Case and Research Letter
Edematous Dermatomyositis with Probable Evans Syndrome
Dermatomiositis edematosa asociada a probable síndrome de Evans
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Note the lentiginous epidermal hyperplasia with hyperpigmented bulbous projections of the epidermal rete ridges and increased basal melanocytes (hematoxylin-eosin [H-E], original magnification ×10; detail (inset), original magnification ×40). B, Findings from a biopsy specimen taken from the back. Notice the dendritic melanocytes in the reticular and papillary dermis and hyperpigmentation of the basal layer of the epidermis (H-E, original magnification ×20).</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "M. García-Arpa, M. Franco-Muñoz, M.A. Flores-Terry, C. Ramos-Rodríguez" "autores" => array:4 [ 0 => array:2 [ "nombre" => "M." "apellidos" => "García-Arpa" ] 1 => array:2 [ "nombre" => "M." "apellidos" => "Franco-Muñoz" ] 2 => array:2 [ "nombre" => "M.A." "apellidos" => "Flores-Terry" ] 3 => array:2 [ "nombre" => "C." 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Blasco-Morente, I. Sánchez-Carpintero" "autores" => array:2 [ 0 => array:2 [ "nombre" => "G." "apellidos" => "Blasco-Morente" ] 1 => array:2 [ "nombre" => "I." 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Flores-Terry, M. García-Arpa, J. Anino-Fernández, M.D. Mínguez-Sánchez" "autores" => array:4 [ 0 => array:4 [ "nombre" => "M.Á." "apellidos" => "Flores-Terry" "email" => array:1 [ 0 => "miguelterry85@hotmail.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" => "M." "apellidos" => "García-Arpa" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "J." "apellidos" => "Anino-Fernández" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "M.D." "apellidos" => "Mínguez-Sánchez" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicio de Dermatología, Hospital General Universitario de Ciudad Real, Ciudad Real, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Reumatología, Hospital General Universitario de Ciudad Real, Ciudad Real, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Dermatomiositis edematosa asociada a probable síndrome de Evans" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 966 "Ancho" => 851 "Tamanyo" => 132496 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Erythematous-violaceous rash affecting the face and neck. The rash is symmetrical and particularly noticeable on the eyelids.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Inflammatory diseases of the muscles and skin are rare and orphan conditions. Dermatomyositis is an idiopathic inflammatory disorder associated with typical muscle and/or skin manifestations. Presentation with edema, ie, edematous dermatomyositis, is an infrequent variant. We report a new case of edematous dermatomyositis and review the literature.</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 52-year-old woman with no clinical history of interest consulted with a pruriginous rash that first appeared 1 month previously. The rash took the form of violaceous erythema on the center of the face and around the eyes, erythematous macules on the dorsum of the metacarpophalangeal joints, and flagellate erythema on the back. The patient also experienced disabling muscle weakness that mainly affected proximal areas and intense dysphagia and dysphonia. The only findings of note in the laboratory workup were increased muscle and liver enzyme values, as follows: creatine phosphokinase, 4005<span class="elsevierStyleHsp" style=""></span>IU/L (><span class="elsevierStyleHsp" style=""></span>140); aldolase, 44.4<span class="elsevierStyleHsp" style=""></span>IU/L (><span class="elsevierStyleHsp" style=""></span>7.5); aspartate aminotransferase, 336<span class="elsevierStyleHsp" style=""></span>IU/L (><span class="elsevierStyleHsp" style=""></span>31); alanine aminotransferase, 187<span class="elsevierStyleHsp" style=""></span>IU/L (><span class="elsevierStyleHsp" style=""></span>40); γ-glutamyl transferase, 109<span class="elsevierStyleHsp" style=""></span>IU/L (><span class="elsevierStyleHsp" style=""></span>30); and lactate dehydrogenase, 1022<span class="elsevierStyleHsp" style=""></span>IU/L (><span class="elsevierStyleHsp" style=""></span>385). The results of antibody testing (antinuclear antibodies, anti-RNP, anti-Jo1, and anti-p155) were negative. Analysis of a skin biopsy specimen revealed vacuolar changes at the dermal-epidermal junction, solitary necrotic keratinocytes, and mucin deposits in the dermis. The electromyogram revealed signs of inflammatory myopathy, and occult underlying neoplasm was ruled out by tumor markers and positron emission tomography and computed tomography imaging. These findings confirmed the diagnosis of dermatomyositis, and treatment was started with intravenous methylprednisolone (1<span class="elsevierStyleHsp" style=""></span>mg/kg/d); after 5 days, 4 doses of immunoglobulin (1.5<span class="elsevierStyleHsp" style=""></span>g/kg/dose) were