Correspondence: Dr. L.F. Flores-Suárez. Department of Immunology and Rheumatology. Instituto Nacional de Ciencias Médicas y Nutrición Salvador Zubirán. Vasco de Quiroga 15, Col. Sección XVI. Tlalpan. México, DF. CP. 14000. México.
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Mexico" "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff1" ] 1 => array:3 [ "entidad" => "Servicio de Otorrinolanringología, Instituto Nacional de Ciencias Médicas y Nutrición Salvador Zubirán (INCMNSZ), Mexico City. Mexico" "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff2" ] 2 => array:3 [ "entidad" => "Servicio de Cirugía Experimental, Instituto Nacional de Ciencias Médicas y Nutrición Salvador Zubirán (INCMNSZ), México DF. México" "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff3" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor1" "etiqueta" => "†" "correspondencia" => "Correspondence: Dr. L.F. Flores-Suárez. Department of Immunology and Rheumatology. Instituto Nacional de Ciencias Médicas y Nutrición Salvador Zubirán. Vasco de Quiroga 15, Col. Sección XVI. Tlalpan. México, DF. CP. 14000. México." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Reflujo laringofaríngeo como simulador de granulomatosis de Wegener limitada" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "f0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 648 "Ancho" => 934 "Tamanyo" => 59248 ] ] "descripcion" => array:1 [ "en" => "<p id="sp0005" class="elsevierStyleSimplePara elsevierViewall">Nasofibrolaryngoscopic findings during an acute dyspnea episode. The Figure shows arytenoid oedema and hyperemia, moderate oedema of the posterior commisure, severe sulcus subglotticus and ventricular obliteration, with a 70% luminal narrowing.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="s0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="st0015">Introduction</span><p id="p0005" class="elsevierStylePara elsevierViewall">Subglottic stenosis (SGS) in Wegener granulomatosis (WG) occurs in 10%-15% of patients.<a class="elsevierStyleCrossRef" href="#bb0005"><span class="elsevierStyleSup">1</span></a> The presence of ANCA in defined clinical settings can be taken as proof to establish the diagnosis. However, misinterpretation when other more common causes of SGS are present can occur. SGS with positive ANCA testing needs exclusion of LPR as cause or contributing factor.</p></span><span id="s0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="st0020">Case report</span><p id="p0010" class="elsevierStylePara elsevierViewall">A 15-year-old woman presented in December 2000 with repetitive upper airway infections and sinusitis. She developed dysphonia, chronic cough, stridor, and frequent dyspnea episodes. ANCA were ordered and positive at a 1:20 dilution by indirect immunofluorescence (IIF) with a cytoplasmic pattern. She was referred to a rheumatologist who started deflazacort (in decreasing doses for one year) and trimethoprim/sulfamethoxazole (TMP-SMZ). Each time steroids were decreased, symptoms returned. In one exacerbation, she had hoarseness and 88% oxygen saturation. Other organ involvement was absent. She was referred to us in January 2003. At that time, all paraclinical studies were normal or negative. A nasofibrolaryngoscopy showed severe inflammatory changes (<a class="elsevierStyleCrossRef" href="#f0005">Figure 1</a>). Evaluation of the distal airway by direct laryngoscopy and flexible bronchoscopy showed no additional pathology. A subglottic tissue biopsy demonstrated nonspecific acute and chronic inflammation with no granulomas or vasculitis. ANCA testing with IIF and ELISA against myeloperoxidase and proteinase-3 was twice negative. An oesophageal manometry