Case and Research Letter
Adult Dermatomyositis Associated With Lipodystrophy
Dermatomiositis del adulto asociada a lipodistrofia
M. Pretel
, M. Navedo, L. Marqués, L. Aguado
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Departamento de Dermatología, Clínica Universidad de Navarra, Pamplona, Navarra, Spain
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Pretel, M. Navedo, L. Marqués, L. Aguado" "autores" => array:4 [ 0 => array:4 [ "nombre" => "M." "apellidos" => "Pretel" "email" => array:1 [ 0 => "mpretel@unav.es" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">¿</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "M." "apellidos" => "Navedo" ] 2 => array:2 [ "nombre" => "L." "apellidos" => "Marqués" ] 3 => array:2 [ "nombre" => "L." "apellidos" => "Aguado" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Departamento de Dermatología, Clínica Universidad de Navarra, Pamplona, Navarra, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Dermatomiositis del adulto asociada a lipodistrofia" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 644 "Ancho" => 900 "Tamanyo" => 72305 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Atrophic erythematous lesions in the interphalangeal joints of the fingers.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0065" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">To the Editor:</span></p><p id="par0005" class="elsevierStylePara elsevierViewall">Lipodystrophy is a rare disease characterized by loss of adipose tissue that can be associated with metabolic disorders and autoimmune diseases, primarily juvenile dermatomyositis. Lipodystrophy in association with adult-onset dermatomyositis, however, is very rare.</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 23-year-old woman consulted for erythema and bilateral palpebral edema that had appeared 1 year earlier and skin lesions on the dorsum of the interphalangeal joints of both hands. The patient reported that she had experienced muscle pain and weakness over the last 6 months, as well as joint pain in her hands, wrists, elbows, and knees. She also mentioned a progressive loss of fat in the facial region and arms over the last 2 months. The patient had no past history of interest and was not taking any medication. She also reported no family history of connective tissue disease.</p><p id="par0015" class="elsevierStylePara elsevierViewall">In the physical examination, we observed a marked loss of adipose tissue in the face and arms; the trunk and lower limbs were unaffected by this loss (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). There were atrophic erythematous lesions on the skin of the elbows and interphalangeal joints of the fingers (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). We also detected the presence of periorbital erythema and periungual telangiectasia (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). The patient had rigidity in the joints of the hands, with no signs of arthritis, and proximal muscle weakness (grade 4-5 according to the Medical Research Council scale).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">The blood tests showed normal levels of creatine kinase and aldolase. The results of the antinuclear antibody test were positive, with a titer of 1:320 and a granular pattern. The C3 and C4 fractions of the complement were within normal limits. The anti-Jo1, anti-Ro, anti-La, anti-nRNP, anti-Sm, anti-ssDNA and anti-dsDNA antibody test results were negative. We detected no abnormalities in serum lipid or glucose levels.</p><p id="par0025" class="elsevierStylePara elsevierViewall">Biopsy of the hand lesions showed lichenoid dermatitis with hydropic degeneration of the basement membrane and upper dermal edema. There were no immunoglobulin (Ig) G, IgA, fibrinogen, or complement deposits in the direct immunofluorescence assay. An electrophysiological study showed no abnormalities. We therefore performed a muscle biopsy, which revealed muscle fiber necrosis and a lymphoplasmacytic inflammatory infiltrate. Magnetic resonance imaging showed thickening of the perimuscular fascia of the arms, shoulders, buttocks, and thighs. Based on the above findings, we arrived at the diagnosis of dermatomyositis. Given this diagnosis in an adult, we followed up with positron emission tomography, which ruled out underlying neoplastic disease at the time of the visit and 2 years later.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Lipodystrophy is a rare disease characterized by the loss of subcutaneous adipose tissue. It is classified as inherited or acquired and, depending on its location, as generalized, partial (relatively extensive but not generalized), or localized.