Case study
Lymphangioma of the maxillary sinus
Linfangioma del seno maxilar
Andrea Migliorellia,
, Angelo Virgilio Pagliarib, Francesca Velenosia, Pasquale Blottab
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mglndr1@unife.it
Corresponding author at: Department of ENT & Audiology, University Hospital of Ferrara, Via A. Moro 8 (Cona), Ferrara, Ferrara 44100, Italy.
Corresponding author at: Department of ENT & Audiology, University Hospital of Ferrara, Via A. Moro 8 (Cona), Ferrara, Ferrara 44100, Italy.
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Otorinolaringoiatria Ospedale Maggiore ASST di Crema, Italy" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author at: Department of ENT & Audiology, University Hospital of Ferrara, Via A. Moro 8 (Cona), Ferrara, Ferrara 44100, Italy." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Linfangioma del seno maxilar" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 2349 "Ancho" => 3008 "Tamanyo" => 445249 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Axial CT scan with opacification of the left maxillary sinus by hypodense soft tissue. (B) Axial MRI highlighting a mass with high signal intensities in T2-weighted scan. (C) Pantographic radiograph with dome-shape radiopacity.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Case report</span><p id="par0005" class="elsevierStylePara elsevierViewall">A 30-year-old Italian man was referred to the Otolaryngology department of our Hospital.</p><p id="par0010" class="elsevierStylePara elsevierViewall">His main complaint was a two-month history of post-nasal drip, nasal obstruction, intense left facial pain and pressure over the left maxillary sinus trigger point.</p><p id="par0015" class="elsevierStylePara elsevierViewall">No other pre-existing symptoms or pathology were reported, he was not taking any medication, and he denied smoking and consuming alcohol.</p><p id="par0020" class="elsevierStylePara elsevierViewall">A complete head and neck examination was carried out.</p><p id="par0025" class="elsevierStylePara elsevierViewall">External examination of the nose was not significant.</p><p id="par0030" class="elsevierStylePara elsevierViewall">On anterior rhinoscopy a left septal deviation and contralateral hypertrophy of the inferior turbinate was found.</p><p id="par0035" class="elsevierStylePara elsevierViewall">In consideration of the negativity of the endoscopic objectivity, and following a dental evaluation that the patient had performed a few days earlier where a pantographic radiograph (OPT) was recommended, it was decided to begin with this type of imaging.</p><p id="par0040" class="elsevierStylePara elsevierViewall">The OPT showing a dome-shaped radiopacity of the left maxillary sinus (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0045" class="elsevierStylePara elsevierViewall">Therefore, a computed tomography (CT) scan of the paranasal sinus was performed, and it revealed a non-specific hypodense soft tissue opacification within the left maxillary sinus without erosion of the bone walls (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>).</p><p id="par0050" class="elsevierStylePara elsevierViewall">All other paranasal sinus were disease-free.</p><p id="par0055" class="elsevierStylePara elsevierViewall">Then was performed a magnetic resonance imagining (MRI) to complete pre-operative patient study, the mass showed low signal intensities in T1-weighted image and high in T2-weighted image (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>).</p><p id="par0060" class="elsevierStylePara elsevierViewall">Infudibulotomy, anterior ethmoidectomy, antrostomy of the medium meatus and curettage of the lesion using 0° 4<span class="elsevierStyleHsp" style=""></span>mm rigid endoscope were performed.</p><p id="par0065" class="elsevierStylePara elsevierViewall">The specimen collected was sent for histological examination.</p><p id="par0070" class="elsevierStylePara elsevierViewall">Histopathology revealed pseudostratified ciliated columnar epithelial lining with underlying loose edematous stroma containing dilated lymphatic channels of various sizes, weak expression CD 34 and CD 31 antigen and the diagnosis of lymphangioma was established.</p><p id="par0075" class="elsevierStylePara elsevierViewall">A multidisciplinary board with ENT, oncologist, hematologist and radiotherapist, was performed and a clinical and radiological follow-up was decided upon.</p><p id="par0080" class="elsevierStylePara elsevierViewall">After 18 months of follow-up, the patient is still asymptomatic and there are no signs of disease recurrence.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Discussion</span><p id="par0085" class="elsevierStylePara elsevierViewall">Lymphangioma is considered a lymphatic malformation of vascular origin that commonly occurs, in two thirds of reported cases, in the head and neck region and in some cases extends to the mediastinum.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a></p><p id="par0090" class="elsevierStylePara elsevierViewall">Lymphatic malformations occur most frequently in infants and children, representing approximately 5% of benign tumours, but rarely occur de novo in adults.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0095" class="elsevierStylePara elsevierViewall">Lymphangioma, although it can be classified as a benign lesion, causes complications by compressing surrounding structures and causing alterations in vision, breathing, swallowing and even disfigurement.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0100" class="elsevierStylePara elsevierViewall">Ultrasound shows multicystic lesion with no blood flow which helps to differentiate lymphatic lesion from haemangiomas and vascular malformations.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> CT and MRI delineate extension and relation with nearby anatomical structures to plan the surgery and differentiate from venous malformations.