showed incompetent lower oesophagic sphincter and 100% relaxation. The 24-hour oesophageal pH confirmed atypical extraoesophageal reflux disease. She had 83 episodes of proximal reflux, 4 of them over 15 minutes, and pH <<span class="elsevierStyleHsp" style=""></span>4.0 for 83 total minutes. The DeMeester score was 23.6 (normal <<span class="elsevierStyleHsp" style=""></span>14.7). During the study she did not record gastrooesophageal symptoms. She was given omeprazole 40<span class="elsevierStyleHsp" style=""></span>mg bid and domperidone 10<span class="elsevierStyleHsp" style=""></span>mg qid. A new nasofibrolaryngoscopy showed improvement (<a class="elsevierStyleCrossRef" href="#f0010">Figure 2</a>). Under treatment for gastrooesophagic reflux disease (GERD) a new study showed absent proximal reflux episodes of ><span class="elsevierStyleHsp" style=""></span>3 minutes, total time of reflux decreasing to 33 minutes. The DeMeester score was 11. She is currently asymptomatic and no new episodes of dyspnea or sinusitis have occurred for more than 6 years.</p><elsevierMultimedia ident="f0005"></elsevierMultimedia><elsevierMultimedia ident="f0010"></elsevierMultimedia></span><span id="s0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="st0025">Discussion</span><p id="p0015" class="elsevierStylePara elsevierViewall">GERD is a common disease in general population.<a class="elsevierStyleCrossRef" href="#bb0010"><span class="elsevierStyleSup">2</span></a> When severe and persistent it can lead to LPR, which in consequence can cause “idiopathic” SGS.<a class="elsevierStyleCrossRef" href="#bb0015"><span class="elsevierStyleSup">3</span></a> However, symptoms are not recalled by a subtantial percentage of adult patients.<a class="elsevierStyleCrossRef" href="#bb0020"><span class="elsevierStyleSup">4</span></a> To accurately diagnose LPR a modified four-port pH measurement is advised.<a class="elsevierStyleCrossRef" href="#bb0025"><span class="elsevierStyleSup">5</span></a> In 1 study, LPR may have contributed to persistent disease in patients with SGS due to WG.<a class="elsevierStyleCrossRef" href="#bb0015"><span class="elsevierStyleSup">3</span></a> Our case emphasizes the notion that SGS can have LPR as cause or important collaborative factor.</p><p id="p0020" class="elsevierStylePara elsevierViewall">A panel of experts has recommended that ANCA be performed in cases of unexplained SGS.<a class="elsevierStyleCrossRef" href="#bb0030"><span class="elsevierStyleSup">6</span></a> However, although upper airway involvement can be present in 70% as part of the initial manifestations of WG,<a class="elsevierStyleCrossRef" href="#bb0035"><span class="elsevierStyleSup">7</span></a> a false positive ANCA result can lead to pitfalls as observed. In a study performed in a general hospital in the UK, McLaren et al<a class="elsevierStyleCrossRef" href="#bb0040"><span class="elsevierStyleSup">8</span></a> showed that routine ANCA testing by IIF ordered in different Internal Medicine departments (other than Rheumatology) yielded a low positive predicted value, making them a tool of poor return when not properly indicated. As in many of their cases, the IIF ANCA was a false positive. Their retrieval increased when both IIF and ELISA were performed. Our case clearly underlines the need to perform both methods to detect ANCA (IIF and ELISA) as proposed by the results of the EC/BCR study.<a class="elsevierStyleCrossRef" href="#bb0045"><span class="elsevierStyleSup">9</span></a> Also, that in doubtful cases of WG with borderline ANCA testing, histological confirmation of the suspected vasculitides is necessary when the procedure is feasible.