Case study
Lacrimonasal duct angioleiomyoma. A rare cause of obstruction of the lagrimal drainage system
Angioleiomioma del conducto lagrimonasal. Una rara causa de obstrucción de la vía lagrimal
Yolanda Escamilla Carpinteroa,
, María Teresa Sellares Fabresb, Carmen Blázquez Mañac, Mario Prenafeta Morenod
Corresponding author
a Servicio de ORL, Hospital Universitario Parc Taulí, Sabadell, Spain
b Servicio de Oftalmología, Hospital Universitario Parc Taulí, Sabadell, Spain
c Servicio de Patología, Hospital Universitario Parc Taulí, Sabadell, Spain
d Servicio de Radiodiagnóstico, UDIAT, Hospital Universitario Parc Taulí, Sabadell, Spain
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Una rara causa de obstrucción de la vía lagrimal" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1202 "Ancho" => 2091 "Tamanyo" => 415506 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Results: A) haematoxylin-eosin ×10; B) positivity for smooth muscle actin; C) positivity for CD31; D) endoscopic result.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Clinical case</span><p id="par0005" class="elsevierStylePara elsevierViewall">A 42-year-old patient, diagnosed with chronic rhinitis, visited the practice due to long-duration onset symptoms of insufficient nasal respiration, epiphora and bulging of the inner corner of the left eye.</p><p id="par0010" class="elsevierStylePara elsevierViewall">ENT examination showed a large lower turbinate hypertrophy and rounded tumour below the lower turbinate which was practically stuck to it.</p><p id="par0015" class="elsevierStylePara elsevierViewall">Computerised tomography (TC) and dacryocystography was requested for evaluation of the lacrimal duct.</p><p id="par0020" class="elsevierStylePara elsevierViewall">TC showed major dilation of the left lacrimonasal duct, focal occupation of soft tissues at the inner corner of the eye level and hypertrophy of the lower left turbinate (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A-B). The dacryocystography showed severe dilation of the lacrimal duct with stenosis of the lacrimonasal duct.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">Endoscopic surgery was programmed to conduct a turbinectomy at the level of the lower left turbinate, explore the lacrimal duct outlet and carry out probing, in collaboration with the ophthalmology unit. During surgery, apart from the turbinate hypertrophy, a tumour of over 3 cm was observed in the Hasner valve and inside the lacrimonasal duct, which was resectioned endoscopic ally in two fragments, being approximately 2 cm the one occupying the inside of the duct, as may be observed in photography (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>C). Despite observing major dilation of the lacrimonasal duct, two-channel catheterisation was performed on the lacrimal duct, which remained during the first month after surgery (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>D).</p><p id="par0030" class="elsevierStylePara elsevierViewall">The anatomopathological results reported on an angioleiomyoma, when a proliferation of fusocellular morphologic cells were found which came from the muscle walls of the vascular structures, with no cytological atypia and showing positivity for actin of the smooth muscle and CD31, the marker with which they observed the endothelia of the vascular structures (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>A–C). At the same time, in the lower turbinate, an inverted Schneiderian papilloma component was found.</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0035" class="elsevierStylePara elsevierViewall">After one year the patient remains asymptomatic and endoscopic examination showed a dilated lacrimonasal duct, with no changes (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>D).</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Discussion</span><p id="par0040" class="elsevierStylePara elsevierViewall">Tumours of the lacrimonasal duct account for 4.6% of all lacrimal obstructions with 72% of them malignant.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> They arise from the ciliated epithelium of the sac and the duct or the serous and mucous glands, giving rise to oncocytomas, adenoid cystic carcinomas, adeoncarcinomas or pleomorphic adenomas.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Vascular leiomyoma or angioleiomyoma is a benign tumour of the smooth muscle which is usually found in the uterus, skin, gut, soft tissues and peritoneum, due to the abundance of smooth muscle cells in these areas, comprising 1% of all benign tumours. Their appearance in the nasal cavity, paranasal sinuses, and lacrimal duct structures is extremely rare due to the low presence of smooth muscle fibres in these locations.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> In a retrospective study on vascular leiomyoma of the head and neck, between 1988 and 2001 Wang et al.