Letter to the Editor
Juvenile dermatomyositis and pneumomediastinum: a case of a very rare complication
Renata Lopes Francisco de Andradea,
, Ana Carolina Lopes Helda, Maria Teresa Terreria, Sonia Mayumi Chibab
Corresponding author
drarenatalf@gmail.com
Corresponding author at: Department of Pediatrics, Section of Rheumatology, Federal University of São Paulo – UNIFESP, Borges Lagoa, St, 802. Vila Clementino, São Paulo, SP, 04083-001, Brazil.
Corresponding author at: Department of Pediatrics, Section of Rheumatology, Federal University of São Paulo – UNIFESP, Borges Lagoa, St, 802. Vila Clementino, São Paulo, SP, 04083-001, Brazil.
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For comparative purposes, the images in each column correspond to identical axial planes.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "T. Oliveira, N. Melo, S. Guimarães, A. Morais" "autores" => array:4 [ 0 => array:2 [ "nombre" => "T." "apellidos" => "Oliveira" ] 1 => array:2 [ "nombre" => "N." "apellidos" => "Melo" ] 2 => array:2 [ "nombre" => "S." "apellidos" => "Guimarães" ] 3 => array:2 [ "nombre" => "A." 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Vila Clementino, São Paulo, SP, 04083-001, Brazil." ] ] ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 424 "Ancho" => 1505 "Tamanyo" => 77996 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Gottron´s papules over the elbows (left). Gottron´s papules on the hands (middle). Heliotrope (right).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Juvenile Dermatomyositis (JDM) is a systemic autoimmune myopathy that affects mostly the muscles and skin. It usually manifests with nonspecific systemic signs preceding typical symptoms of the disease mainly symmetrical proximal muscular weakness and pathognomonic skin lesions, i.e. heliotrope and Gottron papules. Idiopathic inflammatory myopathies are rare in the pediatric group of which the most prevalent is JDM.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Spontaneous pneumomediastinum (PNM) associated with Dermatomyositis (DM) is even rarer, with 81 cases reported overall,<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> 2 of those in children.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,4</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">We report the case of a 10 year-old caucasian boy, who presented to the emergency room with an eight-month history of fever, photosensitivity, cutaneous lesions on hands and face, arthralgia, proximal muscular weakness, significant weight loss (10<span class="elsevierStyleHsp" style=""></span>kg), dyspnea on minimal exertion and dysphonia. The patient was previously healthy, with no personal history of pulmonary disease or tuberculosis. Initial physical examination showed tachycardia, tachypnea, decreased breath sounds at pulmonary bases and diffuse crepitations. He also had heliotrope and Gottron's papules (on the hands, elbows and knees), malar rash, Raynaud's phenomenon, and arthritis in the 3<span class="elsevierStyleSup">rd</span> right proximal interphalangeal joint (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). The Childhood Myositis Assessment Scale(CMAS) score (which assesses the patient's overall muscular strength, with a maximum score of 52) was 15. Laboratory work-up showed elevated muscle enzymes (creatine phosphokinase and aspartate aminotransferase), negative anti-nuclear antibody, negative antibodies to extractable nuclear antigens (ENA), negative double-stranded native DNA and negative anti-neutrophil cytoplasm antibody (ANCA). Specific myositis antibodies were unavailable for testing. Based on the presumed diagnosis of JDM, he received intravenous pulse of methylprednisolone and subcutaneous methotrexate. On the first day of hospitalization, the patient suddenly developed cough, worsened respiratory distress and palpable cervical emphysema. An urgent computed tomography scan showed bilateral mild pneumothorax, extensive PNM with subcutaneous emphysema and bilateral pulmonary consolidations, more prominent in bases, associated with ground-glass attenuation areas (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). Treatment was with conservative, as well as broad spectrum antibiotics due to impossibility of exclude infection, but respiratory distress worsened at day fifteen. Intravenous immunoglobulin was administered, with no response, and he also started sulfamethoxazole<span class="elsevierStyleHsp" style=""></span>+<span class="elsevierStyleHsp" style=""></span>trimethoprim due to leukopenia. The patient was submitted to orotracheal intubation and drainage of PNM, with no significant improvement. By this time, pneumothorax had improved just with conservative measures. However, despite mechanical ventilation support and management of shock the refractory hypoxemia continued and he died after 23 days of hospitalization.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Although JDM affects primarily the muscles and skin, it can involve many tissues and organs, including the lungs.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,5–7</span></a> Lung disease can present as aspiration pneumonia, interstitial lung disease (ILD) or respiratory muscle weakness.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> There are limited data regarding the occurrence of PNM in JDM due to its rarity; its frequency in adults with inflammatory myopathies varies in the literature, but the largest cohort reported a prevalence of 2.2 %.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Risk factors for the occurrence of PNM in DM are: history of ILD, early age of onset (below twenty years old), cutaneous vasculopathy, normal or slightly increased levels of muscle enzymes (amyopathic dermatomyositis), previous use of glucocorticoids and the presence of anti-MDA5<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a>. There is no data for risk factors in JDM. The findings on his CT-scan were also suggestive of ILD, although infection was an important differential diagnosis. The main risk factor for DM-associated PNM is the presence of ILD, with a prevalence of 10–43 % in adult patients<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a>; in pediatric patients, Sato et al. recently reported a prevalence of 27.6 %.