</p><p id="p0025" class="elsevierStylePara elsevierViewall">In young or adult patients presenting with a clinical picture compatible with SGS, LPR must be sought even in absence of GERD symptoms. In such cases, a single, isolated positive ANCA test result, especially if done with only one method cannot be taken as proof of WG. This can lead to serious consequences due to the morbidity associated with treatment.<a class="elsevierStyleCrossRef" href="#bb0050"><span class="elsevierStyleSup">10</span></a> Moreover, in cases of WG, GERD can still be present and contribute to the symptoms resembling SGS.<a class="elsevierStyleCrossRef" href="#bb0015"><span class="elsevierStyleSup">3</span></a> The case provides an important lesson regarding SGS with ANCA positive testing, in where awareness of other causes of SGS is needed. The atypical presentation of the laryngopharyngeal disease, plus the improvement seen with previous steroid and TMP-SMZ treatment during acute dyspnea events further contributed to the impression of WG. Reconsideration of the diagnosis and the mechanism that caused the laryngeal disease led us to term this case as one of WG going backwards.</p><p id="p0030" class="elsevierStylePara elsevierViewall">ANCA testing must be interpreted with caution in patients presenting with SGS as the latter can be due to more common and less severe diseases. As there are individuals who do not recall a history of previous grastrointestinal symptoms this needs special consideration. Practical points about this case are:<ul class="elsevierStyleList" id="l0005"><li class="elsevierStyleListItem" id="o0005"><span class="elsevierStyleLabel">1.</span><p id="p0035" class="elsevierStylePara elsevierViewall">SGS can occur as complication of asymptomatic LPR.</p></li><li class="elsevierStyleListItem" id="o0010"><span class="elsevierStyleLabel">2.</span><p id="p0040" class="elsevierStylePara elsevierViewall">In patients with SGS and positive ANCA, exclusion of LPR as cause or contributing factor is needed.</p></li><li class="elsevierStyleListItem" id="o0015"><span class="elsevierStyleLabel">3.</span><p id="p0045" class="elsevierStylePara elsevierViewall">A combined ANCA test using IIF and ELISA is mandatory when an ANCA-associated vasculitides is suspected. Results need to be weighted against the pretest probability of having a true ANCA-associated disease.</p></li></ul></p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:6 [ 0 => array:2 [ "identificador" => "xpalclavsec112519" "titulo" => "Key words" ] 1 => array:2 [ "identificador" => "xpalclavsec112518" "titulo" => "Palabras clave" ] 2 => array:2 [ "identificador" => "s0005" "titulo" => "Introduction" ] 3 => array:2 [ "identificador" => "s0010" "titulo" => "Case report" ] 4 => array:2 [ "identificador" => "s0015" "titulo" => "Discussion" ] 5 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2007-06-11" "fechaAceptado" => "2007-09-26" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Key words" "identificador" => "xpalclavsec112519" "palabras" => array:3 [ 0 => "Wegener granulomatosis" 1 => "Subglottic stenosis" 2 => "Laryngopharyngeal reflux" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec112518" "palabras" => array:3 [ 0 => "Granulomatosis de Wegener" 1 => "Estenosis subglótica" 2 => "Reflujo laringofaríngeo" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:1 [ "resumen" => "<p id="sp0015" class="elsevierStyleSimplePara elsevierViewall">We present a case of subglottic stenosis (SGS) in a young patient with positive ANCA but a wrong diagnosis of Wegener granulomatosis (WG). Instead, she was diagnosed as having laryngopharyngeal reflux (LPR).