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> found them in 21 patients: only in two were they found in the nasal cavity.</p><p id="par0050" class="elsevierStylePara elsevierViewall">There are 25 published cases on the eyeball<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> and to date two on the lacrimal sac,<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6,7</span></a> with our case being the first to have its origin in the lacrimonasal duct.</p><p id="par0055" class="elsevierStylePara elsevierViewall">Sinonasal leiomyoma accounts for under 1% of all benign non-epithelial tumours. The lower turbinate has a relative large amount of smooth muscle compared with other sinonasal locations and therefore, within the rarity, this is the most common location, followed by the nasal septum and the vestibule.<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">8,9</span></a> According to different authors, they are small masses, no larger than 2 cm.</p><p id="par0060" class="elsevierStylePara elsevierViewall">The cause of vascular leiomyoma is as yet uncertain, but trauma, therapy with steroids and hormonal imbalance have been involved in past cases.</p><p id="par0065" class="elsevierStylePara elsevierViewall">Three theories exist to explain their origin: one is the aberrant differentiation of the mesenchyme, a tumour proliferation in smooth muscle elements in the wall of blood vessels, which is the most accepted theory, or a mixed combination of the previous two theories.</p><p id="par0070" class="elsevierStylePara elsevierViewall">Radiologically, it presents as a well defined tumour, homogeneous in the CT, hypodense or isodensde and in the MR it appears as an isointense lesion in the images weighted in T1 and hyperintense in T2.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> The differential diagnosis must be made with vascular tumours such as the hemangiopericytoma, hemangioma or angiofibroma, leiomyosarcoma, neurofibroma and schwannoma, and often basing this on imaging tests, although in our case CT with contrast was not performed and neither was MR requested. There are no reports of malignant transformation, with recurrence being rare.</p><p id="par0075" class="elsevierStylePara elsevierViewall">Histopathology provides definitive diagnosis, showing lesions composed of prominent vascular structures, with marked proliferation of fussiform cells which form inter-crossed bundles, and which have oval nuclei or long nuclei with no cytological atypia, elongated cytoplasms, eosinophils with changes of vacuolation and which are found in relation to these vascular structures. These cells are positive for smooth muscle actin. There is an absence of mitosis, which is a differential characteristic with regard to leiomyosarcoma, since the mitotic index is the most useful histological indicator of benign lesions.</p><p id="par0080" class="elsevierStylePara elsevierViewall">In patients with lacrimal sac tumours or lacimonasal duct that do not affect the canaliculi, endoscopic surgery has become the treatment of choice, given the advances of this technique. Success rates are equivalent to the external approaches, but with fewer postoperative complications, better aesthetic outcomes and with a shorter recovery time.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Financing</span><p id="par0085" class="elsevierStylePara elsevierViewall">The authors did not receive any source of financing.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">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:5 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Clinical case" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Discussion" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Financing" ] 3 => array:2 [ "identificador" => "sec0020" "titulo" => "Conflict of interests" ] 4 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2020-01-19" "fechaAceptado" => "2020-03-15" "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Escamilla Carpintero Y, Sellares Fabres MT, Blázquez Maña C, Prenafeta Moreno M. Angioleiomioma del conducto lacrimonasal. Una rara causa de obstrucción de la vía lagrimal. Acta Otorrinolaringol Esp. 2021;72:262–264.</p>" ] ] "multimedia" => array:2 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1584 "Ancho" => 1674 "Tamanyo" => 290817 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Images of diagnosis. A) TC coronal; B) Axial CT; C) interior tumour fragment, lacrimonasal duct; D) surgical endoscopic image of the lacrimonasal duct.</p>" ] ] 1 => array:8 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1202 "Ancho" => 2091 "Tamanyo" => 415506 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Results: A) haematoxylin-eosin ×10; B) positivity for smooth muscle actin; C) positivity for CD31; D) endoscopic result.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Tumors of the lacrimal drainage system" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "L.M. 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