Case and Research Letter
Adult Dermatomyositis Associated With Lipodystrophy
Dermatomiositis del adulto asociada a lipodistrofia
M. Pretel
, M. Navedo, L. Marqués, L. Aguado
Corresponding author
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" ]
| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 15 | 11 | 26 |
| 2024 October | 105 | 53 | 158 |
| 2024 September | 138 | 31 | 169 |
| 2024 August | 166 | 94 | 260 |
| 2024 July | 129 | 42 | 171 |
| 2024 June | 152 | 45 | 197 |
| 2024 May | 142 | 48 | 190 |
| 2024 April | 125 | 36 | 161 |
| 2024 March | 129 | 48 | 177 |
| 2024 February | 105 | 47 | 152 |
| 2024 January | 116 | 43 | 159 |
| 2023 December | 119 | 56 | 175 |
| 2023 November | 121 | 62 | 183 |
| 2023 October | 104 | 33 | 137 |
| 2023 September | 102 | 65 | 167 |
| 2023 August | 85 | 38 | 123 |
| 2023 July | 106 | 68 | 174 |
| 2023 June | 80 | 40 | 120 |
| 2023 May | 111 | 61 | 172 |
| 2023 April | 120 | 56 | 176 |
| 2023 March | 106 | 58 | 164 |
| 2023 February | 84 | 45 | 129 |
| 2023 January | 67 | 43 | 110 |
| 2022 December | 77 | 49 | 126 |
| 2022 November | 33 | 59 | 92 |
| 2022 October | 41 | 36 | 77 |
| 2022 September | 45 | 64 | 109 |
| 2022 August | 30 | 64 | 94 |
| 2022 July | 41 | 55 | 96 |
| 2022 June | 29 | 49 | 78 |
| 2022 May | 60 | 48 | 108 |
| 2022 April | 71 | 70 | 141 |
| 2022 March | 60 | 64 | 124 |
| 2022 February | 53 | 44 | 97 |
| 2022 January | 55 | 81 | 136 |
| 2021 December | 56 | 67 | 123 |
| 2021 November | 78 | 60 | 138 |
| 2021 October | 81 | 84 | 165 |
| 2021 September | 81 | 76 | 157 |
| 2021 August | 78 | 63 | 141 |
| 2021 July | 98 | 50 | 148 |
| 2021 June | 69 | 49 | 118 |
| 2021 May | 53 | 45 | 98 |
| 2021 April | 120 | 54 | 174 |
| 2021 March | 87 | 62 | 149 |
| 2021 February | 68 | 40 | 108 |
| 2021 January | 55 | 30 | 85 |
| 2020 December | 48 | 27 | 75 |
| 2020 November | 25 | 18 | 43 |
| 2020 October | 30 | 41 | 71 |
| 2020 September | 37 | 40 | 77 |
| 2020 August | 25 | 30 | 55 |
| 2020 July | 27 | 26 | 53 |
| 2020 June | 46 | 35 | 81 |
| 2020 May | 36 | 50 | 86 |
| 2020 April | 38 | 37 | 75 |
| 2020 March | 35 | 24 | 59 |
| 2020 February | 10 | 19 | 29 |
| 2020 January | 2 | 26 | 28 |
| 2019 December | 8 | 17 | 25 |
| 2019 November | 2 | 2 | 4 |
| 2019 October | 4 | 11 | 15 |
| 2019 September | 8 | 8 | 16 |
| 2019 August | 4 | 8 | 12 |
| 2019 July | 4 | 8 | 12 |
| 2019 June | 6 | 10 | 16 |
| 2019 May | 4 | 49 | 53 |
| 2019 April | 3 | 1 | 4 |
| 2019 March | 2 | 7 | 9 |
| 2019 February | 4 | 0 | 4 |
| 2018 December | 2 | 0 | 2 |
| 2018 November | 1 | 0 | 1 |
| 2018 October | 3 | 2 | 5 |
| 2018 September | 2 | 1 | 3 |
| 2018 August | 0 | 5 | 5 |
| 2018 July | 0 | 3 | 3 |
| 2018 June | 0 | 2 | 2 |
| 2018 May | 0 | 6 | 6 |
| 2018 April | 0 | 1 | 1 |
| 2018 March | 16 | 11 | 27 |
| 2018 February | 105 | 9 | 114 |
| 2018 January | 131 | 8 | 139 |
| 2017 December | 109 | 10 | 119 |
| 2017 November | 84 | 4 | 88 |
| 2017 October | 59 | 10 | 69 |
| 2017 September | 72 | 12 | 84 |
| 2017 August | 80 | 7 | 87 |
| 2017 July | 54 | 18 | 72 |
| 2017 June | 73 | 29 | 102 |
| 2017 May | 75 | 23 | 98 |
| 2017 April | 63 | 10 | 73 |
| 2017 March | 78 | 22 | 100 |
| 2017 February | 69 | 17 | 86 |
| 2017 January | 72 | 13 | 85 |
| 2016 December | 71 | 9 | 80 |
| 2016 November | 131 | 13 | 144 |
| 2016 October | 192 | 28 | 220 |
| 2016 September | 245 | 11 | 256 |
| 2016 August | 195 | 4 | 199 |
| 2016 July | 87 | 9 | 96 |
| 2016 June | 12 | 9 | 21 |
| 2016 May | 11 | 6 | 17 |
| 2016 April | 5 | 3 | 8 |
| 2016 March | 3 | 22 | 25 |
| 2016 February | 6 | 1 | 7 |
| 2016 January | 10 | 4 | 14 |
| 2015 December | 11 | 17 | 28 |
| 2015 November | 19 | 2 | 21 |
| 2015 October | 16 | 15 | 31 |
| 2015 September | 5 | 3 | 8 |
| 2015 August | 9 | 8 | 17 |
| 2015 July | 138 | 8 | 146 |
| 2015 June | 61 | 3 | 64 |
| 2015 May | 71 | 4 | 75 |
| 2015 April | 48 | 12 | 60 |
| 2015 March | 44 | 3 | 47 |
| 2015 February | 56 | 6 | 62 |
| 2015 January | 50 | 1 | 51 |
| 2014 December | 40 | 6 | 46 |
| 2014 November | 46 | 6 | 52 |
| 2014 October | 53 | 7 | 60 |
| 2014 September | 48 | 2 | 50 |
| 2014 August | 39 | 3 | 42 |
| 2014 July | 44 | 5 | 49 |
| 2014 June | 61 | 5 | 66 |
| 2014 May | 59 | 5 | 64 |
| 2014 April | 38 | 6 | 44 |
| 2014 March | 53 | 6 | 59 |
| 2014 February | 41 | 4 | 45 |
| 2014 January | 32 | 2 | 34 |
| 2013 December | 16 | 3 | 19 |
| 2013 November | 1 | 4 | 5 |
| 2013 October | 2 | 3 | 5 |
| 2013 September | 2 | 0 | 2 |
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