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Mounier-Kuhn Syndrome: A Rare Cause of Recurrent Chest Infection
Síndrome de Mounier-Kuhn: una causa rara de infección torácica recurrente
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Mercer, Eihab O. Bedawi, Rachelle Asciak, John Stradling, Najib M. Rahman" "autores" => array:8 [ 0 => array:2 [ "nombre" => "Maged" "apellidos" => "Hassan" ] 1 => array:2 [ "nombre" => "Radhika" "apellidos" => "Banka" ] 2 => array:2 [ "nombre" => "Olalla" "apellidos" => "Castro-Añón" ] 3 => array:2 [ "nombre" => "Rachel M." "apellidos" => "Mercer" ] 4 => array:2 [ "nombre" => "Eihab O." "apellidos" => "Bedawi" ] 5 => array:2 [ "nombre" => "Rachelle" "apellidos" => "Asciak" ] 6 => array:2 [ "nombre" => "John" "apellidos" => "Stradling" ] 7 => array:2 [ "nombre" => "Najib M." "apellidos" => "Rahman" ] ] ] ] ] "idiomaDefecto" => "en" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1579212921003190?idApp=UINPBA00003Z" "url" => "/15792129/0000005700000010/v1_202110020725/S1579212921003190/v1_202110020725/en/main.assets" ] "itemAnterior" => array:18 [ "pii" => "S1579212921003244" "issn" => "15792129" "doi" => "10.1016/j.arbr.2021.01.012" "estado" => "S300" "fechaPublicacion" => "2021-10-01" "aid" => "2746" "copyright" => "SEPAR" "documento" => "article" "crossmark" => 1 "subdocumento" => "sco" "cita" => "Arch Bronconeumol. 2021;57:654" "abierto" => array:3 [ "ES" => false "ES2" => false "LATM" => false ] "gratuito" => false "lecturas" => array:1 [ "total" => 0 ] "en" => array:11 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Clinical Image</span>" "titulo" => "Catathrenia – A Rare But Disturbing Sleep Disorder" "tienePdf" => "en" "tieneTextoCompleto" => "en" "paginas" => array:1 [ 0 => array:1 [ "paginaInicial" => "654" ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Catatrenia: un trastorno del sueño poco común pero perturbador" ] ] "contieneTextoCompleto" => array:1 [ "en" => true ] "contienePdf" => array:1 [ "en" => true ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Video 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1105 "Ancho" => 2508 "Tamanyo" => 193690 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Recording of the cardiorespiratory sleep study showing a long apnea period with synchronous groan sound (Catathrenia).</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Denny Marques Rodrigues, Margarida Pimenta Valério, Teresa Costa" "autores" => array:3 [ 0 => array:2 [ "nombre" => "Denny Marques" "apellidos" => "Rodrigues" ] 1 => array:2 [ "nombre" => "Margarida Pimenta" "apellidos" => "Valério" ] 2 => array:2 [ "nombre" => "Teresa" "apellidos" => "Costa" ] ] ] ] ] "idiomaDefecto" => "en" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1579212921003244?idApp=UINPBA00003Z" "url" => "/15792129/0000005700000010/v1_202110020725/S1579212921003244/v1_202110020725/en/main.assets" ] "en" => array:14 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Clinical Image</span>" "titulo" => "Mounier-Kuhn Syndrome: A Rare Cause of Recurrent Chest Infection" "tieneTextoCompleto" => true "paginas" => array:1 [ 0 => array:1 [ "paginaInicial" => "655" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Narat Srivali, Federica De Giacomi" "autores" => array:2 [ 0 => array:4 [ "nombre" => "Narat" "apellidos" => "Srivali" "email" => array:1 [ 0 => "nsrivali@gmail.com" ] "referencia" => array:3 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] 2 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "Federica" "apellidos" => "De Giacomi" "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Division of Pulmonary Medicine (NS), St. Agnes Hospital, Baltimore, MD 21229, USA" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Respiratory Unit (FDG), Cremona Hospital, Cremona, Italy" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Síndrome de Mounier-Kuhn: una causa rara de infección torácica recurrente" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1091 "Ancho" => 1333 "Tamanyo" => 154750 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Chest radiograph revealed peripheral opacities in the mid and lower lung zones bilaterally. (B) Thoracic CT at a level above the aortic arch shows tracheomegaly severe bullous paraseptal emphysema. Groundglass opacification and nodular consolidation right middle lobe. (C) Thoracic CT at a level slightly below Carina demonstrated dilated right and left main bronchi with severe bullous paraseptal emphysema. Groundglass opacification and nodular consolidation right middle lobe. (D) CT image of the trachea (sagittal view) showing tracheomegaly.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">A 65-year-old man with a past medical history of recurrent pneumonia up to 8 times in the past 5 years presented to our institution with the chief complaint of a productive cough and shortness of breath over the preceding 2 weeks. Physical examination was remarkable with crackle heard on auscultation of both lungs. Complete blood count was remarkable for leukocytosis. The comprehensive metabolic panel was unremarkable. Chest radiography (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A), demonstrated peripheral opacities in the mid and lower lung zones bilaterally. Computerized tomography (CT) of the chest revealed dilation of the trachea (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>B and D) and bronchiomegaly (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>C) with severe bullous paraseptal emphysema. Groundglass opacification and nodular consolidation right middle lobe. The clinical history of recurrent chest infections and a combination of imaging findings were consistent with Mounier-Kuhn Syndrome (MKS). The patient was treated with antibiotic and educated to improve airway clearance techniques.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">The MKS is a rare disorder characterized by the enlarged trachea and main bronchi. It was first described by Pierre-Louis Mounier-Kuhn in 1937 associated with recurrent chest infections<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">1</span></a> and anatomically described as tracheobronchomegaly (TBM) in 1962.