Scientific letter
The first reported case of struma cordis in Spain
El primer caso descrito de struma cordis en España
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] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2530018018301252?idApp=UINPBA00004N" "url" => "/25300180/0000006500000008/v1_201810270629/S2530018018301252/v1_201810270629/en/main.assets" ] "en" => array:15 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Scientific letter</span>" "titulo" => "The first reported case of <span class="elsevierStyleItalic">struma cordis</span> in Spain" "tieneTextoCompleto" => true "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "472" "paginaFinal" => "473" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Cristina María Díaz-Perdigones, Joaquín Cano-Niego, Carmen Hernández-García, José Antonio López-Medina, Sergio González-González" "autores" => array:5 [ 0 => array:4 [ "nombre" => "Cristina María" "apellidos" => "Díaz-Perdigones" "email" => array:1 [ 0 => "cristinnemarie@gmail.com" ] "referencia" => array:3 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] 2 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "Joaquín" "apellidos" => "Cano-Niego" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] ] 2 => array:3 [ "nombre" => "Carmen" "apellidos" => "Hernández-García" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 3 => array:3 [ "nombre" => "José Antonio" "apellidos" => "López-Medina" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 4 => array:3 [ "nombre" => "Sergio" "apellidos" => "González-González" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] ] ] "afiliaciones" => array:3 [ 0 => array:3 [ "entidad" => "Servicio de Endocrinología y Nutrición, Hospital Virgen de la Victoria, Málaga, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "FEA Endocrinología y Nutrición, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Área de Corazón, Hospital El Ángel, Málaga, Spain" "etiqueta" => "c" "identificador" => "aff0015" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "El primer caso descrito de <span class="elsevierStyleItalic">struma cordis</span> en España" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 600 "Ancho" => 950 "Tamanyo" => 64475 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Transthoracic echocardiography showing <span class="elsevierStyleItalic">struma cordis</span> at right ventricle level.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">Struma cordis</span>, or cardiac ectopic thyroid tissue, is a rare condition that affects 1 in every 100,000–300,000 people, and is mostly diagnosed in middle-aged women.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">1</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">The thyroid primordium starts between days 24 and 32 of pregnancy from a thickening of the endodermal epithelium located at the base of the embryonic midline. In turn, the base of the tongue forms and is commonly referred to as the foramen or blind orifice. The appearance of ectopic thyroid tissue is the result of aberrant embryogenesis during migration of the thyroid primordium in the fourth week of pregnancy. Such migration normally takes place from the foramen to the typical thyroid gland location anterior to the second-fourth tracheal cartilage.<a class="elsevierStyleCrossRefs" href="#bib0050"><span class="elsevierStyleSup">1,2</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">According to necropsy studies, the prevalence of ectopic thyroid tissue ranges between 7 and 10%, and the diagnosis is usually established in the first three decades of life. The most common site of appearance is the base of the tongue (in up to 90% of cases), though thyroid remnants may be found over the entire trajectory of the thyroglossal duct, and even in the mediastinum.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">2</span></a> The first case of intrapericardial ectopic thyroid tissue was reported following necropsy in 1941.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">3</span></a> In most reported cases, the thyroid tissue is located in the interventricular septum or right ventricle, with obstruction of the right ventricular outflow tract.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">2,4</span></a></p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Case report</span><p id="par0020" class="elsevierStylePara elsevierViewall">We present the first case of <span class="elsevierStyleItalic">struma cordis</span> described in Spain. The patient was a 28-year-old woman with no relevant disease history or toxic habits, referred to Cardiology for the evaluation of a heart murmur of recent onset.</p><p id="par0025" class="elsevierStylePara elsevierViewall">She had no dyspnea, chest pain or other evidence of heart failure. The physical examination revealed little more than a mild tricuspid systolic murmur at auscultation.