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FEV1&#47;FVC 87&#37;&#59; DLCO 71&#37;&#59; KCO 101&#37;&#41;&#46; Given the suspicion of infectious disease in an immunosuppressed patient&#44; azithromycin was added to the treatment plan&#44; and a computed axial tomography &#40;CT&#41; scan was performed&#44; which revealed discreet tracheal wall thickening&#44; with no evidence of pulmonary consolidations or other type of involvement &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; The bronchoscopy was significant for raised whitish lesions in the tracheal wall and at the entrance of the two main bronchi&#44; predominantly in the posterior tracheal wall &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; An opportunistic infection was suspected&#44; so bronchoalveolar lavage &#40;BAL&#41; was performed and samples were sent to the microbiology lab&#58; standard pathogen panel &#40;bacteria&#44; mycobacteria&#44; fungi&#44; viruses&#44; etc&#46;&#41; was negative&#46; The pathology study of the tracheal wall biopsy reported nonspecific inflammatory signs with areas of ulceration&#46; BAL cellularity consisted mainly of macrophages &#40;80&#37;&#41;&#44; lymphocytes &#40;15&#37;&#41; and polymorphonuclear cells &#40;5&#37;&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">A diagnosis of tracheobronchitis due to Crohn&#8217;s disease was given&#46; Treatment started with inhaled corticosteroids &#40;fluticasone 1&#44;000&#8239;mg every 12&#8239;h&#41;&#46; After 1 month of treatment&#44; the patient&#39;s symptoms had disappeared&#44; so dosing was reduced to 500&#8239;mg every 12&#8239;h&#46; A chest CT scan was repeated&#44; in which chest wall thickening was no longer visualized&#46; Bronchoscopy after treatment confirmed the resolution of the earlier lesions&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">Extraintestinal manifestations may occur in 6&#37;&#8211;47&#37; of patients with inflammatory bowel disease&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Pulmonary involvement is an infrequent event&#44; although underdiagnosis is common in many cases&#44; and it has been suggested that it might occur in up to 60&#37; of patients with inflammatory bowel disease<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a>&#59; the most characteristic manifestation is ulcerative colitis&#46; The entire respiratory system may be involved&#44; from the upper airway to the pulmonary parenchyma&#46; Bronchiectasis is the manifestation most often described in the literature&#58; some series report this type of involvement in up to 66&#37; of cases&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#8211;3</span></a> Tracheobronchitis in Crohn&#39;s disease is very rare and often accompanied by inflammatory bowel disease in remission&#46;<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#44;5</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">These manifestations tend to occur in the fifth decade of life and appear after diagnosis of inflammatory bowel disease in more than 85&#37; of cases&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#8211;5</span></a> The clinical characteristics of this manifestation do not differ much from those of many other respiratory diseases&#46; It usually occurs with cough&#44; fever&#44; and an increase in usual dyspnea&#46; It is not associated with any specific functional alteration&#44; but a low diffusing capacity &#40;as in our patient&#41; or even positivity in a methacholine challenge test may be observed&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;3&#8211;6</span></a> The images obtained on chest X-ray are not usually pathological&#44; but tracheal wall thickening may be seen on chest CT&#46; Fiberoptic bronchoscopy often reveals diffuse edema of the tracheal mucosa and main bronchi&#44; normally concurrently with whitish granular lesions with no clear distribution&#46; In Crohn&#8217;s disease&#44; biopsies tend to show non-specific chronic inflammatory infiltration with a predominance of neutrophils&#44;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> while lymphocytosis is more predominant in other types of pulmonary manifestations of inflammatory bowel diseases&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#8211;3</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Evidence with respect to treatment is scant&#44; and only clinical case series have been published&#46; In most patients&#44; this manifestation resolves with the administration of inhaled corticosteroids&#44; which moreover help to prevent permanent lung damage&#46;<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">8&#44;9</span></a> However&#44; the administration of systemic corticosteroids or even immunosuppressants has been necessary in some series&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> Treatment with glucocorticoids is unsuccessful in approximately one third of patients with pulmonary manifestations&#44; and immunosuppressive therapy may be required&#46; A good option for immunosuppression may be azathioprine&#58; Kar &#38; Thomas reported good results<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> with that approach&#44; although their patient&#44; unlike ours&#44; presented ulcerative colitis&#46; Infliximab has also been shown to greatly improve symptoms of patients