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    "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 &#40;SGS&#41; in Wegener granulomatosis &#40;WG&#41; occurs in 10&#37;-15&#37; of patients&#46;<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&#46; However&#44; misinterpretation when other more common causes of SGS are present can occur&#46; SGS with positive ANCA testing needs exclusion of LPR as cause or contributing factor&#46;</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&#46; She developed dysphonia&#44; chronic cough&#44; stridor&#44; and frequent dyspnea episodes&#46; ANCA were ordered and positive at a 1&#58;20 dilution by indirect immunofluorescence &#40;IIF&#41; with a cytoplasmic pattern&#46; She was referred to a rheumatologist who started deflazacort &#40;in decreasing doses for one year&#41; and trimethoprim&#47;sulfamethoxazole &#40;TMP-SMZ&#41;&#46; Each time steroids were decreased&#44; symptoms returned&#46; In one exacerbation&#44; she had hoarseness and 88&#37; oxygen saturation&#46; Other organ involvement was absent&#46; She was referred to us in January 2003&#46; At that time&#44; all paraclinical studies were normal or negative&#46; A nasofibrolaryngoscopy showed severe inflammatory changes &#40;<a class="elsevierStyleCrossRef" href="#f0005">Figure 1</a>&#41;&#46; Evaluation of the distal airway by direct laryngoscopy and flexible bronchoscopy showed no additional pathology&#46; A subglottic tissue biopsy demonstrated nonspecific acute and chronic inflammation with no granulomas or vasculitis&#46; ANCA testing with IIF and ELISA against myeloperoxidase and proteinase-3 was twice negative&#46; An oesophageal manometry showed incompetent lower oesophagic sphincter and 100&#37; relaxation&#46; The 24-hour oesophageal pH confirmed atypical extraoesophageal reflux disease&#46; She had 83 episodes of proximal reflux&#44; 4 of them over 15 minutes&#44; and pH &#60;<span class="elsevierStyleHsp" style=""></span>4&#46;0 for 83 total minutes&#46; The DeMeester score was 23&#46;6 &#40;normal &#60;<span class="elsevierStyleHsp" style=""></span>14&#46;7&#41;&#46; During the study she did not record gastrooesophageal symptoms&#46; She was given omeprazole 40<span class="elsevierStyleHsp" style=""></span>mg bid and domperidone 10<span class="elsevierStyleHsp" style=""></span>mg qid&#46; A new nasofibrolaryngoscopy showed improvement &#40;<a class="elsevierStyleCrossRef" href="#f0010">Figure 2</a>&#41;&#46; Under treatment for gastrooesophagic reflux disease &#40;GERD&#41; a new study showed absent proximal reflux episodes of &#62;<span class="elsevierStyleHsp" style=""></span>3 minutes&#44; total time of reflux decreasing to 33 minutes&#46; The DeMeester score was 11&#46; She is currently asymptomatic and no new episodes of dyspnea or sinusitis have occurred for more than 6 years&#46;</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&#46;<a class="elsevierStyleCrossRef" href="#bb0010"><span class="elsevierStyleSup">2</span></a> When severe and persistent it can lead to LPR&#44; which in consequence can cause &#8220;idiopathic&#8221; SGS&#46;<a class="elsevierStyleCrossRef" href="#bb0015"><span class="elsevierStyleSup">3</span></a> However&#44; symptoms are not recalled by a subtantial percentage of adult patients&#46;<a class="elsevierStyleCrossRef" href="#bb0020"><span class="elsevierStyleSup">4</span></a> To accurately diagnose LPR a modified four-port pH measurement is advised&#46;<a class="elsevierStyleCrossRef" href="#bb0025"><span class="elsevierStyleSup">5</span></a> In 1 study&#44; LPR may have contributed to persistent disease in patients with SGS due to WG&#46;<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&#46;</p><p id="p0020" class="elsevierStylePara elsevierViewall">A panel of experts has recommended that ANCA be performed in cases of unexplained SGS&#46;<a class="elsevierStyleCrossRef" href="#bb0030"><span class="elsevierStyleSup">6</span></a> However&#44; although upper airway involvement can be present in 70&#37; as part of the initial manifestations of WG&#44;<a class="elsevierStyleCrossRef" href="#bb0035"><span class="elsevierStyleSup">7</span></a> a false positive ANCA result can lead to pitfalls as observed&#46; In