Scientific letter
Synchronous malignant struma ovarii and papillary thyroid carcinoma
Estruma ovárico maligno y cáncer papilar de tiroides sincrónicos
Pablo Fernández Catalina
, Antonia Rego Iraeta, Mónica Lorenzo Solar, Paula Sánchez Sobrino
Corresponding author
Servicio de Endocrinología, Complejo Hospitalario Universitario de Pontevedra, Pontevedra, Spain
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Unilateral oophorectomy was therefore performed during the surgical procedure. The resected ovary weighed 194<span class="elsevierStyleHsp" style=""></span>g and measured 9.5<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>7.5<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>3.5<span class="elsevierStyleHsp" style=""></span>cm. A pathological examination confirmed the presence within a mature cystic teratoma of thyroid tissue representing 20–25% of the tumor. A capillary thyroid carcinoma 2.5<span class="elsevierStyleHsp" style=""></span>cm in largest diameter was seen in the thyroid component (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). There was no histological evidence of vascular, lymphatic, or extraovarian invasion. The results of thyroid function tests, performed before surgery, were normal.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">The patient was referred to the endocrinology department of our hospital. Thyroid ultrasonography was performed, showing a 1.5<span class="elsevierStyleHsp" style=""></span>cm hypoechoic nodule with ill-defined borders, located in the left thyroid lobe; fine needle aspiration (FNA) of the thyroid nodule was consistent with a benign follicular nodule. However, because of the ultrasonographic features of the thyroid lesion and to achieve adequate radioiodine (<span class="elsevierStyleSup">131</span>I) uptake by the primary tumor, total thyroidectomy was performed. The surgical specimen weighed 14<span class="elsevierStyleHsp" style=""></span>g, measured 7.1<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>2.5<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>2.2<span class="elsevierStyleHsp" style=""></span>cm, and contained a poorly delimited nodule 1.4<span class="elsevierStyleHsp" style=""></span>cm in largest diameter. The thyroid tumor turned out to be a classical papillary thyroid carcinoma with metastases to two lymph nodes and invading the perithyroidal soft tissue.</p><p id="par0020" class="elsevierStylePara elsevierViewall">The administration of a dose of 100<span class="elsevierStyleHsp" style=""></span>mCi of radioiodine (<span class="elsevierStyleSup">131</span>I) was decided upon, and a subsequent whole body scan (WBS) revealed strong uptake in the thyroid bed, with no evidence of locoregional or distant pathological uptake. Suppressive therapy with levothyroxine was subsequently started. One year later, due to the persistence of a minimum residue in the neck area in a new WBS and detectable plasma thyroglobulin levels (8.6<span class="elsevierStyleHsp" style=""></span>ng/mL) concurrent with elevated TSH levels (99.4<span class="elsevierStyleHsp" style=""></span>IU/mL) and negative thyroglobulin antibodies (<60<span class="elsevierStyleHsp" style=""></span>IU/mL), an additional dose of 100<span class="elsevierStyleHsp" style=""></span>mCi of radioiodine (<span class="elsevierStyleSup">131</span>I) was administered. The complete ablation of the thyroid remnant and the absence of abnormal extrathyroid uptake were verified during follow-up. Plasma thyroglobulin levels currently remain undetectable with stimulated plasma TSH levels. In addition, there is no evidence of tumor relapse six years after the initial diagnosis of malignant struma ovarii.</p><p id="par0025" class="elsevierStylePara elsevierViewall">The coexistence of differentiated thyroid cancer and malignant struma ovarii in the same patient is an exceptional finding in clinical practice.