Case Report
Spontaneous pneumomediastinum complicating interstitial lung disease, associated with clinically amyopathic dermatomyositis and positive anti-MDA5 antibodies
Neumomediastino espontáneo como complicación de enfermedad pulmonar intersticial, asociada con dermatomiositis clínicamente amiopática y anticuerpos anti-MDA5 positivos
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Otra imagen axial demuestra la existencia de opacidades parcheadas peribronquiales en ambas bases pulmonares, compatibles con un patrón de neumonía organizada (flechas) B). En la reconstrucción coronal se observa un extenso neumomediastino que se extiende desde la región tiroidea (flechas negras) a lo largo de todo el mediastino (flechas blancas) C).</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Walter Alberto Sifuentes-Giraldo, María Jesús García-Villanueva, Luis Gorospe" "autores" => array:3 [ 0 => array:2 [ "nombre" => "Walter Alberto" "apellidos" => "Sifuentes-Giraldo" ] 1 => array:2 [ "nombre" => "María Jesús" "apellidos" => "García-Villanueva" ] 2 => array:2 [ "nombre" => "Luis" "apellidos" => "Gorospe" ] ] ] ] ] "idiomaDefecto" => "es" "Traduccion" => array:1 [ "en" => array:9 [ "pii" => "S2444440518300074" "doi" => "10.1016/j.rcreue.2018.04.001" "estado" => "S300" "subdocumento" => "" "abierto" => array:3 [ "ES" => false "ES2" => false "LATM" => false ] "gratuito" => false "lecturas" => array:1 [ "total" => 0 ] "idiomaDefecto" => "en" "EPUB" => 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"Ancho" => 1740 "Tamanyo" => 254082 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">Computed tomography imaging of (A) axial, (B) sagittal and (C) coronal planes of right calf that demonstrate a heterogenous collection of fluid of 17<span class="elsevierStyleHsp" style=""></span>cm<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>9.4<span class="elsevierStyleHsp" style=""></span>cm<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>7<span class="elsevierStyleHsp" style=""></span>cm in the thickness of the medial gastrocnemius muscle (arrow), with significant soft tissue edema.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Lizeth Natalia Melgarejo Moreno, Andrés Fernando Rodríguez-Gutiérrez, Giovana Alejandra Solano Villamarín, Jorge Alberto Cortés, Nario Cano Arenas" "autores" => array:5 [ 0 => array:2 [ "nombre" => 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Pulmonary emphysema, cylindrical bronchiectasis toward the lower lobes with wall thickening, mixed patchy infiltrates predominantly in the right lung, reticulomicronodular infiltrates resembling a budding tree.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Juan Camilo Díaz Coronado, María Camila Soto Osorio, Johnatan Stiven Sánchez Jaramillo" "autores" => array:3 [ 0 => array:2 [ "nombre" => "Juan Camilo" "apellidos" => "Díaz Coronado" ] 1 => array:2 [ "nombre" => "María Camila" "apellidos" => "Soto Osorio" ] 2 => array:2 [ "nombre" => "Johnatan Stiven" "apellidos" => "Sánchez Jaramillo" ] ] ] ] ] "idiomaDefecto" => "en" "Traduccion" => array:1 [ "es" => array:9 [ "pii" => "S0121812317300889" "doi" => "10.1016/j.rcreu.2017.06.001" "estado" => "S300" "subdocumento" => "" "abierto" => array:3 [ "ES" => false "ES2" => false "LATM" => false ] "gratuito" => false "lecturas" => array:1 [ "total" => 0 ] "idiomaDefecto" => "es" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0121812317300889?idApp=UINPBA00004N" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2444440518300062?idApp=UINPBA00004N" "url" => "/24444405/0000002400000004/v1_201806020530/S2444440518300062/v1_201806020530/en/main.assets" ] "en" => array:20 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Case Report</span>" "titulo" => "Spontaneous pneumomediastinum complicating interstitial lung disease, associated with clinically amyopathic dermatomyositis and positive anti-MDA5 antibodies" "tieneTextoCompleto" => true "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "259" "paginaFinal" => "264" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Walter