Case study
Sclerosing Polycystic Adenosis of the Submandibular Gland
Adenosis poliquística esclerosante de la glándula submaxilar
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Proliferation of acini and ducts, some of them dilated, in a sclerotic stroma with a slight, chronic, inflammatory infiltrate.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Sclerosing polycystic adenosis (SPA) is a rare entity which was first described in 1996. Its anatomopathological appearance is similar to that of fibrocystic breast disease. A total of 51 cases have been described in the literature. Its aetiology is unknown, although it is clonal and, therefore, neoplastic nature has been reported. Most cases appear in the major salivary glands, especially in the parotid gland.</p><p id="par0010" class="elsevierStylePara elsevierViewall">It is important to carry out the differential diagnosis with other salivary gland tumours, mainly acinar cell carcinoma.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Case Report</span><p id="par0015" class="elsevierStylePara elsevierViewall">We present the case of a 63-year-old female who attended consultation due to a left cervical tumour of a few months evolution. The patient was a smoker.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Examination revealed a normal oropharynx, with reddening of the drainage orifice of the left Wharton duct. Fibroscopy was normal, whilst bimanual and cervical palpation noticed an enlarged, left submandibular gland, with a posterior adenopathy to a mobile and rolling gland of about 2<span class="elsevierStyleHsp" style=""></span>cm in diameter.</p><p id="par0025" class="elsevierStylePara elsevierViewall">A cervical computed tomography (CT) scan revealed a solid nodule of 16<span class="elsevierStyleHsp" style=""></span>mm in the left submandibular gland, with an adenopathy of 6<span class="elsevierStyleHsp" style=""></span>mm adjacent to the submaxillary gland.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Ultrasound-guided fine needle aspiration reported a well-differentiated acinar cell carcinoma (low grade).</p><p id="par0035" class="elsevierStylePara elsevierViewall">Given these findings, we performed a left submaxillectomy with neck dissection of group I.</p><p id="par0040" class="elsevierStylePara elsevierViewall">The pathological anatomy of the surgical specimen reported an irregular, whitish tumour of firm consistency, measuring 1.7<span class="elsevierStyleHsp" style=""></span>cm in its greatest dimension, occupying 30% of the gland, without extension into the rest of the gland and without vascular or perineural invasion.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Microscopically, we observed dilated ducts with intraluminal, epithelial hyperplasia and cellular atypia, surrounded by a stroma with abundant sclerosis. The lymph nodes were negative for malignancy. The final diagnosis was SPA (<a class="elsevierStyleCrossRefs" href="#fig0005">Figs. 1 and 2</a>).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0050" class="elsevierStylePara elsevierViewall">Two years after surgery, the patient remains asymptomatic and has not suffered recurrence or distant metastasis.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Discussion</span><p id="par0055" class="elsevierStylePara elsevierViewall">SPA was first described in 1996 by Smith et al.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> At present, there are about 50 cases reported in the literature.</p><p id="par0060" class="elsevierStylePara elsevierViewall">It mainly affects the major salivary glands, especially the parotid gland, although it may exceptionally affect the minor salivary glands.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,3</span></a> It appears in middle-aged subjects (40–50 years) with no clear gender prevalence. The tumours measure between 0.5 and 7<span class="elsevierStyleHsp" style=""></span>cm in diameter.