</p><p id="sp0020" class="elsevierStyleSimplePara elsevierViewall">Pitfalls of ANCA testing in this scenario, the route to diagnosis of LPR and the contribution of this entity to subglottic stenosis (SGS) in WG are discussed. Caution when interpreting ANCA results is mandatory to avoid improper management.</p>" ] "es" => array:1 [ "resumen" => "<p id="sp0025" class="elsevierStyleSimplePara elsevierViewall">Presentamos un caso de estenosis subglótica (ESG) en una paciente joven con ANCA positivos, pero un diagnóstico erróneo de granulomatosis de Wegener (GW). El caso correspondió a reflujo laringofaríngeo (RLF). Se discuten los falsos positivos de la prueba de ANCA, la ruta diagnóstica del RLF y la contribución de esta entidad a la ESG en GW. Debe tenerse precaución en la interpretación de ANCA para evitar el tratamiento inapropiado.</p>" ] ] "multimedia" => array:2 [ 0 => array:7 [ "identificador" => "f0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 648 "Ancho" => 934 "Tamanyo" => 59248 ] ] "descripcion" => array:1 [ "en" => "<p id="sp0005" class="elsevierStyleSimplePara elsevierViewall">Nasofibrolaryngoscopic findings during an acute dyspnea episode. The Figure shows arytenoid oedema and hyperemia, moderate oedema of the posterior commisure, severe sulcus subglotticus and ventricular obliteration, with a 70% luminal narrowing.</p>" ] ] 1 => array:7 [ "identificador" => "f0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 648 "Ancho" => 934 "Tamanyo" => 62266 ] ] "descripcion" => array:1 [ "en" => "<p id="sp0010" class="elsevierStyleSimplePara elsevierViewall">Nasofibrolaryngoscopic findings after treatment for extraoesophageal reflux disease. One month after diagnosis and treatment initiation, there is a decrease in the arytenoid and commisural oedema, important decrese of the sulcus subglotticus, and a 50% luminal patency.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bs0005" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bb0005" "etiqueta" => "1." "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "The management of subglottic stenosis in patients with Wegener’s granulomatosis" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "R.S. Lebovics" 1 => "G.S. Hoffman" 2 => "R.Y. Leavitt" 3 => "G.S. Kerr" 4 => "W.D. Travis" 5 => "W. 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año/Mes | Html | Total | |
---|---|---|---|
2024 Noviembre | 6 | 13 | 19 |
2024 Octubre | 23 | 36 | 59 |
2024 Septiembre | 32 | 30 | 62 |
2024 Agosto | 63 | 36 | 99 |
2024 Julio | 37 | 37 | 74 |
2024 Junio | 42 | 29 | 71 |
2024 Mayo | 45 | 27 | 72 |
2024 Abril | 55 | 29 | 84 |
2024 Marzo | 46 | 34 | 80 |
2024 Febrero | 37 | 27 | 64 |
2024 Enero | 23 | 27 | 50 |
2023 Diciembre | 29 | 32 | 61 |
2023 Noviembre | 41 | 60 | 101 |
2023 Octubre | 43 | 50 | 93 |
2023 Septiembre | 72 | 46 | 118 |
2023 Agosto | 49 | 15 | 64 |
2023 Julio | 38 | 35 | 73 |
2023 Junio | 38 | 28 | 66 |
2023 Mayo | 33 | 22 | 55 |
2023 Abril | 30 | 8 | 38 |
2023 Marzo | 50 | 28 | 78 |
2023 Febrero | 35 | 24 | 59 |
2023 Enero | 36 | 29 | 65 |
2022 Diciembre | 55 | 31 | 86 |
2022 Noviembre | 52 | 34 | 86 |
2022 Octubre | 51 | 37 | 88 |
2022 Septiembre | 31 | 35 | 66 |
2022 Agosto | 45 | 48 | 93 |
2022 Julio | 41 | 44 | 85 |
2022 Junio | 39 | 36 | 75 |
2022 Mayo | 38 | 39 | 77 |
2022 Abril | 47 | 37 | 84 |
2022 Marzo | 59 | 46 | 105 |
2022 Febrero | 32 | 39 | 71 |
2022 Enero | 60 | 65 | 125 |
2021 Diciembre | 45 | 40 | 85 |
2021 Noviembre | 50 | 56 | 106 |
2021 Octubre | 60 | 53 | 113 |
2021 Septiembre | 57 | 36 | 93 |
2021 Agosto | 50 | 58 | 108 |
2021 Julio | 41 | 35 | 76 |
2021 Junio | 65 | 37 | 102 |
2021 Mayo | 53 | 26 | 79 |
2021 Abril | 169 | 71 | 240 |
2021 Marzo | 283 | 37 | 320 |
2021 Febrero | 51 | 33 | 84 |
2021 Enero | 53 | 26 | 79 |
2020 Diciembre | 52 | 23 | 75 |