</p><p id="par0030" class="elsevierStylePara elsevierViewall">The blood tests and electrocardiographic findings were within normal limits. Transthoracic (TTE) and transesophageal echocardiography (TEE) revealed a 3.5<span class="elsevierStyleHsp" style=""></span>cm<span class="elsevierStyleSup">3</span> broad-pedicled and scantly mobile tumor lesion in the intraventricular septum of the right ventricle, close to the tricuspid valve (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0035" class="elsevierStylePara elsevierViewall">A midline sternotomy was performed, with a right atriotomy and removal of the well delimited mass supported by the interventricular septum. The pathology study revealed the presence of ectopic thyroid tissue with focal fibrosis and dystrophic calcifications. There were no signs of malignancy.</p><p id="par0040" class="elsevierStylePara elsevierViewall">Following the histological findings, a neck ultrasound study was carried out to verify the presence of eutopic thyroid tissue, and thyroid function testing after surgery showed TSH 1.3<span class="elsevierStyleHsp" style=""></span>ml<span class="elsevierStyleHsp" style=""></span>U/l (reference range: 0.25–5). Thyroid scintigraphy revealed a single focus of radiopharmaceutical uptake in the anterocervical region.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Discussion</span><p id="par0045" class="elsevierStylePara elsevierViewall">Although the molecular mechanisms underlying thyroid dysgenesis are not fully understood, mutations of genes <span class="elsevierStyleItalic">TITF-1</span> (<span class="elsevierStyleItalic">Nkx2-1</span>), <span class="elsevierStyleItalic">Foxe1</span> (<span class="elsevierStyleItalic">TITF-2</span>) and <span class="elsevierStyleItalic">PAX-8</span> have a crucial impact upon thyroid morphogenesis and differentiation, and may cause alterations in thyroid migration, as well as conditioning the appearance of ectopic thyroid tissue remnants. The literature on this subject is scarce, though cases with familial aggregation have been reported.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">1</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Most cases of thyroid ectopia are located on the midline of the neck along the trajectory of the thyroglossal duct from the base of the tongue to the diaphragm, and coexist with normal thyroid tissue. The clinical manifestations of thyroid ectopia range from the absence of symptoms to altered thyroid gland function. The most common manifestation of thyroid dysfunction is the appearance of hormonal hypofunction, which in turn is related to the amount of ectopic thyroid tissue. However, symptoms of hyperthyroidism have been reported, even with histological features similar to those of Graves’ disease. Depending on the location and size of the mass, some patients may experience dyspnea, stridor, central chest pressure sensation, palpitations and ventricular arrhythmias, particularly in the case of mediastinal or intracardiac presentations, though such lesions usually constitute incidental findings.<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">4,5</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">If the transthoracic or transesophageal echocardiography findings suggest the presence of ectopic thyroid tissue, a neck ultrasound exploration should be performed on a complementary basis. This will allow us to either confirm or discard the presence of a thyroid gland, with a view to determining that <span class="elsevierStyleItalic">struma cordis</span> is not the only existing thyroid tissue. Radionuclide I-123, I-131 or Tc-99 scintigraphy may be useful for determining the existence of other ectopic thyroid tissue remnants, though the identification of two concomitant ectopic sites is very uncommon.<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">4–6</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">Another suitable option for anatomical and functional characterization in the event of suspected mediastinal thyroid tissue is SPECT-CT. The typical CT image of ectopic thyroid tissue is a well delimited homogeneous mass with an attenuation index of 70<span class="elsevierStyleHsp" style=""></span>±<span class="elsevierStyleHsp" style=""></span>10<span class="elsevierStyleHsp" style=""></span>HU, which suggests a benign nature. In addition, magnetic resonance imaging (MRI) is used, in particular for diagnosing a lingual thyroid gland when there are difficulties in differentiating between thyroid tissue and tongue muscle.