with inflammatory bowel disease&#44; and plays a significant role in the control of both gastrointestinal and pulmonary manifestations&#44; as reported by Hayek&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> Treatment duration has not been established and varies greatly&#58; from 2 weeks in the shortest schedules&#44; up to 3 months in the longest&#46; In our case&#44; treatment continued at full dose for 1 month&#44; and was then tapered over the following 2 months until discontinuation&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">The mechanism of how inflammatory bowel disease can lead to pulmonary manifestations is still unknown&#46; In embryonic development&#44; the formation of the gastrointestinal tract and respiratory system originate in the same part of the embryonic structure and have a similar epithelial structure&#44; so this might explain why the lung may become involved in this entity&#46; However&#44; many other alternative mechanisms have been proposed&#44; such as bacterial dysbiosis&#44; environmental pollution&#44; or even genetic factors&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> For this reason&#44; when performing a differential diagnosis in any patient with inflammatory bowel disease&#44; the presence of manifestations of their underlying disease should be taken into account&#44; especially in patients with cough and fever and no obvious infection&#46; Although the incidence of these manifestations is still believed to be low&#44; underdiagnosis seems likely&#44; given the similarity of diagnostic test results and the fact that many patients who respond well to inhaled corticosteroids may be classified as asthmatics&#46;<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6&#44;7</span></a> We must&#44; therefore&#44; take into account this diagnostic possibility when we encounter patients with these characteristics&#46;</p></span>"
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Scientific Letter
Tracheobronchial involvement in Crohn's disease
Afectación traqueobronquial por enfermedad de Crohn
Irene Lojo-Rodríguez, Virginia Leiro-Fernández
Corresponding author
, Cecilia Mouronte-Roibás, Maribel Botana-Rial, Alberto Fernández-Villar
Servicio de Neumología, Hospital Álvaro Cunqueiro, Complexo Hospitalario Univeristario de Vigo, Grupo de Investigación NeumovigoI+i. IIS Galicia Sur, Vigo, Pontevedra, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">We report the case of a 53-year-old man&#44; non-smoker&#44; monitored for Crohn&#8217;s disease of the ileum by the digestive diseases department from the age of 14 years&#46; Ileocecal resection was performed more than 20 years previously&#46; He has been receiving mercaptopurine for 12 years and infliximab for 6 years&#44; and has had no flare-ups in the last 10 years&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">He was referred to our clinic with a 1-month history of non-productive cough and fever&#46; During the course of these symptoms&#44; he received 2 antibiotic regimens&#44; first with levofloxacin and then with amoxicillin-clavulanate&#46; The chest X-ray showed no significant changes and pulmonary function tests were significant only for slightly altered diffusion results &#40;FEV1 3&#44;100 &#91;91&#37;&#93;&#59; FVC 3&#44;550 &#91;81&#37;&#93;&#59; FEV1&#47;FVC 87&#37;&#59; DLCO 71&#37;&#59; KCO 101&#37;&#41;&#46; Given the suspicion of infectious disease in an immunosuppressed patient&#44; azithromycin was added to the treatment plan&#44; and a computed axial tomography &#40;CT&#41; scan was performed&#44; which revealed discreet tracheal wall thickening&#44; with no evidence of pulmonary consolidations or other type of involvement &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; The bronchoscopy was significant for raised whitish lesions in the tracheal wall and at the entrance of the two main bronchi&#44; predominantly in the posterior tracheal wall &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; An opportunistic infection was suspected&#44; so bronchoalveolar lavage &#40;BAL&#41; was performed and samples were sent to the microbiology lab&#58; standard pathogen panel &#40;bacteria&#44; mycobacteria&#44; fungi&#44; viruses&#44; etc&#46;&#41; was negative&#46; The pathology study of the tracheal wall biopsy reported nonspecific inflammatory signs with areas of ulceration&#46; BAL cellularity consisted mainly of macrophages &#40;80&#37;&#41;&#44; lymphocytes &#40;15&#37;&#41; and polymorphonuclear cells &#40;5&#37;&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">A diagnosis of tracheobronchitis due to Crohn&#8217;s disease was given&#46; Treatment started with inhaled corticosteroids &#40;fluticasone 1&#44;000&#8239;mg every 12&#8239;h&#41;&#46; After 1 month of treatment&#44; the patient&#39;s symptoms had disappeared&#44; so dosing was reduced to 500&#8239;mg every 12&#8239;h&#46; A chest CT scan was repeated&#44; in which chest wall thickening was no longer visualized&#46; Bronchoscopy after treatment confirmed the resolution of the earlier lesions&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">Extraintestinal manifestations may occur in 6&#37;&#8211;47&#37; of patients with inflammatory bowel disease&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Pulmonary involvement is an infrequent event&#44; although