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&#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; An electromyographic study showed a pattern indicating a demyelinating disease&#46; Bronchoalveolar lavage demonstrated nonspecific chronic and acute inflammation&#46; Studies focusing on autoimmunity found the patient to be positive for antinuclear antibodies at a titer of 1&#58;320 with a mitochondrial pattern&#44; as well as for perinuclear antineutrophil cytoplasmic antibodies &#40;p-ANCA&#41;&#46; The specificity of anti-myeloperoxidase antibodies &#40;anti-MPO&#41; was positive&#59; whereas that of other autoantibodies &#40;anti-Jo-1&#44; anti-Scl-70&#44; anti-double-stranded DNA and anti-cyclic citrullinated peptide antibodies&#41; was negative&#46; The histopathological study of the lung specimen demonstrated small-vessel vasculitis with extensive areas of fibrosis and pulmonary emphysema &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">The presence of interstitial lung disease &#40;ILD&#41;&#44; ANCA-associated vasculitis &#40;AAV&#41;&#44; proteinuria and neuropathy enabled the classification of the disorder as microscopic polyangiitis &#40;MPA&#41;&#46; The treatment was based on pulses of methylprednisolone&#44; azathioprine and rituximab&#46; The outcome was satisfactory&#44; with improvement of the symptoms and signs 4 weeks after treatment&#44; and the absence of relapse over a 3-month period of observation&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Discussion</span><p id="par0020" class="elsevierStylePara elsevierViewall">In this case of MPA&#44; the presence of early ILD results in an uncommon pattern in lung disease&#44; rather than capillaritis with alveolar hemorrhage and the usual interstitial pneumonia&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#44;2</span></a> In ILD&#44; the main pathological finding lies in the interstitial alveolar structure&#59; however&#44; the airways and pulmonary vasculature can also be affected&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">3&#8211;5</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">The association of MPA and AAV was described by Arulkumaran et al&#46; in 510 patients&#44; in 14 &#40;2&#46;7&#37;&#41; of whom it was associated with ILD&#59; all of them were positive for p-ANCA and MPO and were diagnosed with MPA&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a> Eschun et al&#46; reported similar findings in 6 patients in which the initial manifestation was ILD&#44; which was followed by the development of vasculitis documented by positive p-ANCA and renal biopsy&#46; The histopathological study usually demonstrates rapidly progressive and&#47;or necrotizing crescentic glomerulonephritis&#46;<a class="elsevierStyleCrossRefs" href="#bib0080"><span class="elsevierStyleSup">6&#8211;8</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">The mechanisms of lung injury in ILD with AAV are not fully understood&#46; However&#44; repeated episodes of subclinical alveolar hemorrhage and&#47;or the effects of autoantibodies on the surface of neutrophils could induce proinflammatory cytokine production and the release of free radicals resulting in alveolar epithelial injury and pulmonary fibrosis&#46;<a class="elsevierStyleCrossRefs" href="#bib0095"><span class="elsevierStyleSup">9&#44;10</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">There are no guidelines for the treatment of patients with MPA and ILD&#46; At the present time&#44; they are managed along the lines of small-vessel vasculitis&#46; The prognosis of this association is uncertain&#44; and no significant differences are observed with respect to mortality&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">In conclusion&#44; the association of ILD and MPA is not very common&#46; However&#44; clinicians in their diagnostic approach to ILD must take into account the possibility of underlying AAV&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Ethical Disclosures</span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Protection of human and animal subjects</span><p id="par0045" class="elsevierStylePara elsevierViewall">The authors declare that no experiments were performed on humans or animals for this study&#46;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Confidentiality of data</span><p