Case and Research Letters
Facial Calcinosis Cutis in a Woman with Paraneoplastic Dermatomyositis
Calcinosis cutis facial en una paciente con dermatomiositis paraneoplásica
M. Herrero-Moyano
, C. Martínez-Mera, C. García-García, E. Dauden
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Servicio de Dermatología, Hospital Universitario de La Princesa, Madrid, España
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Herrero-Moyano, C. Martínez-Mera, C. García-García, E. Dauden" "autores" => array:4 [ 0 => array:2 [ "nombre" => "M." "apellidos" => "Herrero-Moyano" ] 1 => array:2 [ "nombre" => "C." "apellidos" => "Martínez-Mera" ] 2 => array:2 [ "nombre" => "C." "apellidos" => "García-García" ] 3 => array:2 [ "nombre" => "E." 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En la epidermis, se observó un subgrupo de linfocitos atípicos negativos para CD4 y CD8. Hay pérdida de expresión de CD7. Pocas células fueron reactivas al marcador CD30. La mayoría de los linfocitos dieron positivo para el receptor de la cadena beta de linfocitos T (BF1) y negativo para el receptor de la cadena gamma de linfocitos T. A) Tinción hematoxilina y eosina, 40x. B) Tinción hematoxilina y eosina, 200x C-F) inmunohistoquímica, 200x.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "D.J. Lewis, T. Hinojosa, W.H. Chan, J.H. Wu, M. Duvic" "autores" => array:5 [ 0 => array:2 [ "nombre" => "D.J." "apellidos" => "Lewis" ] 1 => array:2 [ "nombre" => "T." "apellidos" => "Hinojosa" ] 2 => array:2 [ "nombre" => "W.H." "apellidos" => "Chan" ] 3 => array:2 [ "nombre" => "J.H." "apellidos" => "Wu" ] 4 => array:2 [ "nombre" => "M." 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Herrero-Moyano, C. Martínez-Mera, C. García-García, E. Dauden" "autores" => array:4 [ 0 => array:4 [ "nombre" => "M." "apellidos" => "Herrero-Moyano" "email" => array:1 [ 0 => "mariaherreromoyano@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "C." "apellidos" => "Martínez-Mera" ] 2 => array:2 [ "nombre" => "C." "apellidos" => "García-García" ] 3 => array:2 [ "nombre" => "E." "apellidos" => "Dauden" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Servicio de Dermatología, Hospital Universitario de La Princesa, Madrid, España" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Calcinosis cutis facial en una paciente con dermatomiositis paraneoplásica" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 568 "Ancho" => 750 "Tamanyo" => 36445 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Skin ultrasound (M-mode, 18<span class="elsevierStyleHsp" style=""></span>MHz) image showing hyperechoic lesions (yellow arrows) with posterior acoustic shadow in the subcutaneous cellular tissue compatible with calcinosis.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Calcinosis is an infrequent complication of adult dermatomyositis (DM) and a very rare complication of paraneoplastic DM that predominantly affects areas of the limbs that are frequently exposed to pressure. Descriptions of facial calcinosis are very scarce.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> We describe the case of a patient with paraneoplastic DM and calcinosis cutis of the face.</p><p id="par0010" class="elsevierStylePara elsevierViewall">The patient was a 50-year-old woman who was being followed for DM, which had been diagnosed 8 years earlier. She had extensive DM skin lesions on the face, trunk, and limbs, and electromyography and muscle biopsy revealed involvement of the scapular and pelvic girdles. The results of the antinuclear antibody test were positive. An autoimmunity test for DM revealed positivity for anti-SAE1 (small ubiquitin-like modifier activating enzyme 1) antibody. Treatment with systemic corticosteroids, azathioprine, and methotrexate controlled the myositis but not the skin lesions, which worsened and progressed to necrotic lesions in the lumbar and gluteal regions. Mammography revealed an increase in focal density in the right retroareolar region that was difficult to evaluate; a breast biopsy was not performed until microcalcifications appeared on the breast 3 years after the onset of DM. At this point, the patient was diagnosed with grade III invasive ductal breast carcinoma. Complete remission was achieved following surgery and chemotherapy. The patient's skin condition markedly improved after breast cancer treatment. The only persisting lesions were mild poikilodermal lesions on the face and trunk that did not require immunosuppressive therapy. About 4 years after diagnosis of DM, the patient developed painful, indurated nodules, some of which were ulcerated, first on the arm and subsequently on the biopsy scar, both malar regions (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>), and the forearms, legs, and gluteal region. Radiography confirmed the presence of calcium in these locations. Craniofacial computed tomography showed thickening of the periorbital soft tissue and the presence of subcutaneous calcifications in both malar regions and the left preauricular region (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). Skin ultrasound showed thickening of the dermis and the presence in the subcutaneous cellular tissue of hyperechoic deposits that produced posterior acoustic shadow, with no increase in vascularization evident on Doppler ultrasound (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). Analysis of phosphocalcic metabolism revealed no findings of interest. At the time, the patient was being treated with diltiazem, colchicine, and alendronic acid (70<span class="elsevierStyleHsp" style=""></span>mg/wk), which prevented progression of calcification. Several flare-ups of skin lesions, without myositis, were effectively controlled with methotrexate and cycles of intravenous immunoglobulin. The patient's breast cancer remains in complete remission and no new tumors have been detected.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Dystrophic calcinosis cutis is a relatively common complication of juvenile DM (45%–75% of cases), but is uncommon in adult DM (19%–20% of cases).