Letter to the Editor
Familial idiopathic tracheal subglottic stenosis: Description of three cases
Estenosis subglótica traqueal idiopática familiar: descripción de tres casos
Esperanza Salcedo Lobera
, Francisco M. Páez Codeso, Antonio Dorado Galindo
Corresponding author
Unidad de Gestión Clínica Médico-Quirúrgica de Enfermedades Respiratorias, Hospital Regional Universitario de Málaga, Málaga, Spain
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Páez Codeso, Antonio Dorado Galindo" "autores" => array:3 [ 0 => array:4 [ "nombre" => "Esperanza" "apellidos" => "Salcedo Lobera" "email" => array:1 [ 0 => "esalcedolobera@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "Francisco M." "apellidos" => "Páez Codeso" ] 2 => array:2 [ "nombre" => "Antonio" "apellidos" => "Dorado Galindo" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Unidad de Gestión Clínica Médico-Quirúrgica de Enfermedades Respiratorias, Hospital Regional Universitario de Málaga, Málaga, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Estenosis subglótica traqueal idiopática familiar: descripción de tres casos" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1656 "Ancho" => 2007 "Tamanyo" => 255921 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0345" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A) Sagittal section of the neck CT showing tracheal stenosis (arrow) of case 1. B) Flow/volume loop with fixed airflow obstruction morphology of the case 3. C,D) Endoscopic images showing subglottic stenosis in case 1 and case 3, respectively.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Subglottic stenosis is an inflammatory cicatricial obstruction of the cricoid cartilage. The causes of said stenosis are diverse: infections, prolonged intubation, trauma or systemic inflammatory diseases, but in 5% of cases it is idiopathic.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">Idiopathic subglottic stenosis is more common in women between the ages of 20–50 years, and the familial predisposition of this entity is unknown, with few cases reported and published. We present the cases of three women with idiopathic subglottic stenosis who were first degree relatives.</p><p id="par0015" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">Case 1.</span> 55 year old female, with no medical-surgical history of interest, who was referred to medical care due to symptoms of dyspnoea on minimal exertion with occasional episodes of stridor over the past 3 years, with no symptoms of gastroesophageal disease. The computed tomography (CT) of the neck revealed tracheal stenosis of 4.50<span class="elsevierStyleHsp" style=""></span>mm. Pulmonary function tests (PFT) were then performed, in which a flow-volume loop with fixed airflow obstruction morphology was observed. A conventional bronchoscopy was also performed, which revealed a concentric subglottic stenosis due to the subglottic membrane <span class="elsevierStyleItalic">(wedge like)</span> at 1.5<span class="elsevierStyleHsp" style=""></span>cm from the glottic lumen (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). Two endoscopic procedures were performed with balloon tracheal pneumoplasty and intralesional instillation of triamcinolone with partial improvement but followed by recurrence. Therefore, it was decided to perform surgical intervention with satisfactory follow-up at one year, and clinical improvement finally observed.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">Case 2.</span> 53 year old female (sister of case 1), with no medical-surgical history of interest, who was referred due to symptoms of dyspnoea on moderate exertion over the past 2 years, with no symptoms of gastroesophageal disease. The neck CT revealed tracheal stenosis of 6<span class="elsevierStyleHsp" style=""></span>mm; with the same PFT, the endoscopy showed subglottic stenosis at 2<span class="elsevierStyleHsp" style=""></span>cm from the glottic lumen. Due to the symptoms of moderate dyspnoea and the patient's wishes, follow-up was continued with endoscopy control.</p><p id="par0025" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">Case 3.</span> 39 year old female (daughter of case 1), with no medical-surgical history of interest, who was referred due to symptoms of dyspnoea on minimal exertion over the past year, with no symptoms of gastroesophageal disease. The neck CT revealed tracheal stenosis of 5.56<span class="elsevierStyleHsp" style=""></span>mm; with the same PFT, the endoscopy showed subglottic stenosis at 2.5<span class="elsevierStyleHsp" style=""></span>cm from the glottic lumen. It was decided to continue with endoscopic treatment with balloon tracheal pneumoplasty and intralesional instillation of triamcinolone, resulting in clinical improvement and satisfactory follow-up.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Idiopathic subglottic stenosis is a rare disease, with an unknown incidence, although it is common in women with a mean age of 20–50 years; in our series, the mean age was 49 years. The aetiology of this stenosis is still unknown,<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> although several studies have been carried out on the action of oestrogens in the development pathway of this disease, but without finding evidence to support it. The same occurs with gastroesophageal reflux, where some authors observed clinical improvement in these patients after initiation of antireflux treatment<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a>; even so, our series did not present reflux symptoms.</p><p id="par0035" class="elsevierStylePara elsevierViewall">The treatment includes endoscopic measures, such as resection by thermal ablation (broncholaser, electroblade, etc.) or balloon tracheal pneumoplasty, but it presents a high recurrence rate, around 87% at 5 years. The alternative would be surgery, which is more invasive, but with a lower recurrence rate, around 10%–40% after 8 years.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">This is the first case of familial idiopathic subglottic stenosis described in our country, since the few cases that exist in literature, even in twin sisters, were reported in other countries. All these cases open the door to a possible genetic predisposition for the development of this idiopathic stenosis in adulthood, which could be a working hypothesis in future research.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,5</span></a></p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Ethical considerations</span><p id="par0045" class="elsevierStylePara elsevierViewall">We declare that all the authors of this paper have taken into account the ethical responsibilities necessary for the handling of the information of said paper.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Funding</span><p id="par0050" class="elsevierStylePara elsevierViewall">We declare that we have not received funding from any pharmaceutical industry for this work.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflict of interests</span><p id="par0055" class="elsevierStylePara elsevierViewall">We declare that none of the participants have a conflict of interest with this manuscript.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:4 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Ethical considerations" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Funding" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Conflict of interests" ] 3 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "multimedia" => array:1 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1656 "Ancho" => 2007 "Tamanyo" => 255921 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0345" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A) Sagittal section of the neck CT showing tracheal stenosis (arrow) of case 1. B) Flow/volume loop with fixed airflow obstruction morphology of the case 3. C,D) Endoscopic images showing subglottic stenosis in case 1 and case 3, respectively.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Idiopathic subglottic stenosis: a familial predisposition" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:6 [ 0 => "E. Dumoulin" 1 => "D.R. Stather" 2 => "G. Gelfand" 3 => "B. Maranda" 4 => "P. Maceachern" 5 => "A. Tremblay" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.athoracsur.2012.07.076" "Revista" => array:6 [ "tituloSerie" => "Ann Thorac Surg." 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