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such as cough and dyspnea&#46; We report the case of a patient with CD without respiratory manifestations&#44; in whom pulmonary necrobiotic nodules were an incidental radiological finding&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 26-year-old woman consulted for diarrhea&#44; with a 2-year history of 6&#8211;10 stools per day of liquid consistency with blood and mucus&#44; abdominal pain&#44; and weight loss&#46; She was a smoker of 10 cigarettes a day and had no other clinical history of interest&#46; Physical examination revealed poor general condition and painful abdomen on palpation in the epigastrium&#46; Cardiopulmonary auscultation was normal&#44; and no adenopathies or skin lesions were observed&#46; Clinical laboratory tests were significant for hemoglobin 10&#46;5<span class="elsevierStyleHsp" style=""></span>g&#47;dl and transferrin saturation 5&#46;1&#37;&#44; platelets 393<span class="elsevierStyleHsp" style=""></span>000&#47;ml&#44; and eosinophils 1000&#47;ml&#46; 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1</a>&#41;&#46; Bronchoscopy was normal with no tumor cells or pathogens in bronchoalveolar lavage &#40;BAL&#41;&#46; Other diagnoses&#44; including metastasis and abscesses&#44; were considered in the differential diagnosis&#46; The diagnosis of pulmonary necrobiotic nodules associated with CD was given&#44; in view of the temporal relationship between the diagnosis and IBD flare-up&#44; and the good condition of the patient&#46; She was treated with systemic corticosteroids&#44; and radiological resolution of the nodules was achieved after 1 month of treatment &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">IBD is a chronic inflammation of unknown etiology&#44; which affects the digestive tract&#46; Pathogenesis is due to a recurrent inadequate response of the mucosal immune system&#44; activated by the presence of normal luminal flora in genetically predisposed individuals&#46; It is histologically characterized by a lymphocytic polymorphonuclear infiltrate with formation of granulomas&#44; ulcers&#44; and fissures in the mucosa&#46; Although it mainly affects the intestine&#44; extraintestinal manifestations are well-known&#44; with a prevalence of 21&#37;&#8211;41&#37; that increase as the disease progresses&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">Pulmonary involvement as an extraintestinal manifestation was first described by Kraft et al&#46; in 1976 after observing 6 patients with a diagnosis of IBD who developed chronic bronchial suppuration&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> Different pathogenic mechanisms for the pulmonary involvement in these patients have been described&#44; including the common embryological origin of the airway and the intestine&#44; a similar immune system&#44; and the presence of circulating immune complexes and autoantibodies&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> This is the most unusual extraintestinal manifestation of IBD and is usually seen in patients with UC&#44; distinguishing it from other extraintestinal manifestations&#46; Its real prevalence is unknown because it is sometimes asymptomatic or does not coincide chronologically with the IBD&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> All these factors make diagnosis difficult in the absence of a high suspicion&#46; However&#44; early identification is important to prevent it progressing to a more disabling condition and to avoid complications&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">The most common symptoms are derived from airway inflammation &#40;cough&#44; expectoration&#44; or dyspnea&#41;&#44; which manifests in many ways&#44; ranging from asymptomatic disease to involvement of the tracheobronchial tree &#40;bronchitis&#44; bronchiectasis or bronchiolitis&#41;&#44; the lung parenchyma&#44; and the pleura&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">A distinction must be drawn between pulmonary involvement caused by IBD and that caused by IBD treatment&#44; the latter being the most frequent&#46; IBD treatment that might cause pulmonary involvement includes the long-term use of sulfasalazine&#44; mesalazine&#44; methotrexate&#44; and anti-tumor necrosis factor &#40;anti-TNF&#41;&#44; rather than the underlying disease&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">Pulmonary necrobiotic nodules in IBD are an exceptional complication and are more frequent in UC&#46; This presentation was first described in patients with rheumatoid arthritis or pneumoconiosis &#40;Caplan&#39;s syndrome&#41;&#46; Histologically&#44; necrobiotic nodules are sterile aggregates of neutrophils&#44; which frequently cavitate&#46;<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Radiologically&#44; the