<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">4–6</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">The histological study provides the definitive diagnosis of <span class="elsevierStyleItalic">struma cordis</span>. The differential diagnosis is established mainly with metastasis of follicular thyroid carcinoma or primary cardiac tumors such as myxoma.<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">7</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">Surgery is the treatment of choice, with resection of the ectopic tissue using cardiopulmonary bypass techniques, but should be limited to symptomatic patients in view of the high surgical risk involved. Treatment with I-131 may be considered in patients who are not eligible for surgery, or even as an alternative management option in patients who refuse surgery.<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">7</span></a> In other ectopic thyroid tissues, treatment with levothyroxine may reduce the size of the mass. This has not been demonstrated in the case of <span class="elsevierStyleItalic">struma cordis</span>, however.</p><p id="par0075" class="elsevierStylePara elsevierViewall">Thyroid function should be checked after surgery. The recommended timing for this is between the third and sixth week after the operation. In the absence of a eutopic thyroid gland, it is advisable to start levothyroxine at doses of 1.2–1.8<span class="elsevierStyleHsp" style=""></span>μg/kg/day after surgery.<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">8</span></a> The long term prognosis is good if there have been no complications secondary to surgery.<a class="elsevierStyleCrossRefs" href="#bib0080"><span class="elsevierStyleSup">7–9</span></a></p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:4 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Introduction" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Case report" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Discussion" ] 3 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Díaz-Perdigones CM, Cano-Niego J, Hernández-García C, López-Medina JA, González-González S. El primer caso descrito de <span class="elsevierStyleItalic">struma cordis</span> en España. Endocrinol Diabetes Nutr. 2018;65: 438–438.</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" => 600 "Ancho" => 950 "Tamanyo" => 64475 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Transthoracic echocardiography showing <span class="elsevierStyleItalic">struma cordis</span> at right ventricle level.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:9 [ 0 => array:3 [ "identificador" => "bib0050" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Morphological, diagnostic and surgical features of ectopic thyroid gland. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 October | 8 | 1 | 9 |
| 2024 September | 25 | 5 | 30 |
| 2024 August | 27 | 9 | 36 |
| 2024 July | 21 | 4 | 25 |
| 2024 June | 14 | 3 | 17 |
| 2024 May | 16 | 3 | 19 |
| 2024 April | 22 | 5 | 27 |
| 2024 March | 17 | 3 | 20 |
| 2024 February | 26 | 3 | 29 |
| 2024 January | 18 | 5 | 23 |
| 2023 December | 30 | 5 | 35 |
| 2023 November | 13 | 2 | 15 |
| 2023 October | 14 | 1 | 15 |
| 2023 September | 13 | 1 | 14 |
| 2023 August | 14 | 6 | 20 |
| 2023 July | 7 | 3 | 10 |
| 2023 June | 30 | 2 | 32 |
| 2023 May | 67 | 1 | 68 |
| 2023 April | 50 | 1 | 51 |
| 2023 March | 38 | 0 | 38 |
| 2023 February | 13 | 8 | 21 |
| 2023 January | 18 | 4 | 22 |
| 2022 December | 9 | 5 | 14 |
| 2022 November | 28 | 8 | 36 |
| 2022 October | 27 | 8 | 35 |
| 2022 September | 27 | 5 | 32 |
| 2022 August | 22 | 19 | 41 |
| 2022 July | 17 | 8 | 25 |
| 2022 June | 10 | 10 | 20 |
| 2022 May | 17 | 6 | 23 |
| 2022 April | 18 | 7 | 25 |
| 2022 March | 15 | 9 | 24 |
| 2022 February | 22 | 5 | 27 |
| 2022 January | 43 | 3 | 46 |
| 2021 December | 16 | 14 | 30 |
| 2021 November | 34 | 4 | 38 |
| 2021 October | 24 | 9 | 33 |
| 2021 September | 11 | 10 | 21 |
| 2021 August | 5 | 5 | 10 |
| 2021 July | 12 | 8 | 20 |
| 2021 June | 15 | 7 | 22 |
| 2021 May | 29 | 10 | 39 |
| 2021 April | 98 | 16 | 114 |
| 2021 March | 109 | 12 | 121 |
| 2021 February | 33 | 10 | 43 |
| 2021 January | 27 | 11 | 38 |
| 2020 December | 18 | 9 | 27 |
| 2020 November | 19 | 2 | 21 |
| 2020 October | 6 | 10 | 16 |
| 2020 September | 24 | 7 | 31 |
| 2020 August | 22 | 9 | 31 |
| 2020 July | 21 | 11 | 32 |
| 2020 June | 19 | 6 | 25 |
| 2020 May | 11 | 10 | 21 |
| 2020 April | 18 | 11 | 29 |
| 2020 March | 16 | 1 | 17 |
| 2020 February | 15 | 5 | 20 |
| 2020 January | 12 | 2 | 14 |
| 2019 December | 17 | 6 | 23 |
| 2019 November | 11 | 8 | 19 |
| 2019 October | 6 | 1 | 7 |
| 2019 September | 3 | 3 | 6 |
| 2019 August | 2 | 0 | 2 |
| 2019 July | 2 | 21 | 23 |
| 2019 June | 20 | 9 | 29 |
| 2019 May | 53 | 15 | 68 |
| 2019 April | 26 | 3 | 29 |
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