underdiagnosis is common in many cases&#44; and it has been suggested that it might occur in up to 60&#37; of patients with inflammatory bowel disease<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a>&#59; the most characteristic manifestation is ulcerative colitis&#46; The entire respiratory system may be involved&#44; from the upper airway to the pulmonary parenchyma&#46; Bronchiectasis is the manifestation most often described in the literature&#58; some series report this type of involvement in up to 66&#37; of cases&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#8211;3</span></a> Tracheobronchitis in Crohn&#39;s disease is very rare and often accompanied by inflammatory bowel disease in remission&#46;<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#44;5</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">These manifestations tend to occur in the fifth decade of life and appear after diagnosis of inflammatory bowel disease in more than 85&#37; of cases&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#8211;5</span></a> The clinical characteristics of this manifestation do not differ much from those of many other respiratory diseases&#46; It usually occurs with cough&#44; fever&#44; and an increase in usual dyspnea&#46; It is not associated with any specific functional alteration&#44; but a low diffusing capacity &#40;as in our patient&#41; or even positivity in a methacholine challenge test may be observed&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;3&#8211;6</span></a> The images obtained on chest X-ray are not usually pathological&#44; but tracheal wall thickening may be seen on chest CT&#46; Fiberoptic bronchoscopy often reveals diffuse edema of the tracheal mucosa and main bronchi&#44; normally concurrently with whitish granular lesions with no clear distribution&#46; In Crohn&#8217;s disease&#44; biopsies tend to show non-specific chronic inflammatory infiltration with a predominance of neutrophils&#44;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> while lymphocytosis is more predominant in other types of pulmonary manifestations of inflammatory bowel diseases&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#8211;3</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Evidence with respect to treatment is scant&#44; and only clinical case series have been published&#46; In most patients&#44; this manifestation resolves with the administration of inhaled corticosteroids&#44; which moreover help to prevent permanent lung damage&#46;<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">8&#44;9</span></a> However&#44; the administration of systemic corticosteroids or even immunosuppressants has been necessary in some series&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> Treatment with glucocorticoids is unsuccessful in approximately one third of patients with pulmonary manifestations&#44; and immunosuppressive therapy may be required&#46; A good option for immunosuppression may be azathioprine&#58; Kar &#38; Thomas reported good results<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> with that approach&#44; although their patient&#44; unlike ours&#44; presented ulcerative colitis&#46; Infliximab has also been shown to greatly improve symptoms of patients with inflammatory bowel disease&#44; and plays a significant role in the control of both gastrointestinal and pulmonary manifestations&#44; as reported by Hayek&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> Treatment duration has not been established and varies greatly&#58; from 2 weeks in the shortest schedules&#44; up to 3 months in the longest&#46; In our case&#44; treatment continued at full dose for 1 month&#44; and was then tapered over the following 2 months until discontinuation&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">The mechanism of how inflammatory bowel disease can lead to pulmonary manifestations is still unknown&#46; In embryonic development&#44; the formation of the gastrointestinal tract and respiratory system originate in the same part of the embryonic structure and have a similar epithelial structure&#44; so this might explain why the lung may become involved in this entity&#46; However&#44; many other alternative mechanisms have been proposed&#44; such as bacterial dysbiosis&#44; environmental pollution&#44; or even genetic factors&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> For this reason&#44; when performing a differential diagnosis in any patient with inflammatory bowel disease&#44; the presence of manifestations of their underlying disease should be taken into account&#44; especially in patients with cough and fever and no obvious infection&#46; Although the incidence of these manifestations is still believed to be low&#44; underdiagnosis seems likely&#44; given the similarity of diagnostic test results and the fact that many patients who respond well to inhaled corticosteroids may be classified as asthmatics&#46;<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6&#44;7</span></a> We must&#44; therefore&#44; take into account this diagnostic possibility when we encounter patients with these characteristics&#46;</p></span>"
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Article information
ISSN: 15792129
Original language: English
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