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Documento de Consenso de la Comisión de Otoneurología Sociedad Española de Otorrinolaringología y Cirugía de Cabeza y Cuello" ] ] "contieneResumen" => array:2 [ "en" => true "es" => true ] "contieneTextoCompleto" => array:1 [ "en" => true ] "contienePdf" => array:1 [ "en" => true ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0035" "etiqueta" => "Figure 7" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr7.jpeg" "Alto" => 1750 "Ancho" => 2333 "Tamanyo" => 150297 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0045" class="elsevierStyleSimplePara elsevierViewall">Gufoni manoeuvre for ageotropic nystagmus/Appiani, for cupulolithiasis of the anterior arm of the horizontal canal (right side). We start with the patient sitting on the edge of the bed. 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"apellidos" => "Lopez-Escamez" ] ] ] ] ] "idiomaDefecto" => "en" "Traduccion" => array:1 [ "es" => array:9 [ "pii" => "S0001651917301358" "doi" => "10.1016/j.otorri.2017.05.001" "estado" => "S300" "subdocumento" => "" "abierto" => array:3 [ "ES" => true "ES2" => true "LATM" => true ] "gratuito" => true "lecturas" => array:1 [ "total" => 0 ] "idiomaDefecto" => "es" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0001651917301358?idApp=UINPBA00004N" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2173573518300796?idApp=UINPBA00004N" "url" => "/21735735/0000006900000006/v1_201811180608/S2173573518300796/v1_201811180608/en/main.assets" ] "en" => array:18 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Case study</span>" "titulo" => "Low Grade Mucoepidermoid Carcinoma of a Minor Salivary Gland of the Tongue in a Paediatric Patient" "tieneTextoCompleto" => true "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "367" "paginaFinal" => "369" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "María Fernanda Vargas Gamarra, José María Perolada Vilmaña, Miguel Armengot Carceller" "autores" => array:3 [ 0 => array:4 [ "nombre" => "María Fernanda" "apellidos" => "Vargas Gamarra" "email" => array:1 [ 0 => "mariafernandavargasg@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "José María" "apellidos" => "Perolada Vilmaña" ] 2 => array:2 [ "nombre" => "Miguel" "apellidos" => "Armengot Carceller" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Departamento de Otorrinolaringología, Hospital Universitari i Politecnic La Fe, Valencia, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Carcinoma mucoepidermoide de bajo grado de glándula salival menor de lengua en un paciente pediátrico" ] ] "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" => 1132 "Ancho" => 2500 "Tamanyo" => 211129 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Cervicofacial CAT performed in the emergency department, reporting: boil on the floor of the mouth with areas of abscessification. (B) PET/CT: soft tissue lesions of 49×31×29<span class="elsevierStyleHsp" style=""></span>mm in diameter AP, Ax and <span class="elsevierStyleSmallCaps">CC</span>, located at the tongue base and extending towards the retrohyoid region. Mild metabolic activity with very uneven distribution, SUV<span class="elsevierStyleInf">max</span> of 4.1<span class="elsevierStyleHsp" style=""></span>g/ml, not ruling out malignancy.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">We present the case of a 13-year-old female patient who attended the emergency department with a 3-day history of odynophagia, fever and dyspnoea. Oropharyngoscopy revealed no significant findings. A deep neck space infection was suspected and a cervicofacial CT was performed, which reported a boil at the base of the tongue with small areas of necrosis (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A). A few hours later, after the patient's dyspnoea had worsened and there was no improvement with antibiotics or intravenous corticosteroids we surgically drained the boil using a cervical approach, a retrohyoid mass was seen of solid features with small areas of abscessification. A biopsy was taken of the lesion without entering the pharyngeal cavity and then we then performed a regulated tracheotomy. The laboratory report was inflammatory sialadenitis. The study was extended to viral serologies which were negative for an active infectious process.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">Because there was little improvement after intravenous treatment we requested a PET/CT (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>B), which showed a mass of a tumour-like appearance 49×31×29<span class="elsevierStyleHsp" style=""></span>mm in diameter at the base of the tongue, with low metabolic activity, and no clear features of malignancy.