</p><p id="par0065" class="elsevierStylePara elsevierViewall">It is characterised by a slow growth, which is usually asymptomatic, nodular and similar in many respects to fibrocystic breast disease.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">The pathological anatomy reveals circumscribed, but not encapsulated nodules, with dilated ducts and epithelial hyperplasia, surrounded by a very fibrous stroma and with chronic inflammation.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0075" class="elsevierStylePara elsevierViewall">The aetiology of this condition is currently unknown, although some recent articles have reported conducting trials with human androgen-receptor gene (HUMARA),<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> and demonstrated its clonal and, therefore, neoplastic nature. Furthermore, immunohistochemistry techniques have detected positivity for oestrogen and progesterone receptors which may play a role in its pathogenesis. Moreover, some studies have pointed to the role of Epstein–Barr virus (EBV) in SPA.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a></p><p id="par0080" class="elsevierStylePara elsevierViewall">Its treatment is surgery with disease-free margins, given that 30% of cases have reported recurrences. There have been no reports of distant metastases or mortality in this condition.</p><p id="par0085" class="elsevierStylePara elsevierViewall">In many cases, fine-needle aspiration (FNA) techniques<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> may confuse the tumours with other types of salivary gland tumours, such as acinar cell carcinoma or mucoepidermoid carcinoma. Therefore, we must always consider this entity when conducting the differential diagnosis, since, while still remaining a neoplasm, treatment and prognosis of this disease are different from those of other malignant tumours of the salivary glands.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Conflict of Interests</span><p id="par0090" class="elsevierStylePara elsevierViewall">The authors have no conflict of interests to declare.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:9 [ 0 => array:2 [ "identificador" => "xres94868" "titulo" => "Abstract" ] 1 => array:2 [ "identificador" => "xpalclavsec82018" "titulo" => "Keywords" ] 2 => array:2 [ "identificador" => "xres94869" "titulo" => "Resumen" ] 3 => array:2 [ "identificador" => "xpalclavsec82019" "titulo" => "Palabras clave" ] 4 => array:2 [ "identificador" => "sec0005" "titulo" => "Introduction" ] 5 => array:2 [ "identificador" => "sec0010" "titulo" => "Case Report" ] 6 => array:2 [ "identificador" => "sec0015" "titulo" => "Discussion" ] 7 => array:2 [ "identificador" => "sec0020" "titulo" => "Conflict of Interests" ] 8 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2011-02-27" "fechaAceptado" => "2011-04-12" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec82018" "palabras" => array:3 [ 0 => "Sclerosing polycystic adenosis" 1 => "Submandibular gland" 2 => "Salivary glands tumours" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec82019" "palabras" => array:3 [ 0 => "Adenosis poliquística esclerosante" 1 => "Glándula submaxilar" 2 => "Tumores glándulas salivales" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Sclerosing polycystic adenosis (SPA) is a rare, newly reported lesion of the salivary glands. Most cases occurred within major salivary glands and were frequently confused with other salivary gland carcinomas such as acinar cell carcinoma. We report a case of sclerosing polycystic adenosis within the submandibular gland in a 63-year-old woman.</p>" ] "es" => array:2 [ "titulo" => "Resumen" "resumen" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">La Adenosis Poliquística Esclerosante (APE) es una entidad poco frecuente descrita hace poco tiempo que afecta a las glándulas salivales, principalmente mayores, y que frecuentemente se ha confundido con otros carcinomas de glándula salival como el carcinoma de células acinares. Presentamos un caso de adenosis poliquística esclerosante en la glándula submaxilar de una mujer de 63 años.