added, although there was little improvement. During the following months, the patient's condition progressed with intense edema affecting the face, neck, and upper extremities (visible on the magnetic resonance image) and myositis (<a class="elsevierStyleCrossRefs" href="#fig0005">Figs. 1–3</a>). Therefore, methotrexate was added to the treatment regimen at 2 months, hydroxychloroquine at 4 months, and, given the lack of improvement, rituximab (1<span class="elsevierStyleHsp" style=""></span>g) at 6 months in 2 doses separated by 2 weeks. The response was good, mainly in the skin. During follow-up, occasional thrombocytopenia and anemia (hemoglobin, 10.8<span class="elsevierStyleHsp" style=""></span>g/dL; platelets, 58 000/μL) were recorded, as were increased values for indirect bilirubin (1<span class="elsevierStyleHsp" style=""></span>mg/dL [><span class="elsevierStyleHsp" style=""></span>0.7]) and lactate dehydrogenase (641<span class="elsevierStyleHsp" style=""></span>IU/L [><span class="elsevierStyleHsp" style=""></span>385]). These findings were compatible with Evans syndrome with dermatomyositis occurring alongside worsening muscle enzyme values. However, as the patient was receiving treatment with corticosteroids, it was impossible to perform the Coombs test to confirm this associated autoimmune etiology. Similarly, it was not possible to rule out other etiologies.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Dermatomyositis is an autoimmune disease that mainly affects the skin and muscle. It has traditionally taken the form of mild periorbital edema accompanied by heliotrope rash. However, edematous dermatomyositis involves more extensive swelling. It is also a rare clinical variant of the condition, with only 23 cases reported in the literature.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> The etiology and pathogenesis remain unclear, although some authors have pointed to intense inflammatory activity with activation and deposition of complement, leading to vascular disease with muscle microinfarcts that in turn increase vascular permeability.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1–5</span></a> This manifests clinically as edema affecting muscle and/or subcutaneous tissue, with or without pitting. Age at presentation is variable,<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,2,6</span></a> with most adult cases occurring in women.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> Edema mainly affects the upper extremities, although it may be generalized<a class="elsevierStyleCrossRefs" href="#bib0070"><span class="elsevierStyleSup">4,6,7</span></a>; cases of local edema have also been reported.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2,8,9</span></a> Edematous dermatomyositis usually progresses more rapidly than classic dermatomyositis.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> Edema usually develops after skin involvement, although it may also be the initial presentation<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a>; there have even been reports of edematous dermatomyositis with no other cutaneous findings.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> Muscle involvement and dysphagia are frequently associated with edematous dermatomyositis,<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,4</span></a> as observed in the case we report.</p><p id="par0020" class="elsevierStylePara elsevierViewall">It remains unclear whether this clinical presentation of dermatomyositis implies a greater risk of neoplasm<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a>; according to a recently published literature review, the risk of neoplasm was recorded in fewer than 30% of cases (6 of 23 cases reviewed).<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">Before a diagnosis of edematous dermatomyositis can be confirmed, it is important to rule out other, secondary causes of edema,<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> such as kidney, heart, and thyroid disease, as well as hypoproteinemia. Treatment of edematous dermatomyositis should be intensive and early, given the potentially severe nature of the symptoms<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,4,5,9,10</span></a> and the probable poorer prognosis than with classic dermatomyositis.<a class="elsevierStyleCrossRefs" href="#bib0070"><span class="elsevierStyleSup">4,6,10</span></a> The combination of high doses of intravenous corticosteroids and immunosuppressants seems to be a good alternative; immunoglobulins can be added when there is no response,<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">3,5,8</span></a> and rituximab can be administered in refractory cases.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,2</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Evans syndrome (anemia and/or autoimmune thrombocytopenia) rarely occurs with dermatomyositis. The first case was reported in 1990 in a woman with generalized edematous dermatomyositis.