id="par0050" class="elsevierStylePara elsevierViewall">The authors declare that they have followed the protocols of their work center on the publication of patient data&#46;</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0050">Right to privacy and informed consent</span><p id="par0055" class="elsevierStylePara elsevierViewall">The authors have obtained the written informed consent of the patients or subjects mentioned in the article&#46; The corresponding author is in possession of this document&#46;</p></span></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0055">Conflicts of Interest</span><p id="par0060" class="elsevierStylePara elsevierViewall">The authors declare they have no conflicts of interest&#46;</p></span></span>"
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        "titulo" => "Abstract"
        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Microscopic polyangiitis &#40;MPA&#41; is a systemic disease included in the Chapel Hill 2012 Classification as necrotizing vasculitis affecting capillaries&#44; venules and arterioles&#46; It usually expresses antineutrophil cytoplasmic antibodies &#40;ANCA&#41; and has a perinuclear immunofluorescence pattern and correlation with anti-myeloperoxidase &#40;MPO&#41; antibodies&#46; Capillaritis with alveolar hemorrhage is the most common manifestation of lung disease&#46; Interstitial lung disease &#40;ILD&#41; is uncommon&#44; with usual interstitial pneumonia being the predominant pattern&#46; However&#44; other patterns such as organizing pneumonia have been described&#46; No guidelines exist for treating patients with ILD and&#44; currently&#44; ANCA-associated vasculitis &#40;AAV&#41; is managed along the lines of small vessel vasculitis&#46; The prognosis with this association is uncertain&#44; with possibilities of relapse and a fatal outcome&#46; We present a case in which ILD was the first manifestation of MPA&#44; without alveolar hemorrhage&#44; with subsequent renal involvement and&#44; in which&#44; the established treatment produced a significant clinical improvement&#46;</p></span>"
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        "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">La poliange&#237;tis microsc&#243;pica &#40;PAM&#41; es una enfermedad sist&#233;mica incluida en la clasificaci&#243;n de Chapel Hill 2012 como vasculitis necrosante que afecta capilares&#44; v&#233;nulas y arteriolas&#46; Usualmente expresa anticuerpos anti-citoplasma de neutr&#243;filo &#40;ANCA&#41;&#44; con patr&#243;n perinuclear en la inmunofluorescencia&#44; y correlaci&#243;n con los anticuerpos anti-mieloperoxidasa &#40;MPO&#41;&#46; La capilaritis con hemorragia alveolar es la manifestaci&#243;n m&#225;s usual de afecci&#243;n pulmonar&#46; La enfermedad pulmonar intersticial &#40;EPI&#41; es infrecuente&#44; siendo la neumon&#237;a intersticial com&#250;n el patr&#243;n predominante&#44; sin embargo&#44; otros patrones como la neumon&#237;a organizada han sido descritos&#46; No existen pautas de tratamiento de los pacientes con EPI y vasculitis asociada a ANCA &#40;VAA&#41;&#59; actualmente son tratados con las bases de las vasculitis de vasos peque&#241;os&#46; El pron&#243;stico de esta asociaci&#243;n es incierto&#44; con posibilidad de reca&#237;das y de curso fatal&#46; A continuaci&#243;n se presenta un caso en el que neumopat&#237;a intersticial fue la primera manifestaci&#243;n de una PAM&#44; sin hemorragia alveolar&#44; con posterior involucro renal&#44; y con el tratamiento instaurado presenta mejor&#237;a cl&#237;nica significativa&#46;</p></span>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Garc&#237;a-Nava M&#44; Mateos-Toledo H&#44; Guevara-Canseco APG&#44; Infante-Gonz&#225;lez CE&#44; Reyes-Nava DA&#44; Estrada-Castro E&#46; Enfermedad pulmonar intersticial temprana en poliangeitis microsc&#243;pica&#58; reporte de un caso y revisi&#243;n de la literatura&#46; Reumatol Clin&#46; 2018&#59;14&#58;106-108&#46;</p>"
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Case report
Early Interstitial Lung Disease in Microscopic Polyangiitis: Case Report and Literature Review
Enfermedad pulmonar intersticial temprana en poliangeitis microscópica: reporte de un caso y revisión de la literatura
Marcos García-Navaa,
Corresponding author
mark_g_n@hotmail.com

Corresponding author.
