{"paper_id":"309a8c2b-c7e8-44e9-b8bd-8b2a27ed40e6","body_text":"Archives of the Balkan Medical Union\nCopyright © 2019 Balkan Medical Union\nvol. 54, no. 2, pp. 368-371\nJune 2019\nRÉSUMÉ\nStruma ovarii chez une femme de 56 ans – rapport \nde cas\nIntroduction. Struma ovarii représente une tumeur \nrare, seulement 1% des tumeurs de l’ovaire, avec une \nincidence de 0,3 à 0,7%. Le diagnostic positif est ob-\ntenu par examen microscopique; parfois, des taux sé-\nriques élevés d’hormones thyroïdiennes, de CA 125 et \nd’échographies peuvent suggérer un diagnostic préopé-\nratoire.\nRapport du cas. N o u s  r a p p o r t o n s  l e  c a s  d ’ u n e  \nfemme de 56 ans avec une struma ovarii unilatérale \net aucune preuve clinique ou paraclinique du diagnos-\ntic à venir. L’échographie transvaginale a révélé un \nutérus poly-fibromateux et des annexes apparemment \nABSTRACT\nIntroduction. Struma ovarii represents a rare tumor, \nonly 1% of the ovarian tumors, with an incidence of \n0.3-0.7%. The positive diagnosis is obtained by micro-\nscopic examination; sometimes elevated serum levels \nof thyroid hormone, CA 125 and ultrasound aspects \ncan suggest the preoperative diagnosis.\nCase presentation. W e  r e p o r t  t h e  c a s e  o f  a  \n56-year-old woman with unilateral struma ovarii and \nno clinical or paraclinical evidence of the diagnosis to \ncome. Transvaginal ultrasound revealed polyfibroma-\ntous uterus and apparently normal adnexa. Partial \nhysterectomy with bilateral salpingo-oophorectomy \n(considering the patients age) through laparotomy was \nperformed under spinal anaesthesia. The histopatho-\nlogical result showed an endometrial polyp, multiple \nCASE REPORT\nSTRUMA OVARII IN A 56-YEAR-OLD WOMAN – \nA CASE REPORT\nAndra M. IONESCU1, Bogdan SOCEA2 , Mihai C.T. DIMITRIU3, Vlad D. CONSTANTIN2, \nCringu A. IONESCU3, Alexandra MATEI3, Diana C. GHEORGHIU1, Irina PACU3, \nTeodora VLADESCU4, Mihai B. NICULAE5\n1 Emergency Clinical Hospital „Sf. Pantelimon “ , Obstetrics and Gynecology Department, Bucharest, \nRomania\n2 Emergency Clinical Hospital „Sf. Pantelimon“, General Surgery Clinic, „Carol Davila“ University of \nMedicine and Pharmacy, Bucharest, Romania\n3 Emergency Clinical Hospital „Sf. Pantelimon“, Obstetrics-Gynecology and Neonatology, „Carol Davila“ \nUniversity of Medicine and Pharmacy, Bucharest, Romania\n4 Emergency Clinical Hospital “Sf. Pantelimon“, Pathology Department, Bucharest, Romania\n5 „N. Gh. Lupu“ Clinical Hospital, Pathology Department, Bucharest, Romania\nReceived 02 Apr 2019, Accepted 17 May 2019\nhttps:/ /doi.org/10.31688/ABMU.2019.54.2.24\n Address for correspondence:   Bogdan SOCEA\nEmergency Clinical Hospital “Sfântul Pantelimon“ General Surgery Clinic, \nBucharest, Romania\nAddress: Soseaua Pantelimon no. 340-342, 1st floor, General Surgery \nDepartment, Bucharest, Romania\nEmail bogdansocea@gmail.com;Phone: +40788491091; Fax: \n+40212550064\n\nArchives of the Balkan Medical Union\nJune 2019 / 369\nINTRODUCTION\nStruma ovarii is a mono-dermal teratoma com-\nposed predominantly or solely of thyroid tissue1. This \ntype of tumor accounts for 3% of ovarian teratomas2. \nIt is encountered in the reproductive age, with most \npatients being in the fifth decade of life\n1,3. It is found \nalmost always unilaterally, in only 10% and 15% be-\ning bilateral\n3. It is most commonly asymptomatic, sel-\ndom clinically discernable as a palpable pelvic mass \nand even less frequently associated with ascites (1/3) \nor Meigs syndrome\n1,2. On some occasions, hyperthy-\nroidism is present 2. High serum levels of CA – 125 \nhave been reported 2. At ultrasound examination, \nit can appear complex and nonspecific 2,3. However, \nlow-resistance blood flow or good vascularized solid \ncomponent in the central portion are important clues \nfor the diagnosis of struma ovarii.\nOn gross examination, the tumor has a color \nthat can vary from red to brown and green and a size \nof no more than 10 cm\n1. It consists of predominantly \nsolid, soft tissue 4.\nLike the thyroid tissue it emulates, struma ovarii \nis made of hundreds of thousands of follicles lined by \ncuboidal or low columnar epithelium\n5. Cytology is \nusually with minimal atypia and low mitotic activity 2. \nOne can encounter variants such as the microfollicu-\nlae, the pseudotubular or the solid pattern. The latter \ncan be composed of oxyphilic (abundant eosinophilic \ncytoplasm) or clear cells (pale cytoplasm).