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The patient ultimately developed squamous cell carcinoma of the left upper lobe &#40;LUL&#41; bronchus&#46; To the best of our knowledge&#44; this is the first description in the literature of a patient with both lesions&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">This was a 51-year-old man&#44; smoker of 60 pack-years&#44; with a clinical history of chronic bronchitis&#44; obesity&#44; symptomatic hyperuricemia treated with allopurinol 300<span class="elsevierStyleHsp" style=""></span>mg&#47;day &#40;although compliance was irregular&#41;&#44; arterial hypertension&#44; and metabolic syndrome&#46; One year previously&#44; he had presented in the emergency department on repeated occasions with episodes of dyspnea&#44; even at rest&#44; attributed to COPD exacerbations&#44; treated with bronchodilators and corticosteroids and discharged home with symptomatic treatment&#46; Four months later&#44; he returned to the emergency department with a severe attack of dyspnea&#46; Examination showed increased work of breathing&#44; central and peripheral cyanosis&#44; stridor&#44; and the following arterial blood gases&#58; PaO<span class="elsevierStyleInf">2</span> 55<span class="elsevierStyleHsp" style=""></span>mmHg&#44; PaCO<span class="elsevierStyleInf">2</span> 60<span class="elsevierStyleHsp" style=""></span>mmHg&#44; pH 7&#44; 20 and HCO<span class="elsevierStyleInf">3</span> 22<span class="elsevierStyleHsp" style=""></span>mmol&#46; Chest X-ray revealed mild cardiomegaly and no other findings&#46; An examination of the skin showed multiple giant tophi on the elbows&#44; knees and hands&#44; with deformed joints&#44; on the eyelids&#44; and on the abdominal wall&#44; legs&#44; and arms &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A and B&#41;&#46; He was assessed by the pulmonologist in the emergency department&#44; and urgent evaluation by the ENT specialist was requested&#44; in view of suspected upper airway stenosis&#46; Fiberoptic laryngoscopy showed paralysis of the vocal cords &#40;VC&#41; in adduction&#44; and whitish masses consistent with tophi on the VC and arytenoids &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>C&#41;&#44; requiring urgent tracheostomy&#46; Fiberoptic bronchoscopy showed whitish&#44; excretory lesions in the LMB&#44; similar to those observed in the larynx &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>D&#41;&#46; Bronchial biopsies revealed fibrinous bloody material with isolated atypical epithelial cellularity and fragments of bronchial wall with no significant morphological changes&#46; Laryngeal biopsies showed mucosa with squamous epithelium&#44; with no changes and no evidence of malignancy&#46; Of note in the clinical laboratory tests was uric acid 12<span class="elsevierStyleHsp" style=""></span>mg&#47;dl&#46; After oxygen therapy&#44; bronchodilators&#44; and antibiotics&#44; the patient was discharged with a permanent tracheostomy&#46; During his hospital stay&#44; a respiratory polygraphy with open tracheostomy was performed&#44; showing episodes of severe hypoventilation&#44; and an AHI of 8&#46;5&#47;h&#46; Treatment began with invasive mechanical ventilation via the tracheostomy in the hospital&#44; followed by night time ventilation at home &#40;VIVO 50 VA&#47;C&#58; Vt 1000<span class="elsevierStyleHsp" style=""></span>ml&#44; Fr 12<span class="elsevierStyleHsp" style=""></span>bpm&#44; EPAP 6<span class="elsevierStyleHsp" style=""></span>cmH<span class="elsevierStyleInf">2</span>O&#44; Ti 1&#46;5<span class="elsevierStyleHsp" style=""></span>s&#44; inspiratory trigger 3&#44; and descending ramp&#41;&#44; with good compliance&#46; One year later&#44; the patient was admitted for hemoptysis&#44; with soft tissue density visualized on CT around the LMB&#44; origin of the intermediate bronchus and the LUL bronchus&#46; In addition to the LMB lesions&#44; fiberoptic bronchoscopy showed almost complete stenosis of the lingular bronchus and partial stenosis of the culmen with necrotic mucosa&#46; Biopsies revealed well-differentiated squamous cell carcinoma&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Head and neck tissue involvement in CTG is exceptional&#44; and VC paralysis due to acute gouty arthritis is even rarer&#46; Moreover&#44; bronchial involvement has only been described in 1 patient&#44;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">3</span></a> and there are no more than 20 published cases of laryngeal involvement&#44; none of which appear in the Spanish literature&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">4</span></a> Although very few studies have reported laryngeal involvement in gout&#44; which is more commonly observed in association with the cricothyroid joint&#44; Garrod&#44; in 1863&#44; described a few &#8220;specks&#8221; of sodium urate in the arytenoid cartilage in the autopsy of a man with CTG&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">5</span></a> Years later&#44; Virchow reported the presence of monosodium urate in the VC of a patient with extensive tophaceous deposits&#46;<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">6</span></a> VC paralysis is a potential cause ARF&#44; and exceptionally occurs with acute arthritis&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">2</span></a> As