, Heidegger Mateos-Toledob, Ana Patricia Georgina Guevara-Cansecoa, Cesar Eduardo Infante-Gonzáleza, Diego Alberto Reyes-Navaa, Emilio Estrada-Castroa
a Servicio de Medicina Interna, Centro Médico ISSEMyM Toluca, Metepec, Estado de México, Mexico
b Clínica de enfermedades intersticiales del pulmón, Instituto Nacional de Enfermedades Respiratorias, Mexico
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C-reactive protein 290&#46;4<span class="elsevierStyleHsp" style=""></span>mg&#47;L&#44; total creatine kinase 3&#46;6<span class="elsevierStyleHsp" style=""></span>U&#47;L&#44; creatinine 2&#46;8<span class="elsevierStyleHsp" style=""></span>mg&#47;dL&#44; urea 86&#46;5<span class="elsevierStyleHsp" style=""></span>mg&#47;dL and blood urea nitrogen 40<span class="elsevierStyleHsp" style=""></span>mg&#47;dL&#46; In urine&#44; we observed proteinuria with a level of 75<span class="elsevierStyleHsp" style=""></span>mg&#47;dL and erythrocytes at 260<span class="elsevierStyleHsp" style=""></span>cells&#47;mL&#44; with active sediment characterized by dysmorphic erythrocytes&#46; No evidence of bacteria in urine culture&#44; blood culture&#44; sputum culture or stool culture&#46; The results of a viral panel for hepatitis C virus&#44; hepatitis B virus and human immunodeficiency virus were negative&#46; High-resolution computed tomography of the chest revealed interstitial involvement with a pattern of irregular septal thickening and areas of alveolar filling with air bronchogram in peripherally distributed patches &#40;pattern of organizing pneumonia&#41; &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; An electromyographic study showed a pattern indicating a demyelinating disease&#46; Bronchoalveolar lavage demonstrated nonspecific chronic and acute inflammation&#46; Studies focusing on autoimmunity found the patient to be positive for antinuclear antibodies at a titer of 1&#58;320 with a mitochondrial pattern&#44; as well as for perinuclear antineutrophil cytoplasmic antibodies &#40;p-ANCA&#41;&#46; The specificity of anti-myeloperoxidase antibodies &#40;anti-MPO&#41; was positive&#59; whereas that of other autoantibodies &#40;anti-Jo-1&#44; anti-Scl-70&#44; anti-double-stranded DNA and anti-cyclic citrullinated peptide antibodies&#41; was negative&#46; The histopathological study of the lung specimen demonstrated small-vessel vasculitis with extensive areas of fibrosis and pulmonary emphysema &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">The presence of interstitial lung disease &#40;ILD&#41;&#44; ANCA-associated vasculitis &#40;AAV&#41;&#44; proteinuria and neuropathy enabled the classification of the disorder as microscopic polyangiitis &#40;MPA&#41;&#46; The treatment was based on pulses of methylprednisolone&#44; azathioprine and rituximab&#46; The outcome was satisfactory&#44; with improvement of the symptoms and signs 4 weeks after treatment&#44; and the absence of relapse over a 3-month period of observation&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Discussion</span><p id="par0020" class="elsevierStylePara elsevierViewall">In this case of MPA&#44; the presence of early ILD results in an uncommon pattern in lung disease&#44; rather than capillaritis with alveolar hemorrhage and the usual interstitial pneumonia&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#44;2</span></a> In ILD&#44; the main pathological finding lies in the interstitial alveolar structure&#59; however&#44; the airways and pulmonary vasculature can also be affected&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">3&#8211;5</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">The association of MPA and AAV was described by Arulkumaran et al&#46; in 510 patients&#44; in 14 &#40;2&#46;7&#37;&#41; of whom it was associated with ILD&#59; all of them were positive for p-ANCA and MPO and were diagnosed with MPA&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a> Eschun et al&#46; reported similar findings in 6 patients in which the initial manifestation was ILD&#44; which was followed by the development of vasculitis documented by positive p-ANCA and renal biopsy&#46; The histopathological study usually demonstrates rapidly progressive and&#47;or necrotizing crescentic glomerulonephritis&#46;<a class="elsevierStyleCrossRefs" href="#bib0080"><span class="elsevierStyleSup">6&#8211;8</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">The mechanisms of lung injury in ILD with AAV are not fully understood&#46; However&#44; repeated episodes of subclinical alveolar hemorrhage and&#47;or the effects of autoantibodies on the surface of neutrophils could induce proinflammatory cytokine production and the release of free radicals resulting in alveolar epithelial injury and pulmonary fibrosis&#46;<a class="elsevierStyleCrossRefs" href="#bib0095"><span class="elsevierStyleSup">9&#44;10</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">There are no guidelines for the treatment of patients with MPA and ILD&#46; At the present time&#44; they are managed along the lines of small-vessel vasculitis&#46; The prognosis of this association is uncertain&#44; and no significant differences are