<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> It usually appears in patients with intense DM skin lesions, as in our patient. It is extremely rare in patients with paraneoplastic DM. No cases of calcinosis were reported in one of the largest published series of patients (N<span class="elsevierStyleHsp" style=""></span>=<span class="elsevierStyleHsp" style=""></span>12) with paraneoplastic DM.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> We have found reports of this association only in isolated cases in the literature.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">According to Callen and Wortmann,<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a> the course of paraneoplastic DM may not be dependent on the underlying tumor, but usually is. Our patient's DM did not fully resolve after treatment of the underlying tumor. However, the change in the intensity of the clinical picture and the response to treatment observed after remission of the patient's breast cancer suggest a link between DM and the tumor, which may act as a trigger of DM in predisposed individuals. Cutaneous necrosis, which was observed in our patient, is another clinical feature of DM associated with an underlying tumor.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">In DM patients, calcinosis cutis usually affects proximal parts of the limbs, muscles, and large joints.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> During the course of our patient's condition she developed calcinosis lesions on the face, in addition to the aforementioned classic locations. Isolated cases of facial calcinosis have been described in patients with systemic scleroderma,<a class="elsevierStyleCrossRefs" href="#bib0075"><span class="elsevierStyleSup">5,6</span></a> antisynthetase syndrome,<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a> and inflammatory acne,<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a> and calcinosis of the scalp has been described in 5 cases of juvenile DM.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> However, we have found no reports of facial calcinosis in cases of adult DM or paraneoplastic DM.</p><p id="par0030" class="elsevierStylePara elsevierViewall">While anti-SAE1 antibody positivity has been associated with severe skin diseases and a higher incidence of associated cancer,<a class="elsevierStyleCrossRefs" href="#bib0095"><span class="elsevierStyleSup">9,10</span></a> no association between anti-SAE1 positivity and the development of calcinosis has been demonstrated.</p><p id="par0035" class="elsevierStylePara elsevierViewall">In conclusion, we describe the case of a woman with paraneoplastic DM who developed dystrophic calcinosis cutis in the classic locations and on the face, a location rarely affected by this clinical form.</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: Herrero-Moyano M, Martínez-Mera C, García-García C, Dauden E. Calcinosis cutis facial en una paciente con dermatomiositis paraneoplásica. Actas Dermosifiliogr. 2018;109:760–762.</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" => 563 "Ancho" => 750 "Tamanyo" => 80273 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Stony-hard plaques in both malar regions of about 3<span class="elsevierStyleHsp" style=""></span>cm in diameter with well-defined borders.</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" => 646 "Ancho" => 1250 "Tamanyo" => 67398 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Computed tomography images acquired in coronal (A) and transverse (B) planes showing hyperintense lesions with calcium accumulation (yellow arrows) in both malar regions.</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" => 568 "Ancho" => 750 "Tamanyo" => 36445 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Skin ultrasound (M-mode, 18<span class="elsevierStyleHsp" style=""></span>MHz) image showing hyperechoic lesions (yellow arrows) with posterior acoustic shadow in the subcutaneous cellular tissue compatible with calcinosis.</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" => "Calcinosis cutis in autoimmune connective tissue diseases" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "A. 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| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 5 | 12 | 17 |
| 2024 Octubre | 102 | 44 | 146 |
| 2024 Septiembre | 121 | 41 | 162 |
| 2024 Agosto | 129 | 85 | 214 |
| 2024 Julio | 110 | 70 | 180 |
| 2024 Junio | 118 | 62 | 180 |
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| 2024 Marzo | 123 | 40 | 163 |
| 2024 Febrero | 128 | 45 | 173 |
| 2024 Enero | 157 | 36 | 193 |
| 2023 Diciembre | 165 | 27 | 192 |
| 2023 Noviembre | 239 | 31 | 270 |
| 2023 Octubre | 175 | 38 | 213 |
| 2023 Septiembre | 162 | 45 | 207 |
| 2023 Agosto | 108 | 22 | 130 |
| 2023 Julio | 163 | 40 | 203 |
| 2023 Junio | 125 | 31 | 156 |
| 2023 Mayo | 154 | 33 | 187 |
| 2023 Abril | 152 | 23 | 175 |
| 2023 Marzo | 188 | 37 | 225 |
| 2023 Febrero | 133 | 33 | 166 |
| 2023 Enero | 139 | 43 | 182 |
| 2022 Diciembre | 143 | 56 | 199 |
| 2022 Noviembre | 77 | 35 | 112 |
| 2022 Octubre | 115 | 38 | 153 |
| 2022 Septiembre | 93 | 44 | 137 |
| 2022 Agosto | 82 | 42 | 124 |
| 2022 Julio | 46 | 43 | 89 |
| 2022 Junio | 36 | 45 | 81 |
| 2022 Mayo | 150 | 48 | 198 |
| 2022 Abril | 176 | 32 | 208 |
| 2022 Marzo | 155 | 54 | 209 |
| 2022 Febrero | 138 | 31 | 169 |
| 2022 Enero | 187 | 35 | 222 |
| 2021 Diciembre | 106 | 40 | 146 |
| 2021 Noviembre | 86 | 44 | 130 |
| 2021 Octubre | 66 | 61 | 127 |
| 2021 Septiembre | 72 | 42 | 114 |
| 2021 Agosto | 76 | 33 | 109 |
| 2021 Julio | 91 | 36 | 127 |
| 2021 Junio | 89 | 19 | 108 |
| 2021 Mayo | 81 | 40 | 121 |
| 2021 Abril | 200 | 73 | 273 |
| 2021 Marzo | 95 | 32 | 127 |
| 2021 Febrero | 97 | 28 | 125 |
| 2021 Enero | 47 | 17 | 64 |
| 2020 Diciembre | 48 | 15 | 63 |
| 2020 Noviembre | 40 | 27 | 67 |
| 2020