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">1</span></a> The diagnosis can be made on CT by measuring the diameter of the airway. Woodring et al. suggested the following diagnostic criteria for tracheomegaly in adults based on chest radiography tracheal transverse diameter<span class="elsevierStyleHsp" style=""></span>><span class="elsevierStyleHsp" style=""></span>25<span class="elsevierStyleHsp" style=""></span>mm and sagittal diameter<span class="elsevierStyleHsp" style=""></span>><span class="elsevierStyleHsp" style=""></span>27<span class="elsevierStyleHsp" style=""></span>mm in males and, tracheal transverse diameter<span class="elsevierStyleHsp" style=""></span>><span class="elsevierStyleHsp" style=""></span>21<span class="elsevierStyleHsp" style=""></span>mm and sagittal diameter<span class="elsevierStyleHsp" style=""></span>><span class="elsevierStyleHsp" style=""></span>23<span class="elsevierStyleHsp" style=""></span>mm in the female.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">2</span></a> Different criteria have been suggested diameter of the trachea, right main bronchus, or left main bronchus that exceeds 3.0, 2.4, or 2.3<span class="elsevierStyleHsp" style=""></span>cm, respectively on a standard chest radiograph or bronchogram.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">3</span></a> The diameters in our case were 3.5<span class="elsevierStyleHsp" style=""></span>cm, 2.5<span class="elsevierStyleHsp" style=""></span>cm, and 2.4<span class="elsevierStyleHsp" style=""></span>cm, respectively, fulfilling the diagnostic criteria. This disease is thought to occur from atrophy of the elastic fibers of the trachea and bronchi, leading to thinning of the smooth muscle, and ultimately causing the trachea to become flaccid, dilated and develop tracheobronchomalacia.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">1</span></a> In mild cases, patients are asymptomatic or present only with chronic cough. In rare, severe cases, patients may suffer from pulmonary obstructive diseases such as bronchiectasis.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">2</span></a> Treatment is mainly supportive. Chest physiotherapy can be proposed to improve mucociliary clearance and antibiotics are administered for the treatment of pulmonary infections. Some patients may benefit from noninvasive positive-pressure ventilation, airway stenting, and surgical tracheoplasty.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">1</span></a></p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Funding</span><p id="par0015" class="elsevierStylePara elsevierViewall">None.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Conflict of interest</span><p id="par0020" class="elsevierStylePara elsevierViewall">None declared.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:3 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Funding" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Conflict of interest" ] 2 => array:1 [ "titulo" => "<span class="elsevierStyleBold">References</span>" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "multimedia" => array:1 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1091 "Ancho" => 1333 "Tamanyo" => 154750 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Chest radiograph revealed peripheral opacities in the mid and lower lung zones bilaterally. (B) Thoracic CT at a level above the aortic arch shows tracheomegaly severe bullous paraseptal emphysema. Groundglass opacification and nodular consolidation right middle lobe. (C) Thoracic CT at a level slightly below Carina demonstrated dilated right and left main bronchi with severe bullous paraseptal emphysema. Groundglass opacification and nodular consolidation right middle lobe. (D) CT image of the trachea (sagittal view) showing tracheomegaly.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "<span class="elsevierStyleBold">References</span>" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:3 [ 0 => array:3 [ "identificador" => "bib0020" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Tracheobronchomegaly, cough and recurrent chest infection: Mounier-Kuhn syndrome" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "I. Satia" 1 => "B. Dua" 2 => "N. Singh" 3 => "K. Killian" 4 => "P.M. O’Byrne" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1183/23120541.00138-2020" "Revista" => array:3 [ "tituloSerie" => "ERJ Open Res" "fecha" => "2020" "volumen" => "6" ] ] ] ] ] ] 1 => array:3 [ "identificador" => "bib0025" "etiqueta" => "2" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Congenital tracheobronchomegaly (Mounier-Kuhn syndrome): a report of 10 cases and review of the literature" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "J.H. Woodring" 1 => "R.S. Howard 2nd" 2 => "S.R. Rehm" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:6 [ "tituloSerie" => "J Thorac Imaging" "fecha" => "1991" "volumen" => "6" "paginaInicial" => "1" "paginaFinal" => "10" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/1942191" "web" => "Medline" ] ] ] ] ] ] ] ] 2 => array:3 [ "identificador" => "bib0030" "etiqueta" => "3" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Tracheobronchiomegaly. The Mounier-Kuhn syndrome" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "I. Katz" 1 => "M. Levine" 2 => "P. Herman" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:5 [ "tituloSerie" => "Am J Roentgenol Radium Ther Nucl Med" "fecha" => "1962" "volumen" => "88" "paginaInicial" => "1084" "paginaFinal" => "1094" ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/15792129/0000005700000010/v1_202110020725/S1579212921003220/v1_202110020725/en/main.assets" "Apartado" => array:4 [ "identificador" => "60604" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Clinical Images" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/15792129/0000005700000010/v1_202110020725/S1579212921003220/v1_202110020725/en/main.pdf?idApp=UINPBA00003Z&text.app=https://archbronconeumol.org/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1579212921003220?idApp=UINPBA00003Z" ]
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