Case Study
Laryngeal Lipoma Associated With Madelung's Disease: A Case Report
Lipoma laríngeo asociado a enfermedad de Madelung: a propósito de un caso
Guillermo Arturo Landínez-Cepeda
, Emilio V. Alarcos-Tamayo, Teresa Millás-Gómez, Darío Morais-Pérez
Corresponding author
Servicio de Otorrinolaringología y Patología Cervico-Facial, Hospital Clínico Universitario de Valladolid, Spain
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(A) Left internal carotid arteriography. There is a compartment of the lesion (black arrow) which is irrigated by the caroticotympanic branch of the artery. (B) and (C) Left external carotid arteriography. This image shows the other 2 compartments: one irrigated by the inferior tympanic branch (black arrow) of the ascending pharyngeal, and another, larger one, irrigated by pharyngeal branches of the ascending pharyngeal artery and branches of a hypertrophied, accessory meningeal artery (white arrow). (D) Anteroposterior view showing the extension of the lesion.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Miriam Portero de la Torre, Rafael Sánchez Gutiérrez, Inmaculada Gallardo Muñoz, Francisco A. 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Alarcos-Tamayo, Teresa Millás-Gómez, Darío Morais-Pérez" "autores" => array:4 [ 0 => array:4 [ "nombre" => "Guillermo Arturo" "apellidos" => "Landínez-Cepeda" "email" => array:1 [ 0 => "glandinez@yahoo.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">¿</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "Emilio V." "apellidos" => "Alarcos-Tamayo" ] 2 => array:2 [ "nombre" => "Teresa" "apellidos" => "Millás-Gómez" ] 3 => array:2 [ "nombre" => "Darío" "apellidos" => "Morais-Pérez" ] ] "afiliaciones" => array:1 [ 0 => array:1 [ "entidad" => "Servicio de Otorrinolaringología y Patología Cervico-Facial, Hospital Clínico Universitario de Valladolid, Spain" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Lipoma laríngeo asociado a enfermedad de Madelung: a propósito de un caso" ] ] "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" => 745 "Ancho" => 1000 "Tamanyo" => 126282 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Coronal cervical CT image showing infiltration by unencapsulated (arrows) and endolaryngeal (circles) fat, which reduced the calibre of the glottic lumen.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">The association of multiple cervical lipomatosis and laryngeal infiltration with intrinsic lipomas is rare, with only 3 cases reported in the medical literature.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> We draw attention to this fact and the possibility of dyspnoea symptoms by laryngeal obstruction due to lipomas associated with Madelung's disease.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Clinical Case</span><p id="par0010" class="elsevierStylePara elsevierViewall">We present the case of 48-year-old male with a drinking habit, who smoked over 20 cigarettes/day and who attended the emergency service due to a worsening in his “usual fatigue”. The patient presented inspiratory dyspnoea with stridor, supraclavicular retractions and a marked thickening of the neck which led us to request an emergency CT scan (<a class="elsevierStyleCrossRefs" href="#fig0005">Figs. 1 and 2</a>). The scan revealed the presence of multiple tumours, scattered throughout the neck, with fat density and infiltrating deep tissues, compressing the upper airway up to the sternal notch. Laryngeal examination by fibroscopy found a reddish-yellow mass of 3<span class="elsevierStyleHsp" style=""></span>cm in diameter, which pedicled on the glottic lumen from the right arytenoepiglottic fold and obstructed it intermittently, according to breathing movements. We performed an emergency tracheotomy and subsequently removed the laryngeal tumour by microlaryngoscopy. The anatomopathological report was of lipoma. The patient recovered uneventfully and the tracheostoma closed a few days later. At 4 months we conducted a bilateral cervicotomy to remove the cervical lipomas, which improved the physical appearance and reduced the sensation of cervical compression.