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Superficial Angiomyxoma of the Parotid Region and Review of the Literature
Angiomixoma superficial de la región parotídea y revisión de la literatura
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"apellidos" => "de Vicente" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "Lucas" "apellidos" => "de Villalaín" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 3 => array:3 [ "nombre" => "Verónica" "apellidos" => "Blanco" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicio de Cirugía Oral y Maxilofacial, Hospital Universitario Central de Asturias, Oviedo, Spain" "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Anatomía Patológica, Hospital Universitario Central de Asturias, Oviedo, Spain" "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Angiomixoma superficial de la región parotídea y revisión de la literatura" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 710 "Ancho" => 942 "Tamanyo" => 174241 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Histological examination revealed a proliferation of spindle, stellate and oval cells within a fibromyxoid stroma and accompanied by small capillaries, as well as monocytes and lymphocytes (haematoxylin–eosin; original magnification, ×100).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Superficial angiomyxoma (SA) is a rare cutaneous neoplasm, which was first described in 1988 by Allen et al.,<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> who reported 30 cases in 28 patients. SA is included in the group of soft tissue, myxomatous tumours characterised by their unique ability to produce a large amount of myxoid material, along with the proliferation of tumour constituent cells.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> We report the case of a patient who presented SA in the parotid region, highlighting the importance of distinguishing this entity from other lesions which may appear in this region.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Clinical Case</span><p id="par0010" class="elsevierStylePara elsevierViewall">The patient was a 61-year old male who attended consultation due to a painless, right parotid tumour, with progressive growth for 1 year. The patient had no relevant medical history. Physical examination revealed a mobile and elastic mass, approximately 2<span class="elsevierStyleHsp" style=""></span>cm in size, located in the left parotid region at the level of the mandibular angle. Facial nerve function was normal.</p><p id="par0015" class="elsevierStylePara elsevierViewall">We performed a contrast-enhanced CT scan (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>), which revealed a sharply defined, hypodense lesion, measuring 27<span class="elsevierStyleHsp" style=""></span>mm×16<span class="elsevierStyleHsp" style=""></span>mm and located immediately in front and below the superficial lobe of the right parotid gland. Fine needle aspiration revealed a mesenchymal lesion accompanied by scattered adipocytes and fibroblasts within a myxoid stroma.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">The patient underwent surgical excision of the lesion via a retromandibular approach. Macroscopically, it measured 30<span class="elsevierStyleHsp" style=""></span>mm×2.7<span class="elsevierStyleHsp" style=""></span>mm×20<span class="elsevierStyleHsp" style=""></span>mm, presented a whitish colour, had a myxoid appearance and was circumscribed by a thin capsule. Microscopically, it consisted of a sparse proliferation of spindle, stellate, and oval cells within a fibromyxoid stroma, accompanied by lymphocytes and delicate capillaries which did not form a constant network (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). We did not find any signs of malignancy or cytological aggressiveness. Immunohistochemical analysis showed positive staining for CD34 and negative for cytokeratins, S-100, desmin, actin, CD99 and CD112. These findings were consistent with the diagnosis of superficial angiomyxoma.</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">Given the diagnosis, we performed an echocardiogram which revealed the absence of atrial myxomas. Moreover, physical examination found no other tumours or pigmented lesions which suggested an associated syndrome. A complementary, biochemical analysis showed no signs of endocrine hyperactivity.</p><p id="par0030" class="elsevierStylePara elsevierViewall">After 10 months of follow-up, there were no signs of recurrence.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Discussion</span><p id="par0035" class="elsevierStylePara elsevierViewall">The term “superficial angiomyxoma” was first used in 1989 to designate a benign, myxoid-type neoplasm characterised by the presence of scattered cell nests and small vessels.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> This entity was named “superficial angiomyxoma” in order to distinguish it from “aggressive angiomyxoma”, which primarily affects the female genital region.</p><p id="par0040" class="elsevierStylePara elsevierViewall">Histological examination of SA reveals a noninvasive lesion with well-defined margins. Microscopically, it shares some of the characteristics of other myxomatous lesions (cutaneous mucinosis and aggressive angiomyxoma), such as the presence of spindle, stellate, and oval cells within a myxoid stroma. However, SA typically shows a scattered distribution of blood vessels of small and medium calibre and thin walls. An important diagnostic clue is the presence of inflammatory cells, which are absent in other myxomatous lesions. Occasionally, epithelial inclusions may be present, but this was not so in our case. Immunohistochemical analysis is often nonspecific and may show variable positivity for CD34 and protein S-100, smooth muscle actin and pankeratin.