differential diagnosis must consider pulmonary nodules of infectious origin &#40;tuberculosis&#44; fungi&#44; staphylococci&#41;&#44; autoimmune diseases&#44; cancer&#44; etc&#46; In our case&#44; the absence of fever&#44; absence of neoplastic cells in BAL&#44; and absence of eosinophils in BAL ruling out any association with mesalazine&#44; led us to start empirical treatment with corticosteroids&#44; with good response&#46; Based on these findings&#44; lung biopsy was avoided&#44; pending clinical response and response to the new treatment&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">Very few cases have been reported in the literature on this entity&#44; and all of them presented with respiratory symptoms&#44; including cough and dyspnea&#46;<a class="elsevierStyleCrossRefs" href="#bib0075"><span class="elsevierStyleSup">5&#8211;9</span></a> This makes our patient even more unique&#44; since she did not present respiratory manifestations&#44; and diagnosis was made from an incidental finding&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">Although the spontaneous resolution of necrobiotic nodules in IBD has been described&#44; of the 5 cases associated with CD&#44; only 1 resolved spontaneously&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a> The other patients were treated with oral prednisone&#44; with complete resolution&#46; Infliximab is currently being used for lung nodules that are refractory to systemic steroids&#46;<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a></p></span>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Larrey Ruiz L&#44; Sabater Abad C&#44; Pe&#241;o Mu&#241;oz L&#44; Huguet Malav&#233;s JM&#44; Juan Samper G&#46; N&#243;dulos necrobi&#243;ticos pulmonares&#58; una manifestaci&#243;n excepcional de la enfermedad de Crohn&#46; Arch Bronconeumol&#46; 2019&#59;55&#58;108&#8211;110&#46;</p>"
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Scientific Letter
Pulmonary Necrobiotic Nodules: A Rare Manifestation of Crohn's Disease
Nódulos necrobióticos pulmonares: una manifestación excepcional de la enfermedad de Crohn
Laura Larrey Ruiza, Cristina Sabater Abadb,
Corresponding author
sabaterabadcristina@gmail.com

Corresponding author.
, Laura Peño Muñoza, Jose María Huguet Malavésa, Gustavo Juan Samperb
a Servicio de Patología Digestiva, Consorcio Hospital General Universitario de Valencia, Valencia, Spain
b Servicio de Neumología, Consorcio Hospital General Universitario de Valencia, Valencia, Spain
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such as cough and dyspnea&#46; We report the case of a patient with CD without respiratory manifestations&#44; in whom pulmonary necrobiotic nodules were an incidental radiological finding&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 26-year-old woman consulted for diarrhea&#44; with a 2-year history of 6&#8211;10 stools per day of liquid consistency with blood and mucus&#44; abdominal pain&#44; and weight loss&#46; She was a smoker of 10 cigarettes a day and had no other clinical history of interest&#46; Physical examination revealed poor general condition and painful abdomen on palpation in the epigastrium&#46; Cardiopulmonary auscultation was normal&#44; and no adenopathies or skin lesions were observed&#46; Clinical laboratory tests were significant for hemoglobin 10&#46;5<span class="elsevierStyleHsp" style=""></span>g&#47;dl and transferrin saturation 5&#46;1&#37;&#44; platelets 393<span class="elsevierStyleHsp" style=""></span>000&#47;ml&#44; and eosinophils 1000&#47;ml&#46; Chest radiograph was normal&#46; Stool cultures at the time of the study were negative&#46; Ileocolonoscopy showed swollen and erythematous mucosa with crater-like&#44; serpiginous ulcers alternating with normal mucosa&#46; The pathology report was suggestive of CD&#46; Treatment began with oral budesonide 9<span class="elsevierStyleHsp" style=""></span>mg&#47;day and mesalazine 2<span class="elsevierStyleHsp" style=""></span>g&#47;day&#44; with clinical improvement&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">The patient&#39;s digestive symptoms subsequently improved&#44; but lobar pneumonia developed&#44; which was treated with levofloxacin&#46; However&#44; several lung nodules measuring 8&#8211;10<span class="elsevierStyleHsp" style=""></span>mm in diameter were identified in the X-ray performed to monitor radiological progress when the respiratory symptoms had resolved&#44; and confirmed on a chest CT scan &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; Bronchoscopy was normal with no tumor cells or pathogens in bronchoalveolar lavage &#40;BAL&#41;&#46; Other diagnoses&#44; including metastasis and abscesses&#44; were considered in the differential diagnosis&#46; The