2020 Noviembre | 44 | 25 | 69 |
2020 Octubre | 31 | 29 | 60 |
2020 Septiembre | 58 | 23 | 81 |
2020 Agosto | 45 | 12 | 57 |
2020 Julio | 41 | 16 | 57 |
2020 Junio | 52 | 22 | 74 |
2020 Mayo | 55 | 25 | 80 |
2020 Abril | 49 | 26 | 75 |
2020 Marzo | 50 | 16 | 66 |
2020 Febrero | 63 | 32 | 95 |
2020 Enero | 45 | 24 | 69 |
2019 Diciembre | 51 | 30 | 81 |
2019 Noviembre | 40 | 24 | 64 |
2019 Octubre | 46 | 26 | 72 |
2019 Septiembre | 44 | 31 | 75 |
2019 Agosto | 33 | 29 | 62 |
2019 Julio | 35 | 32 | 67 |
2019 Junio | 43 | 50 | 93 |
2019 Mayo | 71 | 104 | 175 |
2019 Abril | 56 | 56 | 112 |
2019 Marzo | 36 | 20 | 56 |
2019 Febrero | 28 | 22 | 50 |
2019 Enero | 48 | 32 | 80 |
2018 Diciembre | 101 | 60 | 161 |
2018 Noviembre | 86 | 18 | 104 |
2018 Octubre | 67 | 12 | 79 |
2018 Septiembre | 29 | 11 | 40 |
2018 Agosto | 25 | 7 | 32 |
2018 Julio | 15 | 16 | 31 |
2018 Mayo | 4 | 3 | 7 |
2018 Abril | 55 | 8 | 63 |
2018 Marzo | 43 | 10 | 53 |
2018 Febrero | 22 | 5 | 27 |
2018 Enero | 12 | 3 | 15 |
2017 Diciembre | 29 | 7 | 36 |
2017 Noviembre | 27 | 18 | 45 |
2017 Octubre | 28 | 4 | 32 |
2017 Septiembre | 16 | 10 | 26 |
2017 Agosto | 18 | 15 | 33 |
2017 Julio | 21 | 10 | 31 |
2017 Junio | 33 | 9 | 42 |
2017 Mayo | 24 | 13 | 37 |
2017 Abril | 33 | 10 | 43 |
2017 Marzo | 33 | 36 | 69 |
2017 Febrero | 15 | 9 | 24 |
2017 Enero | 20 | 8 | 28 |
2016 Diciembre | 50 | 23 | 73 |
2016 Noviembre | 26 | 7 | 33 |
2016 Octubre | 58 | 23 | 81 |
2016 Septiembre | 49 | 10 | 59 |
2016 Agosto | 18 | 3 | 21 |
2016 Julio | 13 | 16 | 29 |
2016 Abril | 1 | 0 | 1 |
2016 Marzo | 3 | 0 | 3 |
2016 Febrero | 2 | 0 | 2 |
2016 Enero | 1 | 0 | 1 |
2015 Diciembre | 3 | 0 | 3 |
2015 Noviembre | 3 | 0 | 3 |
2015 Octubre | 1 | 0 | 1 |
2015 Agosto | 4 | 0 | 4 |
2015 Julio | 25 | 6 | 31 |
2015 Junio | 44 | 9 | 53 |
2015 Mayo | 45 | 8 | 53 |
2015 Abril | 41 | 8 | 49 |
2015 Marzo | 41 | 7 | 48 |
2015 Febrero | 32 | 7 | 39 |
2015 Enero | 47 | 14 | 61 |
2014 Diciembre | 44 | 10 | 54 |
2014 Noviembre | 26 | 10 | 36 |
2014 Octubre | 36 | 9 | 45 |
2014 Septiembre | 31 | 8 | 39 |
2014 Agosto | 31 | 8 | 39 |
2014 Julio | 35 | 8 | 43 |
2014 Junio | 47 | 11 | 58 |
2014 Mayo | 49 | 18 | 67 |
2014 Abril | 48 | 8 | 56 |
2014 Marzo | 52 | 19 | 71 |
2014 Febrero | 54 | 19 | 73 |
2014 Enero | 50 | 16 | 66 |
2013 Diciembre | 35 | 9 | 44 |
2013 Noviembre | 45 | 15 | 60 |
2013 Octubre | 52 | 20 | 72 |
2013 Septiembre | 46 | 14 | 60 |
2013 Agosto | 34 | 13 | 47 |
2013 Julio | 35 | 9 | 44 |
2013 Junio | 27 | 8 | 35 |
2013 Mayo | 25 | 5 | 30 |
2013 Abril | 32 | 6 | 38 |
2013 Marzo | 27 | 6 | 33 |
2013 Febrero | 26 | 3 | 29 |
2013 Enero | 25 | 5 | 30 |
2012 Diciembre | 27 | 6 | 33 |
2012 Noviembre | 8 | 2 | 10 |
2012 Octubre | 9 | 14 | 23 |
2012 Septiembre | 8 | 2 | 10 |
2011 Mayo | 7 | 0 | 7 |
2011 Abril | 6 | 0 | 6 |
2011 Marzo | 13 | 0 | 13 |
2011 Febrero | 10 | 0 | 10 |
2011 Enero | 18 | 0 | 18 |
2010 Diciembre | 3 | 0 | 3 |
2010 Noviembre | 9 | 0 | 9 |
2010 Octubre | 5 | 0 | 5 |
2010 Septiembre | 14 | 0 | 14 |
2010 Agosto | 21 | 0 | 21 |
2010 Julio | 6 | 0 | 6 |
2010 Junio | 17 | 0 | 17 |
2010 Mayo | 9 | 0 | 9 |
2010 Abril | 1 | 0 | 1 |
2010 Marzo | 2 | 0 | 2 |
2010 Enero | 1 | 0 | 1 |
2009 Diciembre | 2 | 0 | 2 |
2009 Noviembre | 3 | 0 | 3 |
2009 Octubre | 2 | 0 | 2 |
2009 Septiembre | 8 | 0 | 8 |
2009 Agosto | 4 | 0 | 4 |
2009 Julio | 4 | 0 | 4 |
2009 Junio | 4 | 0 | 4 |
2009 Mayo | 4 | 0 | 4 |
2009 Abril | 10 | 0 | 10 |
2009 Marzo | 3 | 0 | 3 |
2009 Febrero | 2 | 0 | 2 |
2009 Enero | 2 | 0 | 2 |
2008 Diciembre | 17 | 0 | 17 |
2008 Noviembre | 24 | 0 | 24 |
2008 Octubre | 29 | 0 | 29 |
2008 Septiembre | 24 | 0 | 24 |
2008 Agosto | 26 | 0 | 26 |
2008 Julio | 41 | 0 | 41 |
2008 Junio | 66 | 0 | 66 |
2008 Mayo | 20 | 0 | 20 |