Alberto Sifuentes-Giraldo, María Jesús García-Villanueva, Luis Gorospe" "autores" => array:3 [ 0 => array:4 [ "nombre" => "Walter Alberto" "apellidos" => "Sifuentes-Giraldo" "email" => array:1 [ 0 => "albertosifuentesg@gmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "María Jesús" "apellidos" => "García-Villanueva" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "Luis" "apellidos" => "Gorospe" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicio de Reumatología, Hospital Universitario Ramón y Cajal, Madrid, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Radiodiagnóstico, Hospital Universitario Ramón y Cajal, Madrid, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "<span class="elsevierStyleItalic">Corresponding author</span>." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Neumomediastino espontáneo como complicación de enfermedad pulmonar intersticial, asociada con dermatomiositis clínicamente amiopática y anticuerpos anti-MDA5 positivos" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0010" "etiqueta" => "Fig. 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1165 "Ancho" => 2333 "Tamanyo" => 164763 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Chest X-ray showing signs of pneumomediastinum that extends to the thyroid region (arrows), without pneumothorax or subcutaneous emphysema, and bibasilar parenchymal opacities (A) and (B).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Dermatomyositis (DM) is a systemic inflammatory disease characterized by proximal muscle weakness, myalgias and typical skin manifestations such as “heliotrope” erythema and Gottron's papules. However, some patients with DM do not develop muscle alterations (amyopathic DM), while others have mild symptoms during the course of the disease, or the muscle commitment is evidenced only by elevation of the enzymes associated with muscle damage or by myopathic alterations in the electromyogram or in the muscle biopsy (hypomyopathic DM), so they are included within the spectrum of the clinically amyopathic DM (CADM), having this group an increased risk of developing interstitial lung disease (ILD).<a class="elsevierStyleCrossRef" href="#bib0140"><span class="elsevierStyleSup">1</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">Although its etiology is unknown, there is evidence that the tissue damage in DM is produced by autoimmune mechanisms, finding circulating antibodies specific for myositis such as anti-Mi2 and anti-Jo1 in 50–70% of cases.<a class="elsevierStyleCrossRef" href="#bib0145"><span class="elsevierStyleSup">2</span></a> Recently, there have been identified antibodies directed against the protein encoded by the melanoma differentiation-associated gene 5 (MDA5), belonging to the family of Rig-I-like receptors that are related with the response to viral infections, which are present in 19–35% of the patients with CADM and are specifically associated with minimal or absent muscle involvement and rapidly progressive ILD, frequently complicated by the appearance of spontaneous pneumomediastinum (SPM).<a class="elsevierStyleCrossRefs" href="#bib0150"><span class="elsevierStyleSup">3,4</span></a> We present the case of a patient of African origin with CADM and positivity for anti-MDA5 who developed ILD and SPM during its evolution.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Case presentation</span><p id="par0015" class="elsevierStylePara elsevierViewall">An 18-year-old black woman, native from Western Sahara, who at the age of 14 years began presenting symmetric polyarthritis, fever and alopecia. At the age of 16 years, she was transferred to Algeria, where treatment with oral prednisone (maximum dose 10<span class="elsevierStyleHsp" style=""></span>mg/day) was started, developing shortly after Raynaud's phenomenon and ulcerations in the finger pads of both hands. The patient was admitted to the hospital at the age of 17 years due to dyspnea and non-productive cough, evidencing bibasilar opacities and altered spirometry with severe restrictive pattern (forced vital capacity 33% of its theoretical value), receiving intravenous empirical antibiotic therapy and being discharged with partial improvement. During this admission, positive rheumatoid factor (RF) (108<span class="elsevierStyleHsp" style=""></span>IU/ml) was detected, so she was diagnosed with rheumatoid arthritis and administration of oral methotrexate (10<span class="elsevierStyleHsp" style=""></span>mg/week) plus folic acid supplement (5<span class="elsevierStyleHsp" style=""></span>mg/week) was started.