\nAssociation with dermoid cysts, mucinous tu-\nmours or Brenner tumours has been described\n2. \nMalignant change in struma ovarii most commonly \nleads to papillary or follicular carcinoma, but it is \nstill unclear whether the criteria used in the thyroid \ngland should be applied\n1,2. Thyroglobulin and TTF1 \nare essential immunohistochemical markers for a \nstruma diagnosis, with the former being more spe-\ncific\n4. When suspecting a carcinoid, additional stains \nsuch as chromogranin or synaptophysin are useful in \ndifferentiating the two entities\n3. Histologic heteroge-\nneity makes the differential diagnosis more difficult. \nThe most important neoplasm a pathologist should \nexclude are ovarian cystadenoma (when confronted \nwith a cystic struma), steroid cell tumours, carcinoid \ntumours, Sertoli-Leydig cell tumours, renal clear cell \ncarcinoma, and metastatic melanoma (in case of ox-\nyphilic struma)\n2,4.\nDifferential diagnoses include: clear cell carcino-\nma (primary or metastatic from the kidney), primary \nor secondary hydatid cyst\n6, metastatic tumors from \nrenal sarcoma 7, endometrioid carcinomas, Sertoli \ncell tumour, hepatoid yolk sac tumour, malignant \nmelanoma, serous cystadenoma, pregnancy lutcomas, \nmetastatic thyroid carcinoma of the ovary\n2,4, second-\nary tumors from retroperitoneum8,9, GIST tumors10,11, \nprimary12 or secondary carcinoid tumors, tumoral or \nbenign appendiceal pathology 13,14.\nMost often, the prognosis is favourable, with \ntypical struma ovarii being benign 1. Even in the \nsmall percentage of histologically malignant results – \n5-10%, the outcome is favourable, with only few pa-\ntients who die of this disease\n1,2. The strumal com-\nponent, abundant ascites, adhesions and defects of \nthe ovarian serosa are correlated with the malignant \ntype\n1. Only half of the malignant tumors associate \nextension beyond the ovaries 2.\nnormales. Une hystérectomie partielle avec salpin-\ngo-ovariectomie bilatérale (compte tenu de l’âge du \npatient) par laparotomie a été réalisée sous anesthésie \nrachidienne. Le résultat histopathologique a montré \nun polype de l’ endomètre, de multiples léiomyomes \nintra-utérins, un ovaire gauche avec des follicules thy-\nroïdiens, qui ressemblait à un tissu thyroïdien normal. \nL ’évolution de la patiente était favorable, sans compli-\ncation au recul de 6 mois.\nConclusions. Dans la littérature, il n’y a que quelques \ncas de struma ovarii bénigne et encore moins de cas de \nstruma ovarii maligne ou de présentation bilatérale de \nla tumeur. La particularité du cas consiste dans le dia-\ngnostic de struma ovarii seulement après une poussée, \nchez une patiente sans preuve clinique ou paraclinique \nde ce diagnostic.\nMots-clés: struma ovarii, tumeur ovarienne, ma-\nligne.\nintrauterine leiomyomas, left ovary with thyroid fol-\nlicles, which looked like normal thyroid tissue. The \nevolution of the patient was favorable, with no compli-\ncations at the 6 months’ follow-up.\nConclusions. In the literature, there are only a few \ncases of benign struma ovarium, and even less cases \nof malignant struma ovarium or bilateral presentation \nof the tumor. The particularity of the case consists in \nthe diagnosis of struma ovarii only after surgery, in a \npatient without clinical or paraclinical evidence of this \ndiagnosis.\nKeywords: struma ovarii, ovarian tumor, malignant.\n\nStruma ovarii in a 56-year-old woman – a case report – IONESCU et al\n370 / vol. 54, no. 2\nThe recommended treatment is oophorectomy \nand, in case of malignant struma, extraovarian tumor \nremoval is advised\n2.\nPatients are advised to have a long-term fol-\nlow-up2.\nThe aim of this case report is to show the diag-\nnostic stages and treatment of a rare case of struma \novarii, which represents no more than 1% of all ovar-\nian tumours and 3% of all dermoid tumors\n1.\nCASE PRESENTATION\nA 56-year-old woman was admitted to the \nDepartment of Obstetrics and Gynaecology of “St. \nPantelimon“ Emergency Clinical Hospital, Bucharest, \nRomania, on November 2018, for persistent vagi-\nnal bleeding during menopause. From the patient’s \npersonal history, we noted one birth through \nC-section in 1994, the occurrence of menopause at \nthe age of 51 years, obesity stage II, fibroadenoma \nof the left breast with surgical intervention in 2000, \nhypercholesterolemia, hypertriglyceridemia, smoker \n(20 cigarettes/day). Dilatation and uterine curettage \nwere performed in May 2018, with the histological \nresult of endometrial polyp.