a study performed in a general hospital in the UK&#44; 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 &#40;other than Rheumatology&#41; yielded a low positive predicted value&#44; making them a tool of poor return when not properly indicated&#46; As in many of their cases&#44; the IIF ANCA was a false positive&#46; Their retrieval increased when both IIF and ELISA were performed&#46; Our case clearly underlines the need to perform both methods to detect ANCA &#40;IIF and ELISA&#41; as proposed by the results of the EC&#47;BCR study&#46;<a class="elsevierStyleCrossRef" href="#bb0045"><span class="elsevierStyleSup">9</span></a> Also&#44; that in doubtful cases of WG with borderline ANCA testing&#44; histological confirmation of the suspected vasculitides is necessary when the procedure is feasible&#46;</p><p id="p0025" class="elsevierStylePara elsevierViewall">In young or adult patients presenting with a clinical picture compatible with SGS&#44; LPR must be sought even in absence of GERD symptoms&#46; In such cases&#44; a single&#44; isolated positive ANCA test result&#44; especially if done with only one method cannot be taken as proof of WG&#46; This can lead to serious consequences due to the morbidity associated with treatment&#46;<a class="elsevierStyleCrossRef" href="#bb0050"><span class="elsevierStyleSup">10</span></a> Moreover&#44; in cases of WG&#44; GERD can still be present and contribute to the symptoms resembling SGS&#46;<a class="elsevierStyleCrossRef" href="#bb0015"><span class="elsevierStyleSup">3</span></a> The case provides an important lesson regarding SGS with ANCA positive testing&#44; in where awareness of other causes of SGS is needed&#46; The atypical presentation of the laryngopharyngeal disease&#44; plus the improvement seen with previous steroid and TMP-SMZ treatment during acute dyspnea events further contributed to the impression of WG&#46; Reconsideration of the diagnosis and the mechanism that caused the laryngeal disease led us to term this case as one of WG going backwards&#46;</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&#46; As there are individuals who do not recall a history of previous grastrointestinal symptoms this needs special consideration&#46; Practical points about this case are&#58;<ul class="elsevierStyleList" id="l0005"><li class="elsevierStyleListItem" id="o0005"><span class="elsevierStyleLabel">1&#46;</span><p id="p0035" class="elsevierStylePara elsevierViewall">SGS can occur as complication of asymptomatic LPR&#46;</p></li><li class="elsevierStyleListItem" id="o0010"><span class="elsevierStyleLabel">2&#46;</span><p id="p0040" class="elsevierStylePara elsevierViewall">In patients with SGS and positive ANCA&#44; exclusion of LPR as cause or contributing factor is needed&#46;</p></li><li class="elsevierStyleListItem" id="o0015"><span class="elsevierStyleLabel">3&#46;</span><p id="p0045" class="elsevierStylePara elsevierViewall">A combined ANCA test using IIF and ELISA is mandatory when an ANCA-associated vasculitides is suspected&#46; Results need to be weighted against the pretest probability of having a true ANCA-associated disease&#46;</p></li></ul></p></span></span>"
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        "resumen" => "<p id="sp0015" class="elsevierStyleSimplePara elsevierViewall">We present a case of subglottic stenosis &#40;SGS&#41; in a young patient with positive ANCA but a wrong diagnosis of Wegener granulomatosis &#40;WG&#41;&#46; Instead&#44; she was diagnosed as having laryngopharyngeal reflux &#40;LPR&#41;&#46;</p><p id="sp0020" class="elsevierStyleSimplePara elsevierViewall">Pitfalls of ANCA testing in this scenario&#44; the route to diagnosis of LPR and the contribution of this entity to subglottic stenosis &#40;SGS&#41; in WG are discussed&#46; Caution when interpreting ANCA results is mandatory to avoid improper management&#46;</p>"
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Casos clínicos
Laryngopharyngeal Reflux Mimicking Limited Wegener Granulomatosis
Reflujo laringofaríngeo como simulador de granulomatosis de Wegener limitada
Luis Felipe Flores-Suáreza,
Autor para correspondencia
felipe98@prodigy.net.mx

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.