</p><p id="par0020" class="elsevierStylePara elsevierViewall">At age 18, she was transferred to Spain and admitted to our center, having at this time diffuse edema of the hands, scarring lesions in the finger pads, periungual erythema, hypopigmented macules of atrophic appearance on areas in which she had previously Gottron's papules in metacarpophalangeal and proximal interphalangeal joints (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>), indurated subcutaneous nodules in forearms and thighs, dyspnea on mild efforts, dysphagia, pyrosis and asthenia, but without muscle weakness or myalgias. The basal oxygen saturation was normal (97%) but with persistence of the restrictive pattern (forced vital capacity 34.8%), being not possible to carry out the test for diffusing capacity of the lungs for carbon monoxide (DLCO) because she mobilized an insufficient volume. The laboratory tests showed hypochromic microcytic anemia (hemoglobin 11.8<span class="elsevierStyleHsp" style=""></span>g/dl, mean corpuscular volume 74.6<span class="elsevierStyleHsp" style=""></span>fl, mean corpuscular hemoglobin 23.7<span class="elsevierStyleHsp" style=""></span>pg), lymphopenia (750/mm<span class="elsevierStyleSup">3</span>), increased acute phase reactants (erythrocyte sedimentation rate 84<span class="elsevierStyleHsp" style=""></span>mm/h, C-reactive protein 17<span class="elsevierStyleHsp" style=""></span>mg/l), polyclonal hypergammaglobulinemia (IgG 2800<span class="elsevierStyleHsp" style=""></span>mg/dl) and increase in thyroid stimulating hormone (TSH) (5.230<span class="elsevierStyleHsp" style=""></span>μIU/ml) with normal T3 and T4 levels. The rest of the biochemical study including muscle enzymes (creatine kinase 40<span class="elsevierStyleHsp" style=""></span>U/l, aldolase 2<span class="elsevierStyleHsp" style=""></span>U/l) was within normal ranges. From the immunological point of view she had ANA 1/80 with nucleolar pattern, positive anti-Ro52 and anti-MDA5, being negative the other antibodies studied (RF, anti-cyclic citrullinated peptide [anti-CCP], anti-DNA, anti-ENA, anti-Scl70, anti-centromere, antiphospholipid antibodies, anti-Mi2, anti-Jo1, anti-PL12, anti-PL7, anti-OJ, anti-EJ, anti-SRP, anti-Ku and anti-PM/Scl). The chest X-ray showed the bibasilar opacities and signs of SPM extending into the perithyroidal region (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>), although crepitation in subcutaneous cellular tissue was not evident in the physical examination, and for this reason it was performed a computed tomography that confirmed the presence of patchy peribronchial opacities of basilar predominance, radiologically compatible with a pattern of organizing pneumonia and an extensive pneumomendiastinum which was expanded from the thyroid region throughout the entire mediastinum dissecting vascular and muscle structures. (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>) There were no signs of pleural honeycombing, mediastinitis or pneumothorax.