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,6</span></a></p><p id="par0105" class="elsevierStylePara elsevierViewall">Though it was first reported by Redenbacher in 1828 an accurate description was given by Virchow in 1854.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> Frequently lymphangioma presents a soft asymptomatic mass in the posterior triangle of the neck or oral cavity and rarely extends into the mediastinum. The localization in sinonasal region is unusual. About 50% of all lesions are noted at birth and around 90% develop by 2 years of age. No clear sex predilection is demonstrated, and Caucasians appear to be affected more often than peoples of other ethnicity. The precise aetiology of lymphangioma still remains unknown.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p><p id="par0110" class="elsevierStylePara elsevierViewall">Lymphangioma are thought to arise from a combination of the following: a failure of lymphatic vessels to connect to the venous system, abnormal budding of lymphatic tissue and sequestered lymphatic rest that retain their embryonic growth potential.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> Lymphangioma can have an acute enlargement with infection, even viral upper respiratory infection, or hormonal changes of puberty.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p><p id="par0115" class="elsevierStylePara elsevierViewall">The principal goal of lymphangioma management is the restoration or preservation of functional and aesthetic integrity. When there is no significant functional deficit treatment can be delayed and may consist only in observation. Although a spontaneous regression is rarely seen is, it has been reported in 1,6–16% of cases.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Preoperative staging is necessary for treatment planning. CT and MRI delineate anatomical extension and relationship with surrounding structures. The management is challenging because of the poor demarcation and the possible association to vital structures.</p><p id="par0120" class="elsevierStylePara elsevierViewall">Resection of lymphangioma can be associated with significant morbidity, blood loss, iatrogenic injuries, deformities and significant recurrence.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0125" class="elsevierStylePara elsevierViewall">Although, surgery with clear margins is the treatment of choice for lymphangioma of the paranasal sinuses.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">9</span></a></p><p id="par0130" class="elsevierStylePara elsevierViewall">These lesions within the sinus cavity, usually present with nasal obstruction or mimic chronic sinusitis. There may also be pain that is misdiagnosed as dental disorder.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">9</span></a></p><p id="par0135" class="elsevierStylePara elsevierViewall">CT is useful to determining site, size and extent of the lesion. In the differential diagnosis cystic like lesions of maxillary sinus, pseudocyst, mucous retention, mucoceles or polyps must be taken into account. The final diagnosis relies on histopathology because of the lesion’s nonspecific clinical and radiologic features.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">9</span></a> A proper resection is the gold standard therapy, and a post-operative follow-up is required. Due to the rarity of the occurrence of this lesion in paranasal sinuses, the tendency for recurrence is unknown. Therefore, appropriate periodic clinical and radiographic follow up evaluations should be performed.</p><p id="par0140" class="elsevierStylePara elsevierViewall">In conclusion, lymphangioma’s localization in paranasal sinus is rare, but should be consider in the differential diagnosis of the disorders of this region.</p><p id="par0145" class="elsevierStylePara elsevierViewall">Surgeons and pathologists should be aware of the occurrence of these lesions in such rare anatomical locations for prompt diagnosis and early treatment.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflicts of interest</span><p id="par0150" class="elsevierStylePara elsevierViewall">The authors certify that there is no conflict of interest with any financial organization regarding the material discussed in the manuscript.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Funding</span><p id="par0155" class="elsevierStylePara elsevierViewall">The authors report no involvement in the research by the sponsor that could have influenced the outcome of this work.</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Declaration of patient consent</span><p id="par0160" class="elsevierStylePara elsevierViewall">The authors certify that they have obtained all appropriate patient consent forms. In the form the patient have given his consent for his images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:6 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Case report" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Discussion" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Conflicts of interest" ] 3 => array:2 [ "identificador" => "sec0020" "titulo" => "Funding" ] 4 => array:2 [ "identificador" => "sec0025" "titulo" => "Declaration of patient consent" ] 5 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2022-11-29" "fechaAceptado" => "2023-01-29" "multimedia" => array:1 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 2349 "Ancho" => 3008 "Tamanyo" => 445249 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Axial CT scan with opacification of the left maxillary sinus by hypodense soft tissue. (B) Axial MRI highlighting a mass with high signal intensities in T2-weighted scan. (C) Pantographic radiograph with dome-shape radiopacity.</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" => "Head and neck malformation treatment: a systematic review" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "M.T. Adams" 1 => "B. Saltzmanm" 2 => "J.A. 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