, Jaqueline Ramírez-Anguianob, Patricio Santillán-Dohertyc
a Servicio de Inmunología y Reumatología, Instituto Nacional de Ciencias Médicas y Nutrición Salvador Zubirán (INCMNSZ), Mexico City. Mexico
b Servicio de Otorrinolanringología, Instituto Nacional de Ciencias Médicas y Nutrición Salvador Zubirán (INCMNSZ), Mexico City. Mexico
c Servicio de Cirugía Experimental, Instituto Nacional de Ciencias Médicas y Nutrición Salvador Zubirán (INCMNSZ), México DF. México
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    "titulo" => "Laryngopharyngeal Reflux Mimicking Limited Wegener Granulomatosis"
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          "en" => "<p id="sp0005" class="elsevierStyleSimplePara elsevierViewall">Nasofibrolaryngoscopic findings during an acute dyspnea episode&#46; The Figure shows arytenoid oedema and hyperemia&#44; moderate oedema of the posterior commisure&#44; severe sulcus subglotticus and ventricular obliteration&#44; with a 70&#37; luminal narrowing&#46;</p>"
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    "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 &#40;SGS&#41; in Wegener granulomatosis &#40;WG&#41; occurs in 10&#37;-15&#37; of patients&#46;<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&#46; However&#44; misinterpretation when other more common causes of SGS are present can occur&#46; SGS with positive ANCA testing needs exclusion of LPR as cause or contributing factor&#46;</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&#46; She developed dysphonia&#44; chronic cough&#44; stridor&#44; and frequent dyspnea episodes&#46; ANCA were ordered and positive at a 1&#58;20 dilution by indirect immunofluorescence &#40;IIF&#41; with a cytoplasmic pattern&#46; She was referred to a rheumatologist who started deflazacort &#40;in decreasing doses for one year&#41; and trimethoprim&#47;sulfamethoxazole &#40;TMP-SMZ&#41;&#46; Each time steroids were decreased&#44; symptoms returned&#46; In one exacerbation&#44; she had hoarseness and 88&#37; oxygen saturation&#46; Other organ involvement was absent&#46; She was referred to us in January 2003&#46; At that time&#44; all paraclinical studies were normal or negative&#46; A nasofibrolaryngoscopy showed severe inflammatory changes &#40;<a class="elsevierStyleCrossRef" href="#f0005">Figure 1</a>&#41;&#46; Evaluation of the distal airway by direct laryngoscopy and flexible bronchoscopy showed no additional pathology&#46; A subglottic tissue biopsy demonstrated nonspecific acute and chronic inflammation with no granulomas or vasculitis&#46; ANCA testing with IIF and ELISA against myeloperoxidase and proteinase-3 was twice negative&#46; An oesophageal manometry showed incompetent lower oesophagic sphincter and 100&#37; relaxation&#46; The 24-hour oesophageal pH confirmed atypical extraoesophageal reflux disease&#46; She had 83 episodes of proximal reflux&#44; 4 of them over 15 minutes&#44; and pH &#60;<span class="elsevierStyleHsp" style=""></span>4&#46;0 for 83 total minutes&#46; The DeMeester score was 23&#46;6 &#40;normal &#60;<span class="elsevierStyleHsp" style=""></span>14&#46;7&#41;&#46; During the study she did not record gastrooesophageal symptoms&#46; She was given omeprazole 40<span class="elsevierStyleHsp" style=""></span>mg bid and domperidone 10<span class="elsevierStyleHsp" style=""></span>mg qid&#46; A new nasofibrolaryngoscopy showed improvement &#40;<a class="elsevierStyleCrossRef" href="#f0010">Figure 2</a>&#41;&#46; Under treatment for gastrooesophagic reflux disease &#40;GERD&#41; a new study showed absent proximal reflux episodes of &#62;<span class="elsevierStyleHsp" style=""></span>3 minutes&#44; total time of reflux decreasing to 33 minutes&#46; The DeMeester score was 11&#46; She is currently asymptomatic and no new episodes of dyspnea or sinusitis have occurred for more than 6 years&#46;</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&#46;<a class="elsevierStyleCrossRef" href="#bb0010"><span class="elsevierStyleSup">2</span></a> When severe and persistent it can lead to LPR&#44; which in consequence can cause &#8220;idiopathic&#8221; SGS&#46;<a class="elsevierStyleCrossRef" href="#bb0015"><span class="elsevierStyleSup">3</span></a> However&#44; symptoms are not recalled by a subtantial percentage of adult patients&#46;<a class="elsevierStyleCrossRef" href="#bb0020"><span class="elsevierStyleSup">4</span></a> To accurately diagnose LPR a modified four-port pH measurement is advised&#46;<a class="elsevierStyleCrossRef" href="#bb0025"><span class="elsevierStyleSup">5</span></a> In 1 study&#44; LPR may have contributed to persistent disease in patients with SGS due to WG&#46;<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&#46;</p><p id="p0020" class="elsevierStylePara elsevierViewall">A panel of experts has recommended that ANCA be performed in cases of unexplained SGS&#46;<a class="elsevierStyleCrossRef" href="#bb0030"><span class="elsevierStyleSup">6</span></a> However&#44; although upper airway involvement can