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">The patient was initially diagnosed with amyopathic DM with associated ILD and SPM, but the electromyogram demonstrated spontaneous activity suggestive of mild inflammatory myopathy, and nonspecific myopathic changes without inflammatory infiltrate were found in the muscle biopsy, therefore, it corresponded actually to hypomyopathic DM. The study was completed with capillaroscopy which showed isolated megacapillaries with preserved capillary density, esophageal manometry with weak contractions at the level of the lower half of the esophagus, and biopsy of the subcutaneous nodules with findings compatible with nonspecific panniculitis. Administration of prednisone 1<span class="elsevierStyleHsp" style=""></span>mg/kg/day was initiated with rapid improvement of dyspnea, cutaneous lesions and asthenia. Methotrexate was discontinued and azathioprine 100<span class="elsevierStyleHsp" style=""></span>mg/day was added. In the evaluation 2 months after discharge the patient was stable, with mild dyspnea and arthralgias without signs of synovitis. The chest X-ray showed an important reduction of the pulmonary infiltrates and disappearance of the signs of SPM. The patient abandoned the follow-up due to change of domicile.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0030" class="elsevierStylePara elsevierViewall">Anti-MDA5 antibodies (formerly called anti-CADM140) were described in 2005 by Sato et al., in Japanese patients with CADM and rapidly progressive ILD.<a class="elsevierStyleCrossRef" href="#bib0160"><span class="elsevierStyleSup">5</span></a> Further studies have found these antibodies in other populations and expanded their spectrum of clinical manifestations, being characteristic the presence of cutaneous and oral ulcerations, painful palmar papules/pustules, periungual erythema, erythema in elbows/knees (Gottron's sign), diffuse alopecia, panniculitis and arthralgias/arthritis.<a class="elsevierStyleCrossRefs" href="#bib0150"><span class="elsevierStyleSup">3,4,6</span></a> Due to these characteristics, as well as to the scarce muscle involvement, the preponderance of the ILD and the infrequent association with neoplasms, it has been proposed to call this picture “dermatopulmonary syndrome associated with MDA5 antibodies” to differentiate it from the classical DM,<a class="elsevierStyleCrossRef" href="#bib0165"><span class="elsevierStyleSup">6</span></a> although these antibodies are not exclusive to CADM and they have been also described in the classical and juvenile forms of the DM.<a class="elsevierStyleCrossRef" href="#bib0170"><span class="elsevierStyleSup">7</span></a> The ability of anti-MDA5 antibodies to identify patients with DM and the risk of developing rapidly progressive ILD is high, with a sensitivity of 77% and a specificity of 86%.<a class="elsevierStyleCrossRef" href="#bib0170"><span class="elsevierStyleSup">7</span></a> The racial factor can influence the clinical expressivity in patients with anti-MDA5. Approximately half of patients coming from East Asia (Japan, China and Korea) have rapidly progressive ILD and it generally corresponds to CADM,<a class="elsevierStyleCrossRef" href="#bib0175"><span class="elsevierStyleSup">8</span></a> while in Caucasian populations the severity of the ILD may be lower and the frequency of clinical myositis higher.<a class="elsevierStyleCrossRef" href="#bib0180"><span class="elsevierStyleSup">9</span></a> CADM associated with anti-MDA5 has been described in African American patients,<a class="elsevierStyleCrossRefs" href="#bib0180"><span class="elsevierStyleSup">9,10</span></a> but according to the literature review we have conducted, this is the first case in a woman of African origin.</p><p id="par0035" class="elsevierStylePara elsevierViewall">In our case, the diagnosis of RA was initially proposed because of the presence of arthritis and positive RF, a fact that is not uncommon in patients with positive anti-MDA5, since a significant proportion (65.5–81.8%) develop symmetric polyarthritis with involvement of the small joints of the hands and morning stiffness that is indistinguishable from RA, some of whom may show positivity for RF or anti-CCP and, more rarely, erosions.