Images in Clinical Rheumatology
Extensive Calcinosis in Adult Dermatomyositis
Calcinosis extensa en dermatomiositis del adulto
Diego Federico Baenas
, Juan Pablo Pirola, Nadia Raquel Benzaquén, Francisco Caeiro, Ana Cecilia Álvarez, Verónica Saurit, Soledad Retamozo, Alejandro Alvarellos
Corresponding author
Servicio de Reumatología, Hospital Privado Universitario de Córdoba, Córdoba, Argentina
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array:4 [ "nombre" => "Diego Federico" "apellidos" => "Baenas" "email" => array:1 [ 0 => "baenashospitalprivado@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "Juan Pablo" "apellidos" => "Pirola" ] 2 => array:2 [ "nombre" => "Nadia Raquel" "apellidos" => "Benzaquén" ] 3 => array:2 [ "nombre" => "Francisco" "apellidos" => "Caeiro" ] 4 => array:2 [ "nombre" => "Ana Cecilia" "apellidos" => "Álvarez" ] 5 => array:2 [ "nombre" => "Verónica" "apellidos" => "Saurit" ] 6 => array:2 [ "nombre" => "Soledad" "apellidos" => "Retamozo" ] 7 => array:2 [ "nombre" => "Alejandro" "apellidos" => "Alvarellos" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Servicio de Reumatología, Hospital Privado Universitario de Córdoba, Córdoba, Argentina" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Calcinosis extensa en dermatomiositis del adulto" ] ] "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" => 2556 "Ancho" => 1400 "Tamanyo" => 239010 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Extensive lineal calcifications of reticular appearance in dermis and hypodermis: (A, B) upper limbs; (C–F) lower limbs.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">A female aged 48 who presented at the surgery with photosensitivity, heliotrope erythema and cervical and upper trunk level erythematous injuries, in addition to myalgias and proximal muscle weakness, 3/5 in upper and lower limbs, and weakness of the flexor tendons of the neck. Laboratory tests revealed erythrocyte sedimentation of 54<span class="elsevierStyleHsp" style=""></span>mm/h, CPK 9.870<span class="elsevierStyleHsp" style=""></span>U/l, significant elevation of transaminase levels and lactate deshydrogenase levels, and ANA 1:1.280 mottled pattern. Magnetic resonance of the thighs showed exaggerated inflammatory changes. Electromyography had potentials of polyphasic motor units of short duration and low amplitude, mainly at gluteus level. Muscular biopsy was compatible with dermatomyositis. Treatment was initiated with 40<span class="elsevierStyleHsp" style=""></span>mg/day prednisone, 20<span class="elsevierStyleHsp" style=""></span>mg/week methotrexate, 5<span class="elsevierStyleHsp" style=""></span>mg/week folic acid, 200<span class="elsevierStyleHsp" style=""></span>mg/12<span class="elsevierStyleHsp" style=""></span>h hydroxychloroquine and muscular physiotherapy. Twenty days later, the patient presented with progression of weakness of 2/5 in lower limbs and 1/5 in upper limbs and was incapacitated and prostrate. Three pulsatile administrations of methylprednisone, cyclophosphamide and gammaglobulins were given. Treatment continued with pulsatile administrations of cyclophosphamide and she received infusions of 1<span class="elsevierStyleHsp" style=""></span>g/kg/2 days in three consecutive stays every 30 days. Improvement occurred with intensive treatment and rehabilitation, with the patient being able to stand up and to walk after 3 months of treatment. There was an improvement in her extensively compromised cutaneous barrier after initiating treatment with 1<span class="elsevierStyleHsp" style=""></span>g rituximab. Cutaneous calcifications progressed in triceps, gluteus, thighs and legs despite treatment with gammaglobulins, rituximab, pamidronate and colchincine (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">Calcinosis is a common finding in inflammatory myopathies, especially in juvenile dermatomyositis (20%–70%), but it is infrequent when the disease presents in the adult (20%).<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> It is associated with the disease activity and progression, a highly compromised cutaneous barrier and a delay or lack of response to treatment.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> It is more common in regions of persistent stress and trauma. Its pathogenesis is unclear.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> Several recent reports associate this complication with the presence of anti-NXP2 antibodies in disseminated form and with anti-PM/Scl<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,3,4</span></a> forms.</p><p id="par0015" class="elsevierStylePara elsevierViewall">This complication is difficult to treat. Several strategies which have demonstrated improvement in case reports are: warfarin (not currently recommended due to risk of bleeding), thalidomide, diltiazem or colchincine (with tolerance-dependence dose adjustment), biphosphonates (cyclical infusions of amidronate or alendronate with oral administration of maintenance treatment), inmunoglobulins, and biologic agents, including infliximab and abatacept.