Lacrimonasal duct angioleiomyoma. A rare cause of obstruction of the lagrimal drainage system

Escamilla Carpintero, Yolanda; Sellares Fabres, María Teresa; Blázquez Maña, Carmen; Prenafeta Moreno, Mario
doi:10.1016/j.otoeng.2020.03.003
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1</a>A-B&#41;&#46; The dacryocystography showed severe dilation of the lacrimal duct with stenosis of the lacrimonasal duct&#46;</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&#44; explore the lacrimal duct outlet and carry out probing&#44; in collaboration with the ophthalmology unit&#46; During surgery&#44; apart from the turbinate hypertrophy&#44; a tumour of over 3&#8239;cm was observed in the Hasner valve and inside the lacrimonasal duct&#44; which was resectioned endoscopic ally in two fragments&#44; being approximately 2&#8239;cm the one occupying the inside of the duct&#44; as may be observed in photography &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>C&#41;&#46; Despite observing major dilation of the lacrimonasal duct&#44; two-channel catheterisation was performed on the lacrimal duct&#44; which remained during the first month after surgery &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>D&#41;&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">The anatomopathological results reported on an angioleiomyoma&#44; when a proliferation of fusocellular morphologic cells were found which came from the muscle walls of the vascular structures&#44; with no cytological atypia and showing positivity for actin of the smooth muscle and CD31&#44; the marker with which they observed the endothelia of the vascular structures &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>A&#8211;C&#41;&#46; At the same time&#44; in the lower turbinate&#44; an inverted Schneiderian papilloma component was found&#46;</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&#44; with no changes &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>D&#41;&#46;</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&#46;6&#37; of all lacrimal obstructions with 72&#37; of them malignant&#46;<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&#44; giving rise to oncocytomas&#44; adenoid cystic carcinomas&#44; adeoncarcinomas or pleomorphic adenomas&#46;<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&#44; skin&#44; gut&#44; soft tissues and peritoneum&#44; due to the abundance of smooth muscle cells in these areas&#44; comprising 1&#37; of all benign tumours&#46; Their appearance in the nasal cavity&#44; paranasal sinuses&#44; and lacrimal duct structures is extremely rare due to the low presence of smooth muscle fibres in these locations&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> In a retrospective study on vascular leiomyoma of the head and neck&#44; between 1988 and 2001 Wang et al&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> found them in 21 patients&#58; only in two were they found in the nasal cavity&#46;</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&#44;<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6&#44;7</span></a> with our case being the first to have its origin in the lacrimonasal duct&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">Sinonasal leiomyoma accounts for under 1&#37; of all benign non-epithelial tumours&#46; The lower turbinate has a relative large amount of smooth muscle compared with other sinonasal locations and therefore&#44; within the rarity&#44; this is the most common location&#44; followed by the nasal septum and the vestibule&#46;<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">8&#44;9</span></a> According to different authors&#44; they are small masses&#44; no larger than 2&#8239;cm&#46;</p><p id="par0060" class="elsevierStylePara elsevierViewall">The cause of vascular leiomyoma is as yet uncertain&#44; but trauma&#44; therapy with steroids and hormonal imbalance have been involved in past cases&#46;</p><p id="par0065" class="elsevierStylePara elsevierViewall">Three theories exist to explain their origin&#58; one is the aberrant differentiation of the mesenchyme&#44; a tumour proliferation in smooth muscle elements in the wall of blood vessels&#44; which is the most accepted theory&#44; or a mixed combination of the previous two theories&#46;</p><p id="par0070" class="elsevierStylePara elsevierViewall">Radiologically&#44; it presents as a well defined tumour&#44; homogeneous in the CT&#44; hypodense or isodensde and in the MR it appears as an isointense lesion in the images weighted in T1 and hyperintense in T2&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> The differential diagnosis must be made with vascular tumours such as the hemangiopericytoma&#44; hemangioma or angiofibroma&#44; leiomyosarcoma&#44; neurofibroma and schwannoma&#44; and often basing this on imaging tests&#44; although in our case CT with contrast was not performed and neither was MR requested&#46; There are no reports of malignant transformation&#44; with recurrence being rare&#46;</p><p id="par0075" class="elsevierStylePara elsevierViewall">Histopathology provides definitive diagnosis&#44; showing lesions composed of prominent vascular structures&#44; with marked proliferation of fussiform cells which form inter-crossed bundles&#44; and which have oval nuclei or long nuclei with no cytological atypia&#44; elongated cytoplasms&#44; eosinophils with changes of vacuolation and which are found in relation to these vascular structures&#46; These cells are positive for smooth muscle actin&#46; There is an absence of mitosis&#44; which is a differential characteristic with regard to leiomyosarcoma&#44; since the mitotic index is the most useful histological indicator of benign lesions&#46;</p><p id="par0080" class="elsevierStylePara elsevierViewall">In patients with lacrimal sac tumours or lacimonasal duct that do not affect the canaliculi&#44; endoscopic surgery has become the treatment of choice&#44; given the advances of this technique&#46; Success rates are equivalent to the external approaches&#44; but with fewer postoperative complications&#44; better aesthetic outcomes and with a shorter recovery time&#46;</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&#46;</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&#46;</p></span></span>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Escamilla Carpintero Y&#44; Sellares Fabres MT&#44; Bl&#225;zquez Ma&#241;a C&#44; Prenafeta Moreno M&#46; Angioleiomioma del conducto lacrimonasal&#46; Una rara causa de obstrucci&#243;n de la v&#237;a lagrimal&#46; Acta Otorrinolaringol Esp&#46; 2021&#59;72&#58;262&#8211;264&#46;</p>"
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