<a class="elsevierStyleCrossRefs" href="#bib0150"><span class="elsevierStyleSup">3,9,11</span></a> As well, is not uncommon the development of arthritis associated with “mechanic's hands”, Raynaud's phenomenon or fever in patients with positive anti-MDA5, which can also give rise to the suspicion of an antisynthetase syndrome (ASS), being recommended the determination of these antibodies when the antisynthetase antibodies are negative.<a class="elsevierStyleCrossRef" href="#bib0180"><span class="elsevierStyleSup">9</span></a> Our patient also had anti-Ro52 antibodies, which have been described as co-stimulators in the antisynthetase syndrome, increasing the severity of ILD in these cases.<a class="elsevierStyleCrossRefs" href="#bib0195"><span class="elsevierStyleSup">12,13</span></a> This has also been reported in patients with anti-MDA5 positive CADM, finding the simultaneous presence of anti-Ro52 in 19–50% of cases.<a class="elsevierStyleCrossRefs" href="#bib0150"><span class="elsevierStyleSup">3,9,14</span></a> The coexistence of both antibodies probably has implications for the pathogenesis of the ILD in this subgroup of DM. MDA5 and Ro52 (TRIM21) are cytoplasmic proteins whose expression is induced by type I interferons, therefore, it has been suggested that the interactions of both molecules could lead to the formation of molecular complexes with increased immunogenicity.<a class="elsevierStyleCrossRef" href="#bib0180"><span class="elsevierStyleSup">9</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">A peculiar characteristic of the ILD associated with DM is the frequent occurrence of SPM, with an incidence ranging between 2.2% and 8.3%, and more than half of these cases corresponding to CADM.<a class="elsevierStyleCrossRefs" href="#bib0210"><span class="elsevierStyleSup">15,16</span></a> The presence of ILD, cutaneous vasculopathy, the use of systemic glucocorticoids, a young patient and normal serum levels of muscle enzymes have been described as risk factors for developing SPM,<a class="elsevierStyleCrossRef" href="#bib0210"><span class="elsevierStyleSup">15</span></a> all of them present in our case. The presence of anti-MDA5 also seems to be a predictive factor for the development of SPM in these patients, as demonstrated by the study conducted by Koga et al., which included 79 patients with DM (58 classical and 21 amyopathic), finding that the presence of mediastinal emphysema was significantly greater in patients with positive anti-MDA5 (35% vs. 2%, <span class="elsevierStyleItalic">p</span><span class="elsevierStyleHsp" style=""></span>=<span class="elsevierStyleHsp" style=""></span>2.1<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>10<span class="elsevierStyleSup">−5</span>).<a class="elsevierStyleCrossRef" href="#bib0155"><span class="elsevierStyleSup">4</span></a> The mechanism by which the SPM develops in DM is not clearly established, but it is speculated that it could be the result of the rupture of subpleural bullae or cysts secondary to the interstitial fibrosis and the increased intra-alveolar pressure. It has also been described a weakening of the alveolar walls due to the treatment with glucocorticoids.<a class="elsevierStyleCrossRefs" href="#bib0220"><span class="elsevierStyleSup">17,18</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Regarding the tomographic patterns of ILD found in patients with positive anti-MDA5, the most frequent is that with basilar consolidation/ground glass areas (50%), followed by ground glass areas with a random distribution (33%), while the basilar reticular and peribronchovascular consolidation patterns suggestive of organizing pneumonia are usually infrequent.<a class="elsevierStyleCrossRef" href="#bib0230"><span class="elsevierStyleSup">19</span></a> The histopathology of ILD associated with anti-MDA5 positive CADM has not been systematically evaluated, but in the case reports with lung biopsy or autopsy, the findings were compatible with diffuse alveolar damage, the fibrotic variant of nonspecific interstitial pneumonia and the usual interstitial pneumonia.