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> ILD was diagnosed in all patients with DM-associated PNM,<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,5,6</span></a> and while ILD is considered a poor prognostic factor,<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,5,6</span></a> isolated spontaneous PNM did not correlate with increased mortality.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> The global mortality rate of DM-associated PNM is 37.5–52.5 %.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> There is no standardized treatment for DM-associated PNM. In most cases, initial treatment was high-dose methylprednisolone, followed by numerous immunosuppressors.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,5,6</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">This case is the third pediatric case reported - previous cases were a eleven-year old girl and a nine-year old boy.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,4</span></a> All three cases had a delayed diagnosis of JDM, varying between six and fourteen months. Corticosteroid treatment was introduced in all three cases, with significant improvement in JDM and PNM in the two cases described above, but a poor outcome in ours. In all three cases, there was concomitant pulmonary disease. Romanelli et al.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> considered it pulmonary infection secondary to immunosuppression; in our case, though suggestive, the diagnosis of ILD was not possible because we didn’t have enough time to confidently rule out infection.</p><p id="par0025" class="elsevierStylePara elsevierViewall">ILD is an important risk factor for the onset of PNM, and those two complications combined are responsible for a high mortality rate in patients with DM.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,5,6</span></a> As there is no data related to PNM in JDM, the extrapolated data from adult studies is important for the identification and management of patients at risk. Thus, we stress the importance of considering the diagnosis of JDM and its complications in the presence of proximal muscle weakness and nonspecific respiratory symptoms All children already diagnosed with JDM must have a thorough evaluation for subjacent lung disease.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Funding</span><p id="par0030" class="elsevierStylePara elsevierViewall">None.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Competing interests</span><p id="par0035" class="elsevierStylePara elsevierViewall">None.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Authors contribution</span><p id="par0040" class="elsevierStylePara elsevierViewall">Active participation in patient care and the search for treatment options. Writing of the clinical case and research of articles that could support the treatment and the case report</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:4 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Funding" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Competing interests" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Authors contribution" ] 3 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2019-07-23" "multimedia" => array:2 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 424 "Ancho" => 1505 "Tamanyo" => 77996 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Gottron´s papules over the elbows (left). Gottron´s papules on the hands (middle). Heliotrope (right).</p>" ] ] 1 => array:8 [ "identificador" => "fig0010" "etiqueta" => "Fig. 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1157 "Ancho" => 1505 "Tamanyo" => 166335 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Bilateral pneumothorax, extensive PNM with subcutaneous emphysema and bilateral consolidations, ground-glass attenuation areas.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:7 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "[1]" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "The lung is involved in juvenile dermatomyositis" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "G. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 October | 44 | 39 | 83 |
| 2024 September | 63 | 20 | 83 |
| 2024 August | 74 | 32 | 106 |
| 2024 July | 64 | 31 | 95 |
| 2024 June | 37 | 33 | 70 |
| 2024 May | 43 | 44 | 87 |
| 2024 April | 37 | 34 | 71 |
| 2024 March | 39 | 22 | 61 |
| 2024 February | 31 | 26 | 57 |
| 2024 January | 27 | 24 | 51 |
| 2023 December | 15 | 24 | 39 |
| 2023 November | 49 | 52 | 101 |
| 2023 October | 23 | 46 | 69 |
| 2023 September | 24 | 30 | 54 |
| 2023 August | 25 | 25 | 50 |
| 2023 July | 29 | 33 | 62 |
| 2023 June | 27 | 22 | 49 |
| 2023 May | 43 | 43 | 86 |
| 2023 April | 29 | 22 | 51 |
| 2023 March | 55 | 26 | 81 |
| 2023 February | 43 | 22 | 65 |
| 2023 January | 34 | 22 | 56 |
| 2022 December | 68 | 29 | 97 |
| 2022 November | 65 | 43 | 108 |
| 2022 October | 103 | 42 | 145 |
| 2022 September | 53 | 32 | 85 |
| 2022 August | 94 | 45 | 139 |
| 2022 July | 104 | 54 | 158 |
| 2022 June | 50 | 23 | 73 |
| 2022 May | 49 | 46 | 95 |
| 2022 April | 54 | 45 | 99 |
| 2022 March | 51 | 45 | 96 |
| 2022 February | 43 | 45 | 88 |
| 2022 January | 71 | 43 | 114 |
| 2021 December | 30 | 42 | 72 |
| 2021 November | 45 | 35 | 80 |
| 2021 October | 56 | 70 | 126 |
| 2021 September | 26 | 35 | 61 |
| 2021 August | 45 | 32 | 77 |
| 2021 July | 35 | 31 | 66 |
| 2021 June | 50 | 40 | 90 |
| 2021 May | 55 | 43 | 98 |
| 2021 April | 149 | 122 | 271 |
| 2021 March | 62 | 57 | 119 |
| 2021 February | 68 | 29 | 97 |
| 2021 January | 37 | 38 | 75 |
| 2020 December | 55 | 33 | 88 |
| 2020 November | 75 | 26 | 101 |
| 2020 October | 90 | 48 | 138 |
| 2020 September | 152 | 100 | 252 |
| 2020 August | 45 | 35 | 80 |
| 2020 July | 35 | 34 | 69 |
| 2020 June | 22 | 18 | 40 |
| 2020 May | 25 | 20 | 45 |
| 2020 April | 31 | 13 | 44 |
| 2020 March | 19 | 18 | 37 |
| 2020 February | 21 | 27 | 48 |
| 2020 January | 27 | 32 | 59 |
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