð7. sitive for recurrent thyroid carcinoma than whole-body di- [9] Joja I, Akurawa T, Mitsumori A, et al. Struma ovarii: appearance at agnostic I131 scanning and thyroglobulin levels have re- MR imaging. Abdominal Imaging 1998;23:652Ð6. placed periodic scanning for monitoring disease status [16]. [10] Canis M, Jardon K, Boulleret C, et al. Management of adnexal tumors: role and risks of laparoscopy. Gynecol Obstet Fertil 2001; Therefore we decided to follow patients with serum thyro- 29:278Ð87. globulin level evaluation. [11] Canis M, Mage G, Pouly JL, Wattiez A, Manhes H, Bruhat MA. In conclusion, we believe that laparoscopic surgery can Laparoscopic dignosis of adnexal cystic tumors: a twelve year expe- be important in removing suspicious ovarian masses but the rience with long term follow-up. Obstet Gynecol 1994;83:707Ð12. technique must be careful and meticulous and, even if lapa- [12] Possover M, Krause N, Plaul K, Kuehne-Heid R, Schneider A. Lapa- roscopic para-aortic and pelvic lymphadenectomy: experience with roscopic staging is not the standard procedure for ovarian 150 patients and review of the literature. Gynecol Oncol 1998;71(1): cancer, it can be accomplished safely and atraumatically in 19Ð28. particular cases of malignant tumors of the ovary. [13] Childers JM, Lang J, Surwitt EA, Hatch KD. Laparoscopic surgical staging of ovarian cancer. Gynecol Oncol 1995;59:25Ð33. [14] Querleu D. Laparoscopic surgical therapy and staging in a case of References early malignant granulosa cell tumor of the ovary. Eur J Obstet Gynecol Biol Reprod 1994;54:215Ð7. [1] Yannopulos D, Yannopulos K, Ossowoski R. Malignant struma ova- [15] Hay ID, Grant CS, Bergstralh EJ, et al. Unilateral total lobectomy: is rii. Pathol Annu 1976;11:410Ð13. it sufficient surgical treatment for patients with AMES low-risk pap- [2] Pardo-Mindan FJ, Vasquez JJ. Malignant struma ovarii. Light and illary thyroid carcinoma? Surgery 1998;124(6):958Ð64. electron microscopic study. Cancer 1983;51(2):337Ð43. [16] Lubin E, Mechlis-Frish Zata S, et al. Serum thyroglobulin and iodine- [3] Dardik RB, Dardik M, Westra W, Montz FJ. Malignant struma ovarii: 131 whole-body scan in the diagnosis and assessment of treatment for two case reports and a review of the literature. Gynecol Oncol metastatic differentiated thyroid carcinoma. J Nuclear Med 1994;35: 1999;73:447Ð51. 257Ð62.