<a class="elsevierStyleCrossRefs" href="#bib0165"><span class="elsevierStyleSup">6,14,18-24</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">The majority of patients with ILD associated with DM have a favorable response to treatment with high-dose glucocorticoids and immunosuppressants. However, those with CADM and rapidly progressive ILD are resistant to multiple treatments and have a poor prognosis, with a mortality of 41% in patients with positive anti-MDA5, so it is recommended to treat them early and aggressively with combinations of drugs including glucocorticoids, calcineurin inhibitors, mycophenolate mofetil, intravenous immunoglobulins, cyclophosphamide and rituximab.<a class="elsevierStyleCrossRefs" href="#bib0155"><span class="elsevierStyleSup">4,8,25</span></a> The presence of SPM also influences prognosis, finding a mortality rate of 34% in patients with DM who have this complication, with 25% of them dying during the first month due to respiratory distress.<a class="elsevierStyleCrossRef" href="#bib0215"><span class="elsevierStyleSup">16</span></a> There have been published 2 cases of rapidly progressive ILD in CADM associated with anti-MDA5 that were refractory to treatment with pulses of methylprednisolone and calcineurin inhibitors and finally underwent hemoperfusion with polymyxin B, a technique that was originally developed to remove the endotoxin from the circulation in patients with sepsis, which has demonstrated to be effective in patients with respiratory distress. One of them showed dramatic improvement and reduction in the levels of anti-MDA5 antibodies,<a class="elsevierStyleCrossRef" href="#bib0265"><span class="elsevierStyleSup">26</span></a> but the other case (who presented SPM and subcutaneous emphysema as complications) only had a slight transient improvement and required plasmapheresis and intravenous immunoglobulins.<a class="elsevierStyleCrossRef" href="#bib0270"><span class="elsevierStyleSup">27</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">In conclusion, the anti-MDA5 antibodies identify a special group of patients with DM who have a tendency to present amyopathic/hypomyopathic forms, with a spectrum of mucocutaneous and articular manifestations and an increased risk to develop ILD, which can follow a rapidly progressive course and be complicated by the appearance of SPM.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Ethical disclosures</span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Protection of human and animal subjects</span><p id="par0060" class="elsevierStylePara elsevierViewall">The authors declare that no experiments were performed on humans or animals for this study.</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0050">Confidentiality of data</span><p id="par0065" class="elsevierStylePara elsevierViewall">The authors declare that they have followed the protocols of their work center on the publication of patient data.</p></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0055">Right to privacy and informed consent</span><p id="par0070" class="elsevierStylePara elsevierViewall">The authors have obtained the written informed consent of the patients or subjects mentioned in the article. The corresponding author is in possession of this document.</p></span></span><span id="sec0040" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0060">Conflict of interest</span><p id="par0075" class="elsevierStylePara elsevierViewall">The authors declare they do not have any conflict of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:10 [ 0 => array:3 [ "identificador" => "xres1033563" "titulo" => "Abstract" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0005" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec990273" "titulo" => "Keywords" ] 2 => array:3 [ "identificador" => "xres1033564" "titulo" => "Resumen" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0010" ] ] ] 3 => array:2 [ "identificador" => "xpalclavsec990272" "titulo" => "Palabras clave" ] 4 => array:2 [ "identificador" => "sec0005" "titulo" => "Introduction" ] 5 => array:2 [ "identificador" => "sec0010" "titulo" => "Case