in our case&#44; VC paralysis is caused by atrophy and denervation of some of the laryngeal muscles of the cricoarytenoid joint&#44; rather than the direct deposit of tophi&#44; causing fibrosis and inflammation on the perineuronal tissue&#44; that ultimately leads to atrophy of the innervated muscle fibers and hypertrophy of the healthy tissue&#46;<a class="elsevierStyleCrossRefs" href="#bib0075"><span class="elsevierStyleSup">7&#44;8</span></a> Our patient also presented endobronchial tophi&#44; although he ultimately developed squamous cell carcinoma&#46; Of course&#44; no cause&#8211;effect relationship between gout and the carcinoma caused by smoking can be established&#46; The diagnosis of cricoarytenoid gout can be established by signs and symptoms observed in patients with extensive CTG&#44; such as dysphagia&#44; dysphonia&#44; dyspnea&#44; etc&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">2</span></a> Gouty laryngeal involvement should be considered in a case of history of hyperuricemia and acute or chronic gouty arthritis&#44; accompanied by snoring&#44; odynophagia&#44; dysphagia&#44; stridor&#44; dyspnea&#44; and&#47;or dysphonia&#46; Sometimes the upper airway is acutely compromised&#44; requiring urgent tracheostomy&#44; and bronchial lesions can also develop&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">3</span></a></p></span>"
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Scientific Letter
Acute Respiratory Failure Due to Chronic Tophaceous Gout With Laryngeal and Bronchial Involvement: An Unusual Complication
Insuficiencia respiratoria aguda secundaria a gota tofácea crónica con afectación laríngea y bronquial: una complicación excepcional
Mar Arlandis, Virginia Molina, Sandra Vañes, Eusebi Chiner
Corresponding author
echinervives@gmail.com

Corresponding author.
Servicio de Neumología, Hospital Universitari Sant Joan d’Alacant, Alicante, Spain
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    "titulo" => "Acute Respiratory Failure Due to Chronic Tophaceous Gout With Laryngeal and Bronchial Involvement&#58; An Unusual Complication"
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          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">&#40;A and B&#41; Gouty tophi in the elbow joint and abdominal wall&#46; &#40;C&#41; Bronchoscopy&#58; vocal cord paralysis in adduction with tophaceous deposit on the arytenoids&#46; &#40;D&#41; Bronchoscopy&#58; submucosal tophaceous gout deposits in the left main bronchus&#46;</p>"
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Sustained hyperuricemia &#40;&#62;7<span class="elsevierStyleHsp" style=""></span>mg&#47;dl&#41;&#44; when it manifests as chronic tophaceous gout &#40;CTG&#41;&#44; can lead to the formation of granulomas &#40;tophi&#41; around the urate crystals&#44; which have a high capacity for erosion&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">1</span></a> Laryngeal involvement in CTG is rare&#44; and can cause upper airway obstruction and acute respiratory failure &#40;ARF&#41;&#44;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">2</span></a> and can affect the tracheobronchial tree&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">3</span></a> We report the case of a patient with CTG who developed ARF after an acute episode of laryngeal gout requiring tracheostomy&#44; and who also presented tophi in the left main bronchus &#40;LMB&#41;&#46; The patient ultimately developed squamous cell carcinoma of the left upper lobe &#40;LUL&#41; bronchus&#46; To the best of our knowledge&#44; this is the first description in the literature of a patient with both lesions&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">This was a 51-year-old man&#44; smoker of 60 pack-years&#44; with a clinical history of chronic bronchitis&#44; obesity&#44; symptomatic hyperuricemia treated with allopurinol 300<span class="elsevierStyleHsp" style=""></span>mg&#47;day &#40;although compliance was irregular&#41;&#44; arterial hypertension&#44; and metabolic syndrome&#46; One year previously&#44; he had presented in the emergency department on repeated occasions with episodes of dyspnea&#44; even at rest&#44; attributed to COPD exacerbations&#44; treated with bronchodilators and corticosteroids and discharged home with symptomatic treatment&#46; Four months later&#44; he returned to the emergency department with a severe attack of dyspnea&#46; Examination showed increased work of breathing&#44; central and peripheral cyanosis&#44; stridor&#44; and the following arterial blood gases&#58; PaO<span class="elsevierStyleInf">2</span> 55<span class="elsevierStyleHsp" style=""></span>mmHg&#44; PaCO<span class="elsevierStyleInf">2</span> 60<span class="elsevierStyleHsp" style=""></span>mmHg&#44; pH 7&#44; 20 and HCO<span class="elsevierStyleInf">3</span> 22<span class="elsevierStyleHsp" style=""></span>mmol&#46; Chest X-ray revealed mild cardiomegaly and no other findings&#46; An examination of the skin showed multiple giant tophi on the elbows&#44; knees and hands&#44; with deformed joints&#44; on the eyelids&#44; and on the abdominal wall&#44; legs&#44; and arms &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A and B&#41;&#46; He was assessed by the pulmonologist in the emergency department&#44; and urgent