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> When lipodystrophy is generalized or partial, it is often associated with metabolic disorders, such as insulin resistance and hypertriglyceridemia.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Acquired lipodystrophy has been associated with infections, antiretroviral therapy for the treatment of human immunodeficiency virus infection and with autoimmune diseases such as rheumatoid arthritis, systemic lupus erythematosus, Sjögren syndrome, and dermatomyositis.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Dermatomyositis (specifically, juvenile dermatomyositis) is the autoimmune disease most frequently related to the development of lipodystrophy.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> The prevalence of lipodystrophy in juvenile dermatomyositis varies among studies from 10% to 40%. In the only study performed with a large number of patients, acquired lipodystrophy was observed in 8% of patients with juvenile dermatomyositis, while only 1 of the 692 adult patients with dermatomyositis had lipodystrophy.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> To date, only 2 cases of lipodystrophy associated with adult-onset dermatomyositis have been published.<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5,6</span></a> The reasons for this lack of association are unknown.</p><p id="par0040" class="elsevierStylePara elsevierViewall">One study has shown that a number of manifestations of dermatomyositis, such as joint rigidity, muscle atrophy, panniculitis, and calcinosis, could be related to the development of lipodystrophy.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Of all these signs, our patient had only joint rigidity in the hands.</p><p id="par0045" class="elsevierStylePara elsevierViewall">The etiology and pathogenesis of lipodystrophy associated with autoimmune disease remain unclear. Patients with partial lipodystrophy often have low C3 levels, which may trigger the alternative complement pathway and promote development of the disease.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">The majority of patients described in the literature developed lipodystrophy years after the onset of dermatomyositis, as a late complication of severe, chronic disease; our patient, however, developed symptoms of dermatomyositis and lipodystrophy practically simultaneously. In contrast to what occurs in most cases of partial lipodystrophy, there were no metabolic abnormalities in our patient. We were also unable to demonstrate etiologic or pathogenic mechanisms associated with immunologic abnormalities such as low levels of C3. In agreement with reports from similar cases in the literature,<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5,6</span></a> treatment with oral prednisone and mycophenolate mofetil did not improve the facial lipoatrophy in our patient.</p><p id="par0055" class="elsevierStylePara elsevierViewall">We have presented a very rare case of adult lipoatrophy simultaneously associated with dermatomyositis.</p><p id="par0060" class="elsevierStylePara elsevierViewall">Based on published data, it would be prudent to closely monitor all patients with dermatomyositis who develop a loss of subcutaneous fat in order to progressively rule out abnormalities in the metabolism of lipids and carbohydrates.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Pretel M, et al. Dermatomiositis del adulto asociada a lipodistrofia. Actas Dermosifiliogr. 2013;104:724–5.</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" => 735 "Ancho" => 900 "Tamanyo" => 92517 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Palpebral erythema and loss of fatty tissue in both cheeks.</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" => 644 "Ancho" => 900 "Tamanyo" => 72305 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Atrophic erythematous lesions in the interphalangeal joints of the fingers.</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" => 672 "Ancho" => 900 "Tamanyo" => 43610 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Periungual telangiectasias observed with capillaroscopy.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:6 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Acquired and inherited lipodystrophies" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:1 [ 0 => "A. 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Abuav" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:5 [ "tituloSerie" => "J Am Acad Dermatol" "fecha" => "2010" "volumen" => "63" "paginaInicial" => "46" "paginaFinal" => "48" ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/15782190/0000010400000008/v1_201309240048/S1578219013001844/v1_201309240048/en/main.assets" "Apartado" => array:4 [ "identificador" => "6157" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Case and Research Letters" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/15782190/0000010400000008/v1_201309240048/S1578219013001844/v1_201309240048/en/main.pdf?idApp=UINPBA000044&text.app=https://actasdermo.org/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1578219013001844?idApp=UINPBA000044" ]
| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 15 | 11 | 26 |
| 2024 Octubre | 105 | 53 | 158 |
| 2024 Septiembre | 138 | 31 | 169 |
| 2024 Agosto | 166 | 94 | 260 |
| 2024 Julio | 129 | 42 | 171 |
| 2024 Junio | 152 | 45 | 197 |
| 2024 Mayo | 142 | 48 | 190 |
| 2024 Abril | 125 | 36 | 161 |
| 2024 Marzo | 129 | 48 | 177 |
| 2024 Febrero | 105 | 47 | 152 |
| 2024 Enero | 116 | 43 | 159 |
| 2023 Diciembre | 119 | 56 | 175 |
| 2023 Noviembre | 121 | 62 | 183 |
| 2023 Octubre | 104 | 33 | 137 |
| 2023 Septiembre | 102 | 65 | 167 |
| 2023 Agosto | 85 | 38 | 123 |
| 2023 Julio | 106 | 68 | 174 |
| 2023 Junio | 80 | 40 | 120 |
| 2023 Mayo | 111 | 61 | 172 |
| 2023 Abril | 120 | 56 | 176 |
| 2023 Marzo | 106 | 58 | 164 |
| 2023 Febrero | 84 | 45 | 129 |
| 2023 Enero | 67 | 43 | 110 |
| 2022 Diciembre | 77 | 49 | 126 |
| 2022 Noviembre | 33 | 59 | 92 |
| 2022 Octubre | 41 | 36 | 77 |
| 2022 Septiembre | 45 | 64 | 109 |
| 2022 Agosto | 30 | 64 | 94 |
| 2022 Julio | 41 | 55 | 96 |
| 2022 Junio | 29 | 49 | 78 |
| 2022 Mayo | 60 | 48 | 108 |
| 2022 Abril | 71 | 70 | 141 |
| 2022 Marzo | 60 | 64 | 124 |
| 2022 Febrero | 53 | 44 | 97 |
| 2022 Enero | 55 | 81 | 136 |
| 2021 Diciembre | 56 | 67 | 123 |
| 2021 Noviembre | 78 | 60 | 138 |
| 2021 Octubre | 81 | 84 | 165 |
| 2021 Septiembre | 81 | 76 | 157 |
| 2021 Agosto | 78 | 63 | 141 |
| 2021 Julio | 98 | 50 | 148 |
| 2021 Junio | 69 | 49 | 118 |
| 2021 Mayo | 53 | 45 | 98 |
| 2021 Abril | 120 | 54 | 174 |
| 2021 Marzo | 87 | 62 | 149 |
| 2021 Febrero | 68 | 40 | 108 |
| 2021 Enero | 55 | 30 | 85 |
| 2020 Diciembre | 48 | 27 | 75 |
| 2020 Noviembre | 25 | 18 | 43 |
| 2020 Octubre | 30 | 41 | 71 |
| 2020 Septiembre | 37 | 40 | 77 |
| 2020 Agosto | 25 | 30 | 55 |
| 2020 Julio | 27 | 26 | 53 |
| 2020 Junio | 46 | 35 | 81 |
| 2020 Mayo | 36 | 50 | 86 |
| 2020 Abril | 38 | 37 | 75 |
| 2020 Marzo | 35 | 24 | 59 |
| 2020 Febrero | 10 | 19 | 29 |
| 2020 Enero | 2 | 26 | 28 |
| 2019 Diciembre | 8 | 17 | 25 |
| 2019 Noviembre | 2 | 2 | 4 |
| 2019 Octubre | 4 | 11 | 15 |
| 2019 Septiembre | 8 | 8 | 16 |
| 2019 Agosto | 4 | 8 | 12 |
| 2019 Julio | 4 | 8 | 12 |
| 2019 Junio | 6 | 10 | 16 |
| 2019 Mayo | 4 | 49 | 53 |
| 2019 Abril | 3 | 1 | 4 |
| 2019 Marzo | 2 | 7 | 9 |
| 2019 Febrero | 4 | 0 | 4 |
| 2018 Diciembre | 2 | 0 | 2 |
| 2018 Noviembre | 1 | 0 | 1 |
| 2018 Octubre | 3 | 2 | 5 |
| 2018 Septiembre | 2 | 1 | 3 |
| 2018 Agosto | 0 | 5 | 5 |
| 2018 Julio | 0 | 3 | 3 |
| 2018 Junio | 0 | 2 | 2 |
| 2018 Mayo | 0 | 6 | 6 |
| 2018 Abril | 0 | 1 | 1 |
| 2018 Marzo | 16 | 11 | 27 |
| 2018 Febrero | 105 | 9 | 114 |
| 2018 Enero | 131 | 8 | 139 |
| 2017 Diciembre | 109 | 10 | 119 |
| 2017 Noviembre | 84 | 4 | 88 |
| 2017 Octubre | 59 | 10 | 69 |
| 2017 Septiembre | 72 | 12 | 84 |
| 2017 Agosto | 80 | 7 | 87 |
| 2017 Julio | 54 | 18 | 72 |
| 2017 Junio | 73 | 29 | 102 |
| 2017 Mayo | 75 | 23 | 98 |
| 2017 Abril | 63 | 10 | 73 |
| 2017 Marzo | 78 | 22 | 100 |
| 2017 Febrero | 69 | 17 | 86 |
| 2017 Enero | 72 | 13 | 85 |
| 2016 Diciembre | 71 | 9 | 80 |
| 2016 Noviembre | 131 | 13 | 144 |
| 2016 Octubre | 192 | 28 | 220 |
| 2016 Septiembre | 245 | 11 | 256 |
| 2016 Agosto | 195 | 4 | 199 |
| 2016 Julio | 87 | 9 | 96 |
| 2016 Junio | 12 | 9 | 21 |
| 2016 Mayo | 11 | 6 | 17 |
| 2016 Abril | 5 | 3 | 8 |
| 2016 Marzo | 3 | 22 | 25 |
| 2016 Febrero | 6 | 1 | 7 |
| 2016 Enero | 10 | 4 | 14 |
| 2015 Diciembre | 11 | 17 | 28 |
| 2015 Noviembre | 19 | 2 | 21 |
| 2015 Octubre | 16 | 15 | 31 |
| 2015 Septiembre | 5 | 3 | 8 |
| 2015 Agosto | 9 | 8 | 17 |
| 2015 Julio | 138 | 8 | 146 |
| 2015 Junio | 61 | 3 | 64 |
| 2015 Mayo | 71 | 4 | 75 |
| 2015 Abril | 48 | 12 | 60 |
| 2015 Marzo | 44 | 3 | 47 |
| 2015 Febrero | 56 | 6 | 62 |
| 2015 Enero | 50 | 1 | 51 |
| 2014 Diciembre | 40 | 6 | 46 |
| 2014 Noviembre | 46 | 6 | 52 |
| 2014 Octubre | 53 | 7 | 60 |
| 2014 Septiembre | 48 | 2 | 50 |
| 2014 Agosto | 39 | 3 | 42 |
| 2014 Julio | 44 | 5 | 49 |
| 2014 Junio | 61 | 5 | 66 |
| 2014 Mayo | 59 | 5 | 64 |
| 2014 Abril | 38 | 6 | 44 |
| 2014 Marzo | 53 | 6 | 59 |
| 2014 Febrero | 41 | 4 | 45 |
| 2014 Enero | 32 | 2 | 34 |
| 2013 Diciembre | 16 | 3 | 19 |
| 2013 Noviembre | 1 | 4 | 5 |
| 2013 Octubre | 2 | 3 | 5 |
| 2013 Septiembre | 2 | 0 | 2 |
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