presentation" ] 6 => array:2 [ "identificador" => "sec0015" "titulo" => "Discussion" ] 7 => array:3 [ "identificador" => "sec0020" "titulo" => "Ethical disclosures" "secciones" => array:3 [ 0 => array:2 [ "identificador" => "sec0025" "titulo" => "Protection of human and animal subjects" ] 1 => array:2 [ "identificador" => "sec0030" "titulo" => "Confidentiality of data" ] 2 => array:2 [ "identificador" => "sec0035" "titulo" => "Right to privacy and informed consent" ] ] ] 8 => array:2 [ "identificador" => "sec0040" "titulo" => "Conflict of interest" ] 9 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2016-12-14" "fechaAceptado" => "2017-06-20" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec990273" "palabras" => array:6 [ 0 => "Anti-MDA5 antibodies" 1 => "Clinically amyopathic dermatomyositis" 2 => "Amyopathic dermatomyositis" 3 => "Dermatomyositis" 4 => "Interstitial lung disease" 5 => "Spontaneous pneumomediastinum" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec990272" "palabras" => array:6 [ 0 => "Anticuerpos anti-MDA5" 1 => "Dermatomiositis clínicamente amiopática" 2 => "Dermatomiositis amiopática" 3 => "Dermatomiositis hipomiopática" 4 => "Enfermedad pulmonar intersticial" 5 => "Neumomediastino espontáneo" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Clinically amyopathic dermatomyositis comprises a special group of patients within the spectrum of dermatomyositis characterized by the presence of typical skin lesions, minimal or absent muscle involvement, and increased risk of interstitial lung disease. The antibodies directed against the protein encoded by melanoma differentiation-associated gene 5 (MDA5) are present in a significant proportion of patients with clinically amyopathic dermatomyositis, who develop rapidly progressive interstitial lung disease, with high mortality and frequently complicated by the onset of spontaneous pneumomediastinum. A case is presented of an African patient with anti-MDA5 positive clinically amyopathic dermatomyositis and interstitial lung disease with tomography pattern of organizing pneumonia who developed spontaneous pneumomediastinum during its clinical course.</p></span>" ] "es" => array:2 [ "titulo" => "Resumen" "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">La dermatomiositis clínicamente amiopática comprende un grupo especial de pacientes dentro del espectro de la dermatomiositis, caracterizados por la presencia de lesiones cutáneas típicas, compromiso muscular mínimo o ausente y riesgo aumentado de enfermedad pulmonar intersticial. Los anticuerpos dirigidos contra la proteína codificada por el gen asociado con la diferenciación del melanoma 5 (MDA5), están presentes en una proporción importante de pacientes con dermatomiositis clínicamente amiopática, los cuales desarrollan enfermedad pulmonar intersticial rápidamente progresiva, con elevada mortalidad y que se complica frecuentemente con la aparición de neumomediastino espontáneo. Presentamos el caso de una paciente de origen africano con dermatomiositis clínicamente amiopática anti-MDA5 positiva y enfermedad pulmonar intersticial con patrón tomográfico de neumonía organizada, que desarrolló neumomediastino espontáneo durante su evolución.</p></span>" ] ] "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Sifuentes-Giraldo WA, García-Villanueva MJ, Gorospe L. Neumomediastino espontáneo como complicación de enfermedad pulmonar intersticial, asociada con dermatomiositis clínicamente amiopática y anticuerpos anti-MDA5 positivos. Rev Colomb Reumatol. 2017;24:259–264.</p>" ] ] "multimedia" => array:3 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 998 "Ancho" => 2333 "Tamanyo" => 227685 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Dorsal aspect of the patient's hands showing diffuse edema to middle phalanges, hypopigmented macules of atrophic appearance in metacarpophalangeal and proximal interphalangeal joints and periungal erythema (A). On the palmar side there are scarring lesions on the finger pads (B).