\nOn admission in our department, the patient \nwas cooperative, the blood pressure was 120/75 \nmmHg, heart rate 73 beats/minute, with moderate \nvaginal bleeding. The gynaecological examination \nwith the speculum showed no macroscopic lesions \non the cervix and moderate bleeding coming from \nthe uterine cavity. On the bimanual examination, \nthe uterus was firm, with increased volume, irregular \ncontour, mobility was preserved, sensitive at mild pal-\npation, bilateral adnexa were normal. Laboratory ex-\nams were in normal range. Transvaginal ultrasound \nwas performed and revealed a polyfibromatous uterus \nand apparently normal adnexa.\nThe informed consent was obtained and explora-\ntory surgery through laparotomy was performed under \nspinal anaesthesia (spinal block/ intradural block/ in-\ntrathecal block) and followed by partial hysterectomy \nFigure 1. Thyroid tissue: intraovarian colloid. \nHE staining, x4.\nFigure 2. Ovary with corpus albicans \nand thyroid follicles. HE staining, x10.\nFigure 3. Thyroid follicles in the ovary. HE staining, x20. Figure 4. Struma ovarii – thyroid follicles. \nHE staining, x4.\n\nArchives of the Balkan Medical Union\nJune 2019 / 371\nwith bilateral salpingo-oophorectomy, considering the \nage and examinations prior to the surgery.\nThe postoperative evolution was favourable, \nwithout any complications. On day 7, the patient was \nreleased from the hospital with good general condi-\ntion and afebrile.\nThe histopathological result showed an endome-\ntrial polyp with glands composed of unistratificate \ncolumnar epithelia, some of them cystically dilated, \nmultiple intrauterine leiomyomas, left ovary which \nshowed lobular display and consisted of thyroid fol-\nlicles, which looked like normal thyroid tissue. The \ncells were cuboidal to columnar and dense colloid is \nseen inside the follicles (Figures 1-4). The right ovary \nwas sclera hyalinized and both salpinges were atrophic.\nAt the 6 months’ follow-up of the patient no \ncomplications occurred and the laboratory investiga-\ntions were within normal limits.\nDISCUSSION\nThe clinical diagnosis of struma ovarii is dif-\nficult due to the rarity of the disease. Most of the \ntimes the patient is asymptomatic, with no abnormal \nparaclinical investigations. This is the case of our pa-\ntient, who had normal serum levels of the thyroid \nhormones or CA 1 25 , with normal vaginal ultra-\nsound of the ovaries and no ascites. If the preopera-\ntive diagnosis occurs, laparotomy surgery is advised, \ndue to the risk of tumor rupture intra-abdominally \nwith dissemination, in case of a malignant tumor.\nOophorectomy is preferred and in case the malig-\nnant struma ovarii is confirmed, second intervention \nis planned and scheduled for pelvic and para-aortic \nlymph nodes sampling, peritoneal cytologic washing, \npartial or total omentectomy. Thyroidectomy and \nfollow-up with Iodine-131 whole body scanner are rec-\nommended. In some cases, fertility can be preserved \nas needed and follow-up with the intervention as ex-\nplained earlier.\nThe postoperative management of the patients \nafter surgery is difficult, due to the rarity of the cases. \nBoth guidelines of ovarian cancer and thyroid cancer \nare used.\nCONCLUSIONS\nThe treatment of choice has to be carefully select-\ned because of the high possibility of clinically misdiag-\nnosing this pathology. If the diagnosis is established, \nlaparotomy is advised due to the better accessibility and \nmanipulation of the tumor by the surgeon. Fertility \ncan be preserved, but caution must be implied. The \nmajority of the patients has a good outcome.\nCompliance with Ethics Requirements:\n„The authors declare no conflict of interest regarding \nthis article“\n„The authors declare that all the procedures and ex-\nperiments of this study respect the ethical standards in the \nHelsinki Declaration of 1975, as revised in 2008(5), as \nwell as the national law. Informed consent was obtained \nfrom the patient included in the study“\n„No funding for this study“\nREFERENCES\n1. Kurman RJ, Carcangiu ML, Herrington CS, Young RH.  \nWHO – Classification of Tumours of Female Reproductive \nOrgans, V o l um e 6, in WH O C lass ifi ca ti o n o f T um o rs,  \nWHO ed., 2014.\n2. Nucci MR, Oliva E – series editor  Goldblum JR. \nGynecologic Pathology. In Foundations in Diagnostic \nPathology, Elsevier, 2009.\n3. Crum C, Hirsch M, Peters III W ,  Quick CM, Laury A. \nHigh Yield Pathology, Gynecologic and Obstetric Pathology \nVolume, Elsevier, 2015.\n4. Clement P, Stall J, Young R. Atlas of Gynecologic Surgical \nPathology, 4th Edition, Elsevier, 2019.\n5. Ross MH, Pawlina W. Histology – A Text And Atlas, sixth \nedition, Lippincott Williams & Wilkins,  2011.\n6. Iorga L, Anghel R, Marcu D, et al. Primary renal hydatid \ncyst – a review. J Mind Med Sci . 2019; 6(1): 47-51.\n7. Iorga L, Anghel R, Marcu D, et al. 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