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a> Such synchronous occurrence raises the dilemma of whether the tumors are separate primary neoplasms coinciding in time, or whether one of them results from the distant metastatic dissemination of the other. Although this dilemma may be difficult to resolve, a number of phenotypic variables of the tumor and clinical data may be helpful in this regard.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Phenotypic variables include the morphological, immunohistochemical, and molecular characteristics of both neoplasms. In the reported case, both tumors showed morphological characteristics of a classical papillary thyroid cancer with a papillary and follicular pattern. The immunohistochemical pattern with HBME-1 (antibody against the microvillous surface of mesothelial cells), CK-19 (cytokeratin 19 of high molecular weight), and galectin-3 may also be of help in tumor characterization, but in our case the only difference shown was the absence of a positive result for galectin-3 in the thyroid neoplasm and a focal positive result for galectin-3 in the ovarian tumor. While the expression of some oncogenes such as BRAF, RAS, or RET/PTC has not been shown to be useful for characterizing synchronous primary tumors, the study of the clonal origin of these tumors by analyzing the differences in length of the polyglutamine tract of the human androgen receptor may be helpful.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">The following clinical variables have been used to establish the difference between synchronous primary tumors and differentiated metastatic disease: the dissemination pattern in WBS with radioiodine (<span class="elsevierStyleSup">131</span>I) or positron emission tomography-computed tomography (PET-CT), stimulated plasma thyroglobulin levels before radioablation, histological characteristics, and uni or bilateral ovarian tumor, as well as favorable or unfavorable prognosis during the course of the disease.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Our patient had only left ovary involvement with a tumor of teratomatous characteristics; WBS after ablation therapy with radioiodine (<span class="elsevierStyleSup">131</span>I) only showed uptake in the cervical bed, but no advanced metastatic disease; finally, thyroglobulin levels before thyroid ablation with radioiodine were not excessively high (16<span class="elsevierStyleHsp" style=""></span>g/mL). After six years of clinical monitoring, the patient remains free of tumor recurrence. All of the foregoing suggests that this case had the clinical characteristics of two synchronous primary tumors.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Although the therapeutic management of malignant struma ovarii is controversial, it is advisable to perform neck ultrasonography and thyroid FNA if any thyroid nodule is detected. Total thyroidectomy is recommended if there is any suspicion of a malignant thyroid nodule.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">The synchronous presence of malignant struma ovarii and papillary thyroid cancer is usually associated with a favorable prognosis, unlike in the presence of metastatic disease having one or the other origin. The main barrier for making recommendations on the clinical management of these synchronous tumors is the rarity of their coexistence and the lack of sound scientific evidence.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Fernández Catalina P, Rego Iraeta A, Lorenzo Solar M, Sánchez Sobrino P. Estruma ovárico maligno y cáncer papilar de tiroides sincrónicos. Endocrinol Nutr. 2016;63:366–367.</p>" ] ] "multimedia" => array:1 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 742 "Ancho" => 990 "Tamanyo" => 135651 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Histological image of mature cystic teratoma showing the typical nuclear characteristics of papillary thyroid cancer, as well as positive TTF1 (thyroid transcription factor-1).