diagnosis of pulmonary necrobiotic nodules associated with CD was given&#44; in view of the temporal relationship between the diagnosis and IBD flare-up&#44; and the good condition of the patient&#46; She was treated with systemic corticosteroids&#44; and radiological resolution of the nodules was achieved after 1 month of treatment &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">IBD is a chronic inflammation of unknown etiology&#44; which affects the digestive tract&#46; Pathogenesis is due to a recurrent inadequate response of the mucosal immune system&#44; activated by the presence of normal luminal flora in genetically predisposed individuals&#46; It is histologically characterized by a lymphocytic polymorphonuclear infiltrate with formation of granulomas&#44; ulcers&#44; and fissures in the mucosa&#46; Although it mainly affects the intestine&#44; extraintestinal manifestations are well-known&#44; with a prevalence of 21&#37;&#8211;41&#37; that increase as the disease progresses&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">Pulmonary involvement as an extraintestinal manifestation was first described by Kraft et al&#46; in 1976 after observing 6 patients with a diagnosis of IBD who developed chronic bronchial suppuration&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> Different pathogenic mechanisms for the pulmonary involvement in these patients have been described&#44; including the common embryological origin of the airway and the intestine&#44; a similar immune system&#44; and the presence of circulating immune complexes and autoantibodies&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> This is the most unusual extraintestinal manifestation of IBD and is usually seen in patients with UC&#44; distinguishing it from other extraintestinal manifestations&#46; Its real prevalence is unknown because it is sometimes asymptomatic or does not coincide chronologically with the IBD&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> All these factors make diagnosis difficult in the absence of a high suspicion&#46; However&#44; early identification is important to prevent it progressing to a more disabling condition and to avoid complications&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">The most common symptoms are derived from airway inflammation &#40;cough&#44; expectoration&#44; or dyspnea&#41;&#44; which manifests in many ways&#44; ranging from asymptomatic disease to involvement of the tracheobronchial tree &#40;bronchitis&#44; bronchiectasis or bronchiolitis&#41;&#44; the lung parenchyma&#44; and the pleura&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">A distinction must be drawn between pulmonary involvement caused by IBD and that caused by IBD treatment&#44; the latter being the most frequent&#46; IBD treatment that might cause pulmonary involvement includes the long-term use of sulfasalazine&#44; mesalazine&#44; methotrexate&#44; and anti-tumor necrosis factor &#40;anti-TNF&#41;&#44; rather than the underlying disease&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">Pulmonary necrobiotic nodules in IBD are an exceptional complication and are more frequent in UC&#46; This presentation was first described in patients with rheumatoid arthritis or pneumoconiosis &#40;Caplan&#39;s syndrome&#41;&#46; Histologically&#44; necrobiotic nodules are sterile aggregates of neutrophils&#44; which frequently cavitate&#46;<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Radiologically&#44; the differential diagnosis must consider pulmonary nodules of infectious origin &#40;tuberculosis&#44; fungi&#44; staphylococci&#41;&#44; autoimmune diseases&#44; cancer&#44; etc&#46; In our case&#44; the absence of fever&#44; absence of neoplastic cells in BAL&#44; and absence of eosinophils in BAL ruling out any association with mesalazine&#44; led us to start empirical treatment with corticosteroids&#44; with good response&#46; Based on these findings&#44; lung biopsy was avoided&#44; pending clinical response and response to the new treatment&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">Very few cases have been reported in the literature on this entity&#44; and all of them presented with respiratory symptoms&#44; including cough and dyspnea&#46;<a class="elsevierStyleCrossRefs" href="#bib0075"><span class="elsevierStyleSup">5&#8211;9</span></a> This makes our patient even more unique&#44; since she did not present respiratory manifestations&#44; and diagnosis was made from an incidental finding&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">Although the spontaneous resolution of necrobiotic nodules in IBD has been described&#44; 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ISSN: 15792129
Original language: English
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