</p><p id="par0015" class="elsevierStylePara elsevierViewall">With no definitive diagnosis and given the uncertainty of whether this was a malignant lesion we decided to perform a cervicotomy for removal of the lesion. The hyoid bone was resected, entering the pharynx via the vallecula and extracting the lesion that was compromising a large part of the tongue base. The hypoglossal nerves and lingual arteries were preserved. The intraoperative biopsy reported an epithelial tumour with no clear signs of malignancy.</p><p id="par0020" class="elsevierStylePara elsevierViewall">The final lab report showed a proliferation of irregular salivary glands arranged at random with intermediate cells, with “lakes” of mucus and inflammatory infiltrate, CK7 stain positive (epithelial cell marker), concluding a low-grade minor salivary gland mucoepidermoid carcinoma (MEC) at the tongue base (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>) In retrospect, we can conclude that the initial biopsy, although broad, was not sufficiently deep, therefore the tissue analysed corresponded to peritumoural inflammation.</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">The peritumoural musculature was infiltrated by the lesion, therefore postoperative radiotherapy (RT) was decided (54<span class="elsevierStyleHsp" style=""></span>Gy).</p><p id="par0030" class="elsevierStylePara elsevierViewall">Eighteen months after completion of treatment the patient is free from disease.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Discussion</span><p id="par0035" class="elsevierStylePara elsevierViewall">Salivary gland MEC is a rare cancer in children, although it is the most common malignant salivary tumour in this age group, particularly in adolescents.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">We present the case of a 13-year-old female patient with a low-grade MEC of the base of the tongue, we show how a diagnosis was reached, the treatment given, and the patient's progress. The case's interest lies in the complexity and rareness of its location, which gave rise to major diagnostic and therapeutic controversy. In the literature reviewed we found few case reports of this disease, the minor salivary glands of the palate are the most frequently affected, and we found no case of the tongue base in children.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Only 5% of salivary gland tumours occur in children and adolescents.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">2</span></a> Those located in the tongue base are a rarity.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a> The case we present exemplifies the diagnostic and therapeutic difficulties of this location.</p><p id="par0050" class="elsevierStylePara elsevierViewall">In tumours of the tongue, the most common symptom is dysphagia, as in our case, with or without odynophagia and pharyngeal voice depending on how advanced and aggressive the tumour. Although the aetiology is not known, previous RT to the head and neck and chemotherapy (CT) are risk factors for malignant tumours of the salivary glands.<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">3,4</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">Diagnosis is histopathological and requires special stains to check mucin production and to differentiate it from epidermoid carcinomas.<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">1,3</span></a> This makes diagnosis difficult, especially in cases of low-grade tumours.</p><p id="par0060" class="elsevierStylePara elsevierViewall">Salivary gland MEC are treated surgically. The benefit of postoperative CT and/or RT has not been established. However there is consensus that patients with affected margins, a high histological grade and advanced stage (positive lymph nodes or remote metastases) will benefit from postoperative RT.<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">4,5</span></a> In our patient, the site, tumour size and the affected margins were determining factors for prescribing postoperative RT.</p><p id="par0065" class="elsevierStylePara elsevierViewall">Survival at 5 years is very variable according to the tumour grade: from 92% to 100% for low-grade tumours, 63% for intermediate grade, while for high-grade tumours survival is poor, at 49% at 5 years.</p><p id="par0070" class="elsevierStylePara elsevierViewall">Long-term follow-up is necessary, since recurrences have been described up to 15–20 years after resection of the tumour.