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> Although we were unable to confirm an autoimmune etiology in the case we report, the clinical and laboratory data were consistent with this diagnosis.</p><p id="par0035" class="elsevierStylePara elsevierViewall">We report a new case of severe edematous dermatomyositis that proved refractory to several systemic treatments but responded well to rituximab. The condition probably occurred with Evans syndrome.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of Interest</span><p id="par0040" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:2 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Conflicts of Interest" ] 1 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Flores-Terry MÁ, García-Arpa M, Anino-Fernández J, Mínguez-Sánchez MD. Dermatomiositis edematosa asociada a probable síndrome de Evans. Actas Dermosifiliogr. 2017;108:673–675.</p>" ] ] "multimedia" => array:3 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 966 "Ancho" => 851 "Tamanyo" => 132496 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Erythematous-violaceous rash affecting the face and neck. The rash is symmetrical and particularly noticeable on the eyelids.</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 699 "Ancho" => 975 "Tamanyo" => 105340 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Erythematous macular lesions on the metacarpophalangeal joints and the dorsum of the fingers, with edema (the arrow indicates the pressure exerted by the patient's ring).</p>" ] ] 2 => array:7 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 1271 "Ancho" => 850 "Tamanyo" => 92818 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Magnetic resonance image of the left arm, where a hyperintense signal in several muscle groups is compatible with myositis. It is also possible to observe edema affecting the muscle and subcutaneous cellular tissue (the large red arrow points to an affected muscle group, and the small red arrow points to an area of edema in the subcutaneous cellular tissue).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0055" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Severe edematous dermatomyositis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "R. Goussot" 1 => "C. Wettlé" 2 => "C. Le Coz" 3 => "B. Cribier" 4 => "D. Lipsker" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.annder.2015.10.594" "Revista" => array:6 [ "tituloSerie" => "Ann Dermatol Venereol." 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| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 22 | 5 | 27 |
| 2024 Octubre | 143 | 58 | 201 |
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| 2023 Noviembre | 172 | 25 | 197 |
| 2023 Octubre | 216 | 21 | 237 |
| 2023 Septiembre | 190 | 34 | 224 |
| 2023 Agosto | 105 | 19 | 124 |
| 2023 Julio | 130 | 30 | 160 |
| 2023 Junio | 121 | 25 | 146 |
| 2023 Mayo | 131 | 38 | 169 |
| 2023 Abril | 131 | 31 | 162 |
| 2023 Marzo | 137 | 38 | 175 |
| 2023 Febrero | 104 | 26 | 130 |
| 2023 Enero | 82 | 36 | 118 |
| 2022 Diciembre | 102 | 53 | 155 |
| 2022 Noviembre | 33 | 17 | 50 |
| 2022 Octubre | 51 | 22 | 73 |
| 2022 Septiembre | 26 | 31 | 57 |
| 2022 Agosto | 40 | 33 | 73 |
| 2022 Julio | 29 | 43 | 72 |
| 2022 Junio | 28 | 20 | 48 |
| 2022 Mayo | 137 | 37 | 174 |
| 2022 Abril | 144 | 47 | 191 |
| 2022 Marzo | 123 | 53 | 176 |
| 2022 Febrero | 109 | 36 | 145 |
| 2022 Enero | 86 | 56 | 142 |
| 2021 Diciembre | 99 | 48 | 147 |
| 2021 Noviembre | 80 | 63 | 143 |
| 2021 Octubre | 75 | 57 | 132 |
| 2021 Septiembre | 75 | 42 | 117 |
| 2021 Agosto | 54 | 42 | 96 |
| 2021 Julio | 51 | 44 | 95 |
| 2021 Junio | 48 | 37 | 85 |
| 2021 Mayo | 72 | 49 | 121 |
| 2021 Abril | 149 | 68 | 217 |
| 2021 Marzo | 87 | 41 | 128 |
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| 2021 Enero | 47 | 46 | 93 |
| 2020 Diciembre | 38 | 33 | 71 |
| 2020 Noviembre | 46 | 34 | 80 |
| 2020 Octubre | 34 | 23 | 57 |
| 2020 Septiembre | 44 | 19 | 63 |
| 2020 Agosto | 38 | 31 | 69 |
| 2020 Julio | 37 | 26 | 63 |
| 2020 Junio | 37 | 35 | 72 |
| 2020 Mayo | 13 | 16 | 29 |
| 2020 Abril | 21 | 12 | 33 |
| 2020 Marzo | 26 | 20 | 46 |
| 2020 Febrero | 7 | 0 | 7 |
| 2019 Diciembre | 8 | 0 | 8 |
| 2019 Noviembre | 6 | 0 | 6 |
| 2019 Septiembre | 10 | 0 | 10 |
| 2019 Agosto | 4 | 0 | 4 |
| 2019 Julio | 4 | 0 | 4 |
| 2019 Junio | 4 | 0 | 4 |
| 2019 Mayo | 6 | 0 | 6 |
| 2019 Abril | 2 | 1 | 3 |
| 2019 Febrero | 6 | 0 | 6 |
| 2019 Enero | 1 | 0 | 1 |
| 2018 Diciembre | 2 | 0 | 2 |
| 2018 Noviembre | 2 | 0 | 2 |
| 2018 Octubre | 3 | 0 | 3 |
| 2018 Septiembre | 2 | 0 | 2 |
| 2018 Febrero | 21 | 7 | 28 |
| 2018 Enero | 39 | 7 | 46 |
| 2017 Diciembre | 42 | 9 | 51 |
| 2017 Noviembre | 37 | 8 | 45 |
| 2017 Octubre | 33 | 9 | 42 |
| 2017 Septiembre | 182 | 25 | 207 |
| 2017 Agosto | 45 | 22 | 67 |
| 2017 Julio | 7 | 9 | 16 |
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