evaluation by the ENT specialist was requested&#44; in view of suspected upper airway stenosis&#46; Fiberoptic laryngoscopy showed paralysis of the vocal cords &#40;VC&#41; in adduction&#44; and whitish masses consistent with tophi on the VC and arytenoids &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>C&#41;&#44; requiring urgent tracheostomy&#46; Fiberoptic bronchoscopy showed whitish&#44; excretory lesions in the LMB&#44; similar to those observed in the larynx &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>D&#41;&#46; Bronchial biopsies revealed fibrinous bloody material with isolated atypical epithelial cellularity and fragments of bronchial wall with no significant morphological changes&#46; Laryngeal biopsies showed mucosa with squamous epithelium&#44; with no changes and no evidence of malignancy&#46; Of note in the clinical laboratory tests was uric acid 12<span class="elsevierStyleHsp" style=""></span>mg&#47;dl&#46; After oxygen therapy&#44; bronchodilators&#44; and antibiotics&#44; the patient was discharged with a permanent tracheostomy&#46; During his hospital stay&#44; a respiratory polygraphy with open tracheostomy was performed&#44; showing episodes of severe hypoventilation&#44; and an AHI of 8&#46;5&#47;h&#46; Treatment began with invasive mechanical ventilation via the tracheostomy in the hospital&#44; followed by night time ventilation at home &#40;VIVO 50 VA&#47;C&#58; Vt 1000<span class="elsevierStyleHsp" style=""></span>ml&#44; Fr 12<span class="elsevierStyleHsp" style=""></span>bpm&#44; EPAP 6<span class="elsevierStyleHsp" style=""></span>cmH<span class="elsevierStyleInf">2</span>O&#44; Ti 1&#46;5<span class="elsevierStyleHsp" style=""></span>s&#44; inspiratory trigger 3&#44; and descending ramp&#41;&#44; with good compliance&#46; One year later&#44; the patient was admitted for hemoptysis&#44; with soft tissue density visualized on CT around the LMB&#44; origin of the intermediate bronchus and the LUL bronchus&#46; In addition to the LMB lesions&#44; fiberoptic bronchoscopy showed almost complete stenosis of the lingular bronchus and partial stenosis of the culmen with necrotic mucosa&#46; Biopsies revealed well-differentiated squamous cell carcinoma&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Head and neck tissue involvement in CTG is exceptional&#44; and VC paralysis due to acute gouty arthritis is even rarer&#46; Moreover&#44; bronchial involvement has only been described in 1 patient&#44;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">3</span></a> and there are no more than 20 published cases of laryngeal involvement&#44; none of which appear in the Spanish literature&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">4</span></a> Although very few studies have reported laryngeal involvement in gout&#44; which is more commonly observed in association with the cricothyroid joint&#44; Garrod&#44; in 1863&#44; described a few &#8220;specks&#8221; of sodium urate in the arytenoid cartilage in the autopsy of a man with CTG&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">5</span></a> Years later&#44; Virchow reported the presence of monosodium urate in the VC of a patient with extensive tophaceous deposits&#46;<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">6</span></a> VC paralysis is a potential cause ARF&#44; and exceptionally occurs with acute arthritis&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">2</span></a> As in our case&#44; VC paralysis is caused by atrophy and denervation of some of the laryngeal muscles of the cricoarytenoid joint&#44; rather than the direct deposit of tophi&#44; causing fibrosis and inflammation on the perineuronal tissue&#44; that ultimately leads to atrophy of the innervated muscle fibers and hypertrophy of the healthy tissue&#46;<a class="elsevierStyleCrossRefs" href="#bib0075"><span class="elsevierStyleSup">7&#44;8</span></a> Our patient also presented endobronchial tophi&#44; although he ultimately developed squamous cell carcinoma&#46; Of course&#44; no cause&#8211;effect relationship between gout and the carcinoma caused by smoking can be established&#46; The diagnosis of cricoarytenoid gout can be established by signs and symptoms observed in patients with extensive CTG&#44; such as dysphagia&#44; dysphonia&#44; dyspnea&#44; etc&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">2</span></a> Gouty laryngeal involvement should be considered in a case of history of hyperuricemia and acute or chronic gouty arthritis&#44; accompanied by snoring&#44; odynophagia&#44; dysphagia&#44; stridor&#44; dyspnea&#44; and&#47;or dysphonia&#46; Sometimes the upper airway is acutely compromised&#44; requiring urgent tracheostomy&#44; and bronchial lesions can also develop&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">3</span></a></p></span>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Arlandis M&#44; Molina V&#44; Va&#241;es S&#44; Chiner E&#46; Insuficiencia respiratoria aguda secundaria a gota tof&#225;cea cr&#243;nica con afectaci&#243;n lar&#237;ngea y bronquial&#58; una complicaci&#243;n excepcional&#46; Arch Bronconeumol&#46; 2018&#59;54&#58;399&#8211;400&#46;</p>"
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Article information
ISSN: 15792129
Original language: English
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