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Clinically, SA often appears as a skin nodule, papule or polypoid lesion, mainly affecting the trunk and lower limbs, followed by the head and neck and, finally, the upper limbs.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">We conducted a retrospective review of medical literature in English in Medline and found only 28 cases in the head and neck region.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1–11</span></a> We observed a slightly higher prevalence in men (57%) than in women. The age of patients ranged from 12 to 82 years with a mean age of 36.45 years. These data are consistent with those reported in other locations.</p><p id="par0055" class="elsevierStylePara elsevierViewall">The present case represents the first one with parotid involvement described in the literature. In this location, the differential diagnosis must include parotid gland tumours and cysts of the first branchial arch. Fine needle aspiration and MRI studies may be useful in this regard.</p><p id="par0060" class="elsevierStylePara elsevierViewall">The differential diagnosis must include focal cutaneous mucinosis, trichodiscoma, fibrofolliculoma, perifollicular fibroma, trichofolliculoma, trichogenic myxoma and other cutaneous lesions, such as epidermoid cysts, lipomas, neurofibromas, abscesses, lymphangioma, fibroma, and dermatofibrosarcoma. It is important to rule out an association with Carney complex in all patients, especially in those forms located in the external ear.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">12</span></a> This autosomal dominant syndrome includes the presence of cardiac and cutaneous myxomas, skin hyperpigmentation, and endocrine hyperactivity (Cushing syndrome, sexual precocity, and acromegaly). All these characteristics were absent in our patient.</p><p id="par0065" class="elsevierStylePara elsevierViewall">Although they do not generate distant metastases, a general tendency to recur has been described in up to 30%–40% of cases, indicating the need for postoperative monitoring. However, the recurrence rate was lower (16%) in those cases described in the head and neck region.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Conflict of Interests</span><p id="par0070" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:9 [ 0 => array:2 [ "identificador" => "xres94958" "titulo" => "Abstract" ] 1 => array:2 [ "identificador" => "xpalclavsec82110" "titulo" => "Keywords" ] 2 => array:2 [ "identificador" => "xres94959" "titulo" => "Resumen" ] 3 => array:2 [ "identificador" => "xpalclavsec82111" "titulo" => "Palabras clave" ] 4 => array:2 [ "identificador" => "sec0005" "titulo" => "Introduction" ] 5 => array:2 [ "identificador" => "sec0010" "titulo" => "Clinical Case" ] 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 "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec82110" "palabras" => array:3 [ 0 => "Superficial angiomyxoma" 1 => "Parotid gland tumours" 2 => "Parotid" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec82111" "palabras" => array:3 [ 0 => "Angiomixoma superficial" 1 => "Tumores de la glándula parótida" 2 => "Parótida" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Superficial angiomyxoma (SA) is a rare benign cutaneous neoplasm first described by Allen et al. in 1988. To the best of our knowledge, we report the first case of superficial angiomyxoma located in the parotid region. We also stress the importance of distinguishing this entity from other lesions that may be involved in this location such as cutaneous neoplasms, parotid tumours or cysts. We emphasise the need to rule out the Carney complex, which has been associated with these tumours.</p>" ] "es" => array:2 [ "titulo" => "Resumen" "resumen" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">El angiomixoma superficial (AS) es una neoplasia benigna cutánea infrecuente que fue descrita por primera vez por Allen y colaboradores en 1988. Presentamos el primer caso publicado en la literatura médica de angiomixoma superficial localizado en la región parotídea, destacando la importancia de distinguir esta entidad de otras lesiones que pueden aparecer en esta área, como neoplasias cutáneas, tumores o quistes. Así mismo hacemos hincapié en la necesidad de descartar la asociación con el complejo Carney ante el diagnóstico de este tipo de lesiones.</p>" ] ] "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara">Please cite this article as: Rosado Rodríguez P, et al. Angiomixoma superficial de la región parotídea y revisión de la literatura. Acta Otorrinolaringol Esp. 2012;63:147–9.</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" => 726 "Ancho" => 942 "Tamanyo" => 104708 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Contrast-enhanced CT revealed an oval, hypodense lesion, measuring 27<span class="elsevierStyleHsp" style=""></span>mm×16<span class="elsevierStyleHsp" style=""></span>mm, with well-defined margins, located in front and below the superficial lobe of the right parotid gland and lateral to the masseteric muscle.</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" => 710 "Ancho" => 942 "Tamanyo" => 174241 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Histological examination revealed a proliferation of spindle, stellate and oval cells within a fibromyxoid stroma and accompanied by small capillaries, as well as monocytes and lymphocytes (haematoxylin–eosin; original magnification, ×100).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:12 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Superficial angiomyxomas with and without epithelial components. 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