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0030" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "<span class="elsevierStyleItalic">Struma ovarii</span> con histología maligna: un caso con carcinoma papilar de tiroides" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "W. 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Angelos" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.4158/EP.15.2.167" "Revista" => array:6 [ "tituloSerie" => "Endocr Pract" "fecha" => "2009" "volumen" => "15" "paginaInicial" => "167" "paginaFinal" => "173" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/19289330" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/21735093/0000006300000007/v2_201609300106/S2173509316300666/v2_201609300106/en/main.assets" "Apartado" => array:4 [ "identificador" => "5823" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Scientific letters" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/21735093/0000006300000007/v2_201609300106/S2173509316300666/v2_201609300106/en/main.pdf?idApp=UINPBA00004N&text.app=https://www.elsevier.es/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2173509316300666?idApp=UINPBA00004N" ]
| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 October | 26 | 3 | 29 |
| 2024 September | 67 | 11 | 78 |
| 2024 August | 48 | 9 | 57 |
| 2024 July | 37 | 2 | 39 |
| 2024 June | 36 | 5 | 41 |
| 2024 May | 26 | 8 | 34 |
| 2024 April | 27 | 19 | 46 |
| 2024 March | 44 | 8 | 52 |
| 2024 February | 53 | 12 | 65 |
| 2024 January | 33 | 7 | 40 |
| 2023 December | 39 | 7 | 46 |
| 2023 November | 52 | 15 | 67 |
| 2023 October | 39 | 17 | 56 |
| 2023 September | 39 | 6 | 45 |
| 2023 August | 20 | 3 | 23 |
| 2023 July | 18 | 10 | 28 |
| 2023 June | 28 | 6 | 34 |
| 2023 May | 33 | 4 | 37 |
| 2023 April | 34 | 4 | 38 |
| 2023 March | 40 | 3 | 43 |
| 2023 February | 19 | 8 | 27 |
| 2023 January | 29 | 6 | 35 |
| 2022 December | 46 | 5 | 51 |
| 2022 November | 38 | 5 | 43 |
| 2022 October | 36 | 13 | 49 |
| 2022 September | 38 | 9 | 47 |
| 2022 August | 49 | 11 | 60 |
| 2022 July | 19 | 7 | 26 |
| 2022 June | 27 | 12 | 39 |
| 2022 May | 14 | 18 | 32 |
| 2022 April | 39 | 8 | 47 |
| 2022 March | 37 | 8 | 45 |
| 2022 February | 54 | 3 | 57 |
| 2022 January | 52 | 7 | 59 |
| 2021 December | 52 | 12 | 64 |
| 2021 November | 46 | 13 | 59 |
| 2021 October | 67 | 19 | 86 |
| 2021 September | 43 | 15 | 58 |
| 2021 August | 46 | 10 | 56 |
| 2021 July | 32 | 9 | 41 |
| 2021 June | 37 | 13 | 50 |
| 2021 May | 62 | 14 | 76 |
| 2021 April | 91 | 19 | 110 |
| 2021 March | 77 | 16 | 93 |
| 2021 February | 41 | 10 | 51 |
| 2021 January | 52 | 24 | 76 |
| 2020 December | 47 | 23 | 70 |
| 2020 November | 82 | 21 | 103 |
| 2020 October | 47 | 10 | 57 |
| 2020 September | 61 | 18 | 79 |
| 2020 August | 43 | 5 | 48 |
| 2020 July | 37 | 11 | 48 |
| 2020 June | 20 | 25 | 45 |
| 2020 May | 48 | 15 | 63 |
| 2020 April | 48 | 11 | 59 |
| 2020 March | 29 | 17 | 46 |
| 2020 February | 59 | 23 | 82 |
| 2020 January | 49 | 5 | 54 |
| 2019 December | 26 | 12 | 38 |
| 2019 November | 17 | 12 | 29 |
| 2019 October | 28 | 0 | 28 |
| 2019 September | 27 | 9 | 36 |
| 2019 August | 24 | 8 | 32 |
| 2019 July | 36 | 29 | 65 |
| 2019 June | 35 | 21 | 56 |
| 2019 May | 112 | 18 | 130 |
| 2019 April | 41 | 18 | 59 |
| 2019 March | 12 | 4 | 16 |
| 2019 February | 15 | 8 | 23 |
| 2019 January | 13 | 4 | 17 |
| 2018 December | 7 | 22 | 29 |
| 2018 November | 20 | 13 | 33 |
| 2018 October | 19 | 7 | 26 |
| 2018 September | 35 | 5 | 40 |
| 2018 August | 10 | 6 | 16 |
| 2018 July | 11 | 2 | 13 |
| 2018 June | 12 | 3 | 15 |
| 2018 May | 8 | 4 | 12 |
| 2018 April | 23 | 2 | 25 |
| 2018 March | 17 | 4 | 21 |
| 2018 February | 20 | 4 | 24 |
| 2018 January | 20 | 1 | 21 |
| 2017 December | 19 | 0 | 19 |
| 2017 November | 19 | 1 | 20 |
| 2017 October | 22 | 3 | 25 |
| 2017 September | 15 | 1 | 16 |
| 2017 August | 18 | 3 | 21 |
| 2017 July | 23 | 2 | 25 |
| 2017 June | 24 | 2 | 26 |
| 2017 May | 39 | 1 | 40 |
| 2017 April | 29 | 2 | 31 |
| 2017 March | 31 | 1 | 32 |
| 2017 February | 51 | 3 | 54 |
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