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a></p><p id="par0075" class="elsevierStylePara elsevierViewall">Minor salivary gland MEC is a rare tumour in childhood, it should be considered in lesions of the oral cavity and oropharynx that do not improve with conservative treatment, or if they present in patients who have been previously treated with CT and/or RT. The clinical symptoms are non-specific and imaging studies can lead to confusion with other non-malignant diseases, the same applies to histopathological study. Surgery with complete resection is the treatment standard, reserving complementary treatment, essentially with RT, for situations associated with factors of poor prognosis.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Conflict of Interests</span><p id="par0080" class="elsevierStylePara elsevierViewall">The authors have no conflict of interests to declare.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:4 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Discussion" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Conflict of Interests" ] 2 => array:2 [ "identificador" => "xack376529" "titulo" => "Acknowledgements" ] 3 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2017-05-21" "fechaAceptado" => "2017-08-08" "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Vargas Gamarra MF, Perolada Vilmaña JM, Armengot Carceller M. Carcinoma mucoepidermoide de bajo grado de glándula salival menor de lengua en un paciente pediátrico. Acta Otorrinolaringol Esp. 2018;69:367–369.</p>" ] ] "multimedia" => array:2 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1132 "Ancho" => 2500 "Tamanyo" => 211129 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Cervicofacial CAT performed in the emergency department, reporting: boil on the floor of the mouth with areas of abscessification. (B) PET/CT: soft tissue lesions of 49×31×29<span class="elsevierStyleHsp" style=""></span>mm in diameter AP, Ax and <span class="elsevierStyleSmallCaps">CC</span>, located at the tongue base and extending towards the retrohyoid region. Mild metabolic activity with very uneven distribution, SUV<span class="elsevierStyleInf">max</span> of 4.1<span class="elsevierStyleHsp" style=""></span>g/ml, not ruling out malignancy.</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 2828 "Ancho" => 3167 "Tamanyo" => 1059942 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Pathological anatomy: low-grade salivary gland mucoepidermoid carcinoma: (A–C) Eosin haematoxylin stain: proliferation of uneven lands with cystic dilatation full of mucus. (D) Positive CK7 stain (epithelial cell marker).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0030" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:3 [ "comentario" => "78:175–181" "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Salivary gland neoplasms in children and adolescents" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "P.J. Bradley" 1 => "D.W. 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Odin" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.jpeds.2012.09.045" "Revista" => array:6 [ "tituloSerie" => "J Pediatr" "fecha" => "2013" "volumen" => "162" "paginaInicial" => "839" "paginaFinal" => "843" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/23140879" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] "agradecimientos" => array:1 [ 0 => array:4 [ "identificador" => "xack376529" "titulo" => "Acknowledgements" "texto" => "<p id="par0085" class="elsevierStylePara elsevierViewall">We would like to thank all the staff at the La Fe University and Polytechnic Hospital, Valencia, involved in this case, particularly the paediatric, oncology and pathological anatomy departments.</p>" "vista" => "all" ] ] ] "idiomaDefecto" => "en" "url" => "/21735735/0000006900000006/v1_201811180608/S2173573518300814/v1_201811180608/en/main.assets" "Apartado" => array:4 [ "identificador" => "5881" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Case Studies" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/21735735/0000006900000006/v1_201811180608/S2173573518300814/v1_201811180608/en/main.pdf?idApp=UINPBA00004N&text.app=https://www.elsevier.es/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2173573518300814?idApp=UINPBA00004N" ]
Journal Information
Vol. 69. Issue 6.
Pages 367-369 (November - December 2018)
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Vol. 69. Issue 6.
Pages 367-369 (November - December 2018)
Case study
Low Grade Mucoepidermoid Carcinoma of a Minor Salivary Gland of the Tongue in a Paediatric Patient
Carcinoma mucoepidermoide de bajo grado de glándula salival menor de lengua en un paciente pediátrico
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María Fernanda Vargas Gamarra
, José María Perolada Vilmaña, Miguel Armengot Carceller
Corresponding author
Departamento de Otorrinolaringología, Hospital Universitari i Politecnic La Fe, Valencia, Spain
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