</p>" ] ] "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara">Please cite this article as: Beato Martínez A, et al. Adenosis poliquística esclerosante de la glándula submaxilar. Acta Otorrinolaringol Esp. 2013;64:78–80.</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" => 714 "Ancho" => 951 "Tamanyo" => 214162 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Sclerosing polycystic adenosis (×2) (H-E staining). Circumscribed, partially encapsulated lesion consisting of a proliferation of ducts and acini, with a lobar pattern and without atypia. By comparison, the adjacent gland is not affected and presents no fibrosis, inflammatory infiltrates or atrophy.</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" => 715 "Ancho" => 951 "Tamanyo" => 289691 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Sclerosing polycystic adenosis (×10) (H-E staining). Proliferation of acini and ducts, some of them dilated, in a sclerotic stroma with a slight, chronic, inflammatory infiltrate.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:8 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Sclerosing polycystic adenosis of major salivary glands. A clinicopathologic analysis of nine cases" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "B.C. Smith" 1 => "G.L. Ellis" 2 => "L.J. Slater" 3 => "R.D. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2023 March | 3 | 2 | 5 |
| 2021 February | 0 | 2 | 2 |
| 2021 January | 0 | 1 | 1 |
| 2020 December | 0 | 2 | 2 |
| 2020 November | 0 | 3 | 3 |
| 2020 October | 0 | 1 | 1 |
| 2020 August | 0 | 1 | 1 |
| 2020 June | 0 | 1 | 1 |
| 2020 March | 19 | 7 | 26 |
| 2020 February | 11 | 6 | 17 |
| 2020 January | 16 | 6 | 22 |
| 2019 December | 27 | 20 | 47 |
| 2019 November | 21 | 11 | 32 |
| 2019 October | 18 | 5 | 23 |
| 2019 September | 30 | 16 | 46 |
| 2019 August | 18 | 5 | 23 |
| 2019 July | 39 | 11 | 50 |
| 2019 June | 45 | 30 | 75 |
| 2019 May | 79 | 25 | 104 |
| 2019 April | 37 | 45 | 82 |
| 2019 March | 17 | 10 | 27 |
| 2019 February | 19 | 8 | 27 |
| 2019 January | 13 | 5 | 18 |
| 2018 December | 18 | 9 | 27 |
| 2018 November | 15 | 3 | 18 |
| 2018 October | 22 | 5 | 27 |
| 2018 May | 1 | 0 | 1 |
| 2018 April | 8 | 1 | 9 |
| 2018 March | 11 | 2 | 13 |
| 2018 February | 5 | 2 | 7 |
| 2018 January | 12 | 2 | 14 |
| 2017 December | 6 | 2 | 8 |
| 2017 November | 9 | 1 | 10 |
| 2017 October | 11 | 1 | 12 |
| 2017 September | 16 | 9 | 25 |
| 2017 August | 18 | 1 | 19 |
| 2017 July | 25 | 2 | 27 |
| 2017 June | 30 | 3 | 33 |
| 2017 May | 36 | 3 | 39 |
| 2017 April | 30 | 1 | 31 |
| 2017 March | 34 | 30 | 64 |
| 2017 February | 122 | 5 | 127 |
| 2017 January | 35 | 3 | 38 |
| 2016 December | 30 | 9 | 39 |
| 2016 November | 37 | 4 | 41 |
| 2016 October | 34 | 6 | 40 |
| 2016 September | 37 | 8 | 45 |
| 2016 August | 16 | 2 | 18 |
| 2016 July | 34 | 3 | 37 |
| 2016 June | 29 | 11 | 40 |
| 2016 May | 27 | 10 | 37 |
| 2016 April | 50 | 22 | 72 |
| 2016 March | 36 | 16 | 52 |
| 2016 February | 34 | 16 | 50 |
| 2016 January | 49 | 15 | 64 |
| 2015 December | 30 | 14 | 44 |
| 2015 November | 13 | 13 | 26 |
| 2015 October | 46 | 16 | 62 |
| 2015 September | 21 | 8 | 29 |
| 2015 August | 54 | 7 | 61 |
| 2015 July | 52 | 7 | 59 |
| 2015 June | 26 | 4 | 30 |
| 2015 May | 31 | 3 | 34 |
| 2015 April | 40 | 8 | 48 |
| 2015 March | 37 | 9 | 46 |
| 2015 February | 13 | 8 | 21 |
| 2015 January | 35 | 4 | 39 |
| 2014 December | 23 | 7 | 30 |
| 2014 November | 18 | 4 | 22 |
| 2014 October | 26 | 3 | 29 |
| 2014 September | 16 | 0 | 16 |
| 2014 August | 24 | 3 | 27 |
| 2014 July | 30 | 5 | 35 |
| 2014 June | 12 | 1 | 13 |
| 2014 May | 19 | 3 | 22 |
| 2014 April | 18 | 2 | 20 |
| 2014 March | 20 | 2 | 22 |
| 2014 February | 12 | 3 | 15 |
| 2014 January | 11 | 1 | 12 |
| 2013 December | 16 | 2 | 18 |
| 2013 November | 13 | 3 | 16 |
| 2013 October | 29 | 3 | 32 |
| 2013 September | 26 | 3 | 29 |
| 2013 August | 17 | 1 | 18 |
| 2013 July | 13 | 2 | 15 |
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