observed with respect to mortality&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">In conclusion&#44; the association of ILD and MPA is not very common&#46; However&#44; clinicians in their diagnostic approach to ILD must take into account the possibility of underlying AAV&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Ethical Disclosures</span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Protection of human and animal subjects</span><p id="par0045" class="elsevierStylePara elsevierViewall">The authors declare that no experiments were performed on humans or animals for this study&#46;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Confidentiality of data</span><p id="par0050" class="elsevierStylePara elsevierViewall">The authors declare that they have followed the protocols of their work center on the publication of patient data&#46;</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0050">Right to privacy and informed consent</span><p id="par0055" class="elsevierStylePara elsevierViewall">The authors have obtained the written informed consent of the patients or subjects mentioned in the article&#46; The corresponding author is in possession of this document&#46;</p></span></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0055">Conflicts of Interest</span><p id="par0060" class="elsevierStylePara elsevierViewall">The authors declare they have no conflicts of interest&#46;</p></span></span>"
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            0 => "Poliange&#237;tis microsc&#243;pica"
            1 => "Enfermedad pulmonar intersticial"
            2 => "Patr&#243;n de neumon&#237;a organizada"
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        "titulo" => "Abstract"
        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Microscopic polyangiitis &#40;MPA&#41; is a systemic disease included in the Chapel Hill 2012 Classification as necrotizing vasculitis affecting capillaries&#44; venules and arterioles&#46; It usually expresses antineutrophil cytoplasmic antibodies &#40;ANCA&#41; and has a perinuclear immunofluorescence pattern and correlation with anti-myeloperoxidase &#40;MPO&#41; antibodies&#46; Capillaritis with alveolar hemorrhage is the most common manifestation of lung disease&#46; Interstitial lung disease &#40;ILD&#41; is uncommon&#44; with usual interstitial pneumonia being the predominant pattern&#46; However&#44; other patterns such as organizing pneumonia have been described&#46; No guidelines exist for treating patients with ILD and&#44; currently&#44; ANCA-associated vasculitis &#40;AAV&#41; is managed along the lines of small vessel vasculitis&#46; The prognosis with this association is uncertain&#44; with possibilities of relapse and a fatal outcome&#46; We present a case in which ILD was the first manifestation of MPA&#44; without alveolar hemorrhage&#44; with subsequent renal involvement and&#44; in which&#44; the established treatment produced a significant clinical improvement&#46;</p></span>"
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        "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">La poliange&#237;tis microsc&#243;pica &#40;PAM&#41; es una enfermedad sist&#233;mica incluida en la clasificaci&#243;n de Chapel Hill 2012 como vasculitis necrosante que afecta capilares&#44; v&#233;nulas y arteriolas&#46; Usualmente expresa anticuerpos anti-citoplasma de neutr&#243;filo &#40;ANCA&#41;&#44; con patr&#243;n perinuclear en la inmunofluorescencia&#44; y correlaci&#243;n con los anticuerpos anti-mieloperoxidasa &#40;MPO&#41;&#46; La capilaritis con hemorragia alveolar es la manifestaci&#243;n m&#225;s usual de afecci&#243;n pulmonar&#46; La enfermedad pulmonar intersticial &#40;EPI&#41; es infrecuente&#44; siendo la neumon&#237;a intersticial com&#250;n el patr&#243;n predominante&#44; sin embargo&#44; otros patrones como la neumon&#237;a organizada han sido descritos&#46; No existen pautas de tratamiento de los pacientes con EPI y vasculitis asociada a ANCA &#40;VAA&#41;&#59; actualmente son tratados con las bases de las vasculitis de vasos peque&#241;os&#46; El pron&#243;stico de esta asociaci&#243;n es incierto&#44; con posibilidad de reca&#237;das y de curso fatal&#46; A continuaci&#243;n se presenta un caso en el que neumopat&#237;a intersticial fue la primera manifestaci&#243;n de una PAM&#44; sin hemorragia alveolar&#44; con posterior involucro renal&#44; y con el tratamiento instaurado presenta mejor&#237;a cl&#237;nica significativa&#46;</p></span>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Garc&#237;a-Nava M&#44; Mateos-Toledo H&#44; Guevara-Canseco APG&#44; Infante-Gonz&#225;lez CE&#44; Reyes-Nava DA&#44; Estrada-Castro E&#46; Enfermedad pulmonar intersticial temprana en poliangeitis microsc&#243;pica&#58; reporte de un caso y revisi&#243;n de la literatura&#46; Reumatol Clin&#46; 2018&#59;14&#58;106-108&#46;</p>"
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Article information
ISSN: 21735743
Original language: English
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Idiomas
Reumatología Clínica (English Edition)
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