be present in 70&#37; as part of the initial manifestations of WG&#44;<a class="elsevierStyleCrossRef" href="#bb0035"><span class="elsevierStyleSup">7</span></a> a false positive ANCA result can lead to pitfalls as observed&#46; In a study performed in a general hospital in the UK&#44; 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 &#40;other than Rheumatology&#41; yielded a low positive predicted value&#44; making them a tool of poor return when not properly indicated&#46; As in many of their cases&#44; the IIF ANCA was a false positive&#46; Their retrieval increased when both IIF and ELISA were performed&#46; Our case clearly underlines the need to perform both methods to detect ANCA &#40;IIF and ELISA&#41; as proposed by the results of the EC&#47;BCR study&#46;<a class="elsevierStyleCrossRef" href="#bb0045"><span class="elsevierStyleSup">9</span></a> Also&#44; that in doubtful cases of WG with borderline ANCA testing&#44; histological confirmation of the suspected vasculitides is necessary when the procedure is feasible&#46;</p><p id="p0025" class="elsevierStylePara elsevierViewall">In young or adult patients presenting with a clinical picture compatible with SGS&#44; LPR must be sought even in absence of GERD symptoms&#46; In such cases&#44; a single&#44; isolated positive ANCA test result&#44; especially if done with only one method cannot be taken as proof of WG&#46; This can lead to serious consequences due to the morbidity associated with treatment&#46;<a class="elsevierStyleCrossRef" href="#bb0050"><span class="elsevierStyleSup">10</span></a> Moreover&#44; in cases of WG&#44; GERD can still be present and contribute to the symptoms resembling SGS&#46;<a class="elsevierStyleCrossRef" href="#bb0015"><span class="elsevierStyleSup">3</span></a> The case provides an important lesson regarding SGS with ANCA positive testing&#44; in where awareness of other causes of SGS is needed&#46; The atypical presentation of the laryngopharyngeal disease&#44; plus the improvement seen with previous steroid and TMP-SMZ treatment during acute dyspnea events further contributed to the impression of WG&#46; Reconsideration of the diagnosis and the mechanism that caused the laryngeal disease led us to term this case as one of WG going backwards&#46;</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&#46; As there are individuals who do not recall a history of previous grastrointestinal symptoms this needs special consideration&#46; Practical points about this case are&#58;<ul class="elsevierStyleList" id="l0005"><li class="elsevierStyleListItem" id="o0005"><span class="elsevierStyleLabel">1&#46;</span><p id="p0035" class="elsevierStylePara elsevierViewall">SGS can occur as complication of asymptomatic LPR&#46;</p></li><li class="elsevierStyleListItem" id="o0010"><span class="elsevierStyleLabel">2&#46;</span><p id="p0040" class="elsevierStylePara elsevierViewall">In patients with SGS and positive ANCA&#44; exclusion of LPR as cause or contributing factor is needed&#46;</p></li><li class="elsevierStyleListItem" id="o0015"><span class="elsevierStyleLabel">3&#46;</span><p id="p0045" class="elsevierStylePara elsevierViewall">A combined ANCA test using IIF and ELISA is mandatory when an ANCA-associated vasculitides is suspected&#46; Results need to be weighted against the pretest probability of having a true ANCA-associated disease&#46;</p></li></ul></p></span></span>"
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        "resumen" => "<p id="sp0015" class="elsevierStyleSimplePara elsevierViewall">We present a case of subglottic stenosis &#40;SGS&#41; in a young patient with positive ANCA but a wrong diagnosis of Wegener granulomatosis &#40;WG&#41;&#46; Instead&#44; she was diagnosed as having laryngopharyngeal reflux &#40;LPR&#41;&#46;</p><p id="sp0020" class="elsevierStyleSimplePara elsevierViewall">Pitfalls of ANCA testing in this scenario&#44; the route to diagnosis of LPR and the contribution of this entity to subglottic stenosis &#40;SGS&#41; in WG are discussed&#46; Caution when interpreting ANCA results is mandatory to avoid improper management&#46;</p>"
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        "resumen" => "<p id="sp0025" class="elsevierStyleSimplePara elsevierViewall">Presentamos un caso de estenosis subgl&#243;tica &#40;ESG&#41; en una paciente joven con ANCA positivos&#44; pero un diagn&#243;stico err&#243;neo de granulomatosis de Wegener &#40;GW&#41;&#46; El caso correspondi&#243; a reflujo laringofar&#237;ngeo &#40;RLF&#41;&#46; Se discuten los falsos positivos de la prueba de ANCA&#44; la ruta diagn&#243;stica del RLF y la contribuci&#243;n de esta entidad a la ESG en GW&#46; Debe tenerse precauci&#243;n en la interpretaci&#243;n de ANCA para evitar el tratamiento inapropiado&#46;</p>"
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Información del artículo
ISSN: 1699258X
Idioma original: Inglés
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2010 Enero 1 0 1
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