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Discussion</span><p id="par0015" class="elsevierStylePara elsevierViewall">Madelung's disease, Launois-Bensaude syndrome or multiple symmetric lipomatosis is an increasingly rare entity, with over 300 cases reported in the medical literature. It consists in the infiltration by unencapsulated fat of the anterior region of the neck, the proximal regions of the upper limbs, chest and facial region. It can cause cosmetic deformity, extrinsic vascular compression symptoms, upper airway compression symptoms and, more rarely, compression of the recurrent laryngeal nerve by fatty infiltration, leading to dyspnoea and dysphonia.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> There are 2 types; type I is more localised and manifests in middle-aged Mediterranean men with a history of alcohol intake in 60%–90% of cases,<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> metabolic disorders such as hyperuricemia, diabetes, goitre, and hypertension, whereas type II is more diffuse and more common among women with a high body mass index.<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4,5</span></a> Presumably, its aetiology involves a local defect in lipolysis induced by catecholamines and the lipogenic and antilipolytic effect of alcohol. It displays greater susceptibility towards developing squamous cell carcinomas of the upper aerodigestive tract in alcoholics and smokers,<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> related to these risk factors.</p><p id="par0020" class="elsevierStylePara elsevierViewall">At present, there have been about 115 reported cases of isolated fat infiltration in the larynx, representing 0.6% of benign tumours of the head and neck.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> However, the association of Madelung's disease with the appearance of intrinsic laryngeal lipomas is more uncommon, with only 3 reported cases in the specialised literature.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">The diagnostic approach is based on examination and palpation of the fatty deformity. A CT or MRI imaging study may show densities and intensities similar to those of normal fat infiltrating cervical spaces and structures.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> Laryngeal infiltration and the possibility of developing a lipoma which occludes the lumen should be suspected upon observing symptoms of inspiratory dyspnoea with stridor. In such cases, airway preservation and diagnostic laryngeal endoscopy, along with excision of the tumour, should be the attitude to follow with urgency. Surgical resection of cervical lipomas by cervicotomy would be indicated in cases of extrinsic vascular compression and upper aerodigestive tract compression with visceral infiltration, while liposuction should be reserved for cases of strict cosmetic deformity without infiltration of important structures.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Conclusion</span><p id="par0030" class="elsevierStylePara elsevierViewall">We wish to draw attention to Madelung's disease and its possible association with laryngeal lipomas. When this happens, symptoms of subacute dyspnoea may appear which may require, as in our case, rapid action including tracheotomy and subsequent excision of the lipoma obstructing the laryngeal lumen.</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Conflict of Interests</span><p id="par0035" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:10 [ 0 => array:2 [ "identificador" => "xres95016" "titulo" => "Abstract" ] 1 => array:2 [ "identificador" => "xpalclavsec82168" "titulo" => "Keywords" ] 2 => array:2 [ "identificador" => "xres95017" "titulo" => "Resumen" ] 3 => array:2 [ "identificador" => "xpalclavsec82169" "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" => "Conclusion" ] 8 => array:2 [ "identificador" => "sec0025" "titulo" => "Conflict of Interests" ] 9 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec82168" "palabras" => array:3 [ 0 => "Laryngeal lipoma" 1 => "Madelung's syndrome" 2 => "Dyspnoea" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec82169" "palabras" => array:3 [ 0 => "Lipoma laríngeo" 1 => "Enfermedad de Madelung" 2 => "Disnea" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Multiple symmetric lipomatosis is an alteration in the neck, upper trunk and upper extremities fat deposits. It produces an aesthetic problem and sometimes upper airway obstruction when the larynx is infiltrated by the mass. We report the case of a male with Madelung's disease, which began with acute dyspnoea caused by laryngeal fat deposits and obstructive lipoma.