</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Fig. 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1165 "Ancho" => 2333 "Tamanyo" => 164763 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Chest X-ray showing signs of pneumomediastinum that extends to the thyroid region (arrows), without pneumothorax or subcutaneous emphysema, and bibasilar parenchymal opacities (A) and (B).</p>" ] ] 2 => array:7 [ "identificador" => "fig0015" "etiqueta" => "Fig. 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 622 "Ancho" => 2500 "Tamanyo" => 196743 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">Axial CT image of the thorax which confirms the presence of gas dissecting the structure of the mediastinum (arrows) (A). Another axial image demonstrates the existence of patchy peribronchial opacities in both lung bases, compatible with a pattern of organizing pneumonia (arrows) (B). An extensive pneumomediastinum that extends from the thyroid region (black arrows) throughout all the mediastinum (white arrows) is observed on the coronal reconstruction (C).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:27 [ 0 => array:3 [ "identificador" => "bib0140" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:3 [ "comentario" => "48–49" "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "The clinical features, diagnosis and classification of dermatomyositis" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "L. Iaccarino" 1 => "A. Ghirardello" 2 => "S. Bettio" 3 => "M. Zen" 4 => "M. Gatto" 5 => "L. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 0 | 1 | 1 |
| 2024 October | 40 | 6 | 46 |
| 2024 September | 69 | 7 | 76 |
| 2024 August | 50 | 5 | 55 |
| 2024 July | 74 | 6 | 80 |
| 2024 June | 49 | 4 | 53 |
| 2024 May | 45 | 7 | 52 |
| 2024 April | 39 | 4 | 43 |
| 2024 March | 44 | 4 | 48 |
| 2024 February | 78 | 9 | 87 |
| 2024 January | 84 | 7 | 91 |
| 2023 December | 94 | 7 | 101 |
| 2023 November | 97 | 5 | 102 |
| 2023 October | 120 | 3 | 123 |
| 2023 September | 57 | 2 | 59 |
| 2023 August | 61 | 2 | 63 |
| 2023 July | 64 | 1 | 65 |
| 2023 June | 97 | 17 | 114 |
| 2023 May | 168 | 4 | 172 |
| 2023 April | 124 | 5 | 129 |
| 2023 March | 129 | 6 | 135 |
| 2023 February | 97 | 5 | 102 |
| 2023 January | 123 | 9 | 132 |
| 2022 December | 83 | 20 | 103 |
| 2022 November | 79 | 4 | 83 |
| 2022 October | 60 | 9 | 69 |
| 2022 September | 76 | 13 | 89 |
| 2022 August | 90 | 14 | 104 |
| 2022 July | 56 | 7 | 63 |
| 2022 June | 51 | 11 | 62 |
| 2022 May | 54 | 13 | 67 |
| 2022 April | 39 | 8 | 47 |
| 2022 March | 73 | 9 | 82 |
| 2022 February | 74 | 10 | 84 |
| 2022 January | 95 | 6 | 101 |
| 2021 December | 57 | 14 | 71 |
| 2021 November | 54 | 10 | 64 |
| 2021 October | 57 | 18 | 75 |
| 2021 September | 50 | 18 | 68 |
| 2021 August | 65 | 11 | 76 |
| 2021 July | 30 | 12 | 42 |
| 2021 June | 41 | 10 | 51 |
| 2021 May | 68 | 19 | 87 |
| 2021 April | 127 | 23 | 150 |
| 2021 March | 86 | 16 | 102 |
| 2021 February | 35 | 16 | 51 |
| 2021 January | 54 | 14 | 68 |
| 2020 December | 44 | 10 | 54 |
| 2020 November | 50 | 13 | 63 |
| 2020 October | 28 | 8 | 36 |
| 2020 September | 33 | 8 | 41 |
| 2020 August | 39 | 5 | 44 |
| 2020 July | 36 | 10 | 46 |
| 2020 June | 30 | 6 | 36 |
| 2020 May | 42 | 14 | 56 |
| 2020 April | 24 | 14 | 38 |
| 2020 March | 29 | 4 | 33 |
| 2020 February | 29 | 6 | 35 |
| 2020 January | 37 | 5 | 42 |
| 2019 December | 41 | 7 | 48 |
| 2019 November | 40 | 6 | 46 |
| 2019 October | 32 | 8 | 40 |
| 2019 September | 44 | 12 | 56 |
| 2019 August | 25 | 8 | 33 |
| 2019 July | 49 | 12 | 61 |
| 2019 June | 127 | 13 | 140 |
| 2019 May | 163 | 11 | 174 |
| 2019 April | 126 | 18 | 144 |
| 2019 March | 18 | 5 | 23 |
| 2019 February | 22 | 7 | 29 |
| 2019 January | 21 | 2 | 23 |
| 2018 December | 17 | 2 | 19 |
| 2018 November | 35 | 5 | 40 |
| 2018 October | 29 | 9 | 38 |
| 2018 September | 17 | 31 | 48 |
| 2018 August | 8 | 3 | 11 |
| 2018 July | 15 | 10 | 25 |
| 2018 June | 16 | 6 | 22 |
| 2018 May | 3 | 5 | 8 |
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