<a class="elsevierStyleCrossRefs" href="#bib0075"><span class="elsevierStyleSup">5–10</span></a> Some recent case reports suggest that rituximab could be a favourable therapeutic option in severe and refractory cases.<a class="elsevierStyleCrossRefs" href="#bib0075"><span class="elsevierStyleSup">5,6</span></a> Surgery is reserved for discrete lesions due to the risk of infection.<a class="elsevierStyleCrossRefs" href="#bib0080"><span class="elsevierStyleSup">6–10</span></a></p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Ethical Liabilities</span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Protection of people and animals</span><p id="par0020" class="elsevierStylePara elsevierViewall">The authors declare that no experiments using human beings or animals have been carried out for this research study.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Data confidentiality</span><p id="par0025" class="elsevierStylePara elsevierViewall">The authors declare they have followed the protocols of their centre of work on patient data publication.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Right to privacy and informed consent</span><p id="par0030" class="elsevierStylePara elsevierViewall">The authors declare that no patient data appear in this article.</p></span></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Conflicts of Interest</span><p id="par0035" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:3 [ 0 => array:3 [ "identificador" => "sec0005" "titulo" => "Ethical Liabilities" "secciones" => array:3 [ 0 => array:2 [ "identificador" => "sec0010" "titulo" => "Protection of people and animals" ] 1 => array:2 [ "identificador" => "sec0015" "titulo" => "Data confidentiality" ] 2 => array:2 [ "identificador" => "sec0020" "titulo" => "Right to privacy and informed consent" ] ] ] 1 => array:2 [ "identificador" => "sec0025" "titulo" => "Conflicts of Interest" ] 2 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2017-01-03" "fechaAceptado" => "2017-03-03" "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Baenas DF, Pirola JP, Benzaquén NR, Caeiro F, Álvarez AC, Saurit V, et al. Calcinosis extensa en dermatomiositis del adulto. Reumatol Clin. 2019;15:121–123.</p>" ] ] "multimedia" => array:1 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 2556 "Ancho" => 1400 "Tamanyo" => 239010 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Extensive lineal calcifications of reticular appearance in dermis and hypodermis: (A, B) upper limbs; (C–F) lower limbs.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0055" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Identification of clinical features and autoantibodies associated with calcinosis in dermatomyositis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "A. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 October | 66 | 25 | 91 |
| 2024 September | 37 | 16 | 53 |
| 2024 August | 48 | 29 | 77 |
| 2024 July | 46 | 19 | 65 |
| 2024 June | 59 | 41 | 100 |
| 2024 May | 64 | 19 | 83 |
| 2024 April | 57 | 25 | 82 |
| 2024 March | 72 | 24 | 96 |
| 2024 February | 51 | 35 | 86 |
| 2024 January | 54 | 17 | 71 |
| 2023 December | 36 | 25 | 61 |
| 2023 November | 60 | 18 | 78 |
| 2023 October | 39 | 26 | 65 |
| 2023 September | 89 | 29 | 118 |
| 2023 August | 48 | 20 | 68 |
| 2023 July | 41 | 16 | 57 |
| 2023 June | 47 | 24 | 71 |
| 2023 May | 59 | 20 | 79 |
| 2023 April | 46 | 6 | 52 |
| 2023 March | 71 | 21 | 92 |
| 2023 February | 56 | 26 | 82 |
| 2023 January | 30 | 11 | 41 |
| 2022 December | 53 | 24 | 77 |
| 2022 November | 58 | 25 | 83 |
| 2022 October | 53 | 29 | 82 |
| 2022 September | 50 | 21 | 71 |
| 2022 August | 32 | 40 | 72 |
| 2022 July | 43 | 40 | 83 |
| 2022 June | 45 | 22 | 67 |
| 2022 May | 62 | 42 | 104 |
| 2022 April | 60 | 35 | 95 |
| 2022 March | 51 | 45 | 96 |
| 2022 February | 58 | 34 | 92 |
| 2022 January | 74 | 39 | 113 |
| 2021 December | 67 | 44 | 111 |
| 2021 November | 44 | 32 | 76 |
| 2021 October | 54 | 44 | 98 |
| 2021 September | 41 | 36 | 77 |
| 2021 August | 57 | 27 | 84 |
| 2021 July | 25 | 34 | 59 |
| 2021 June | 44 | 27 | 71 |
| 2021 May | 46 | 46 | 92 |
| 2021 April | 82 | 93 | 175 |
| 2021 March | 46 | 26 | 72 |
| 2021 February | 42 | 16 | 58 |
| 2021 January | 38 | 25 | 63 |
| 2020 December | 43 | 10 | 53 |
| 2020 November | 41 | 25 | 66 |
| 2020 October | 24 | 19 | 43 |
| 2020 September | 58 | 21 | 79 |
| 2020 August | 31 | 20 | 51 |
| 2020 July | 21 | 21 | 42 |
| 2020 June | 36 | 14 | 50 |
| 2020 May | 33 | 20 | 53 |
| 2020 April | 26 | 13 | 39 |
| 2020 March | 20 | 10 | 30 |
| 2019 July | 1 | 2 | 3 |
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