</p>" ] "es" => array:2 [ "titulo" => "Resumen" "resumen" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">La lipomatosis cervical múltiple es una alteración del depósito graso cervical, parte alta de tórax y extremidades superiores que genera un problema estético y eventualmente cuadros obstructivos de la vía respiratoria en aquellos casos de infiltración grasa de la laringe. Presentamos el caso de un hombre con enfermedad de Madelung y cuadro disneico agudo por infiltración grasa laríngea y lipoma obstructivo.</p>" ] ] "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara">Please cite this article as: Landínez-Cepeda GA, et al. Lipoma laríngeo asociado a enfermedad de Madelung: a propósito de un caso. Acta Otorrinolaringol Esp. 2012;63:311–3.</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" => 533 "Ancho" => 801 "Tamanyo" => 57021 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Appearance of cervical fat accumulation in our patient in an anterior view.</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" => 745 "Ancho" => 1000 "Tamanyo" => 126282 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Coronal cervical CT image showing infiltration by unencapsulated (arrows) and endolaryngeal (circles) fat, which reduced the calibre of the glottic lumen.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:7 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Laryngeal lipoma associated with diffuse lipomatosis: case report and literature review" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "M.L. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2023 March | 4 | 0 | 4 |
| 2021 March | 0 | 1 | 1 |
| 2021 February | 0 | 1 | 1 |
| 2021 January | 0 | 1 | 1 |
| 2020 December | 0 | 5 | 5 |
| 2020 November | 0 | 16 | 16 |
| 2020 October | 0 | 5 | 5 |
| 2020 September | 0 | 5 | 5 |
| 2020 August | 0 | 7 | 7 |
| 2020 July | 0 | 1 | 1 |
| 2020 June | 0 | 2 | 2 |
| 2020 May | 0 | 4 | 4 |
| 2020 March | 12 | 9 | 21 |
| 2020 February | 24 | 8 | 32 |
| 2020 January | 38 | 8 | 46 |
| 2019 December | 33 | 9 | 42 |
| 2019 November | 15 | 16 | 31 |
| 2019 October | 35 | 4 | 39 |
| 2019 September | 36 | 12 | 48 |
| 2019 August | 31 | 7 | 38 |
| 2019 July | 43 | 14 | 57 |
| 2019 June | 96 | 26 | 122 |
| 2019 May | 198 | 63 | 261 |
| 2019 April | 89 | 31 | 120 |
| 2019 March | 18 | 11 | 29 |
| 2019 February | 16 | 13 | 29 |
| 2019 January | 21 | 9 | 30 |
| 2018 December | 12 | 12 | 24 |
| 2018 November | 23 | 9 | 32 |
| 2018 October | 26 | 12 | 38 |
| 2018 May | 4 | 0 | 4 |
| 2018 April | 7 | 3 | 10 |
| 2018 March | 8 | 0 | 8 |
| 2018 February | 8 | 1 | 9 |
| 2018 January | 9 | 5 | 14 |
| 2017 December | 15 | 0 | 15 |
| 2017 November | 15 | 2 | 17 |
| 2017 October | 14 | 1 | 15 |
| 2017 September | 10 | 4 | 14 |
| 2017 August | 17 | 5 | 22 |
| 2017 July | 21 | 4 | 25 |
| 2017 June | 19 | 6 | 25 |
| 2017 May | 26 | 4 | 30 |
| 2017 April | 17 | 4 | 21 |
| 2017 March | 32 | 3 | 35 |
| 2017 February | 32 | 4 | 36 |
| 2017 January | 21 | 4 | 25 |
| 2016 December | 38 | 8 | 46 |
| 2016 November | 27 | 9 | 36 |
| 2016 October | 32 | 8 | 40 |
| 2016 September | 41 | 14 | 55 |
| 2016 August | 29 | 4 | 33 |
| 2016 July | 24 | 5 | 29 |
| 2016 June | 36 | 18 | 54 |
| 2016 May | 42 | 17 | 59 |
| 2016 April | 40 | 12 | 52 |
| 2016 March | 44 | 18 | 62 |
| 2016 February | 38 | 27 | 65 |
| 2016 January | 47 | 22 | 69 |
| 2015 December | 32 | 14 | 46 |
| 2015 November | 26 | 14 | 40 |
| 2015 October | 33 | 19 | 52 |
| 2015 September | 27 | 42 | 69 |
| 2015 August | 53 | 9 | 62 |
| 2015 July | 47 | 6 | 53 |
| 2015 June | 29 | 2 | 31 |
| 2015 May | 34 | 7 | 41 |
| 2015 April | 53 | 7 | 60 |
| 2015 March | 43 | 15 | 58 |
| 2015 February | 34 | 8 | 42 |
| 2015 January | 39 | 5 | 44 |
| 2014 December | 39 | 8 | 47 |
| 2014 November | 23 | 3 | 26 |
| 2014 October | 19 | 7 | 26 |
| 2014 September | 35 | 2 | 37 |
| 2014 August | 25 | 9 | 34 |
| 2014 July | 39 | 4 | 43 |
| 2014 June | 8 | 2 | 10 |
| 2014 May | 18 | 1 | 19 |
| 2014 April | 12 | 4 | 16 |
| 2014 March | 11 | 6 | 17 |
| 2014 February | 12 | 8 | 20 |
| 2014 January | 12 | 2 | 14 |
| 2013 December | 12 | 2 | 14 |
| 2013 November | 23 | 3 | 26 |
| 2013 October | 40 | 9 | 49 |
| 2013 September | 15 | 5 | 20 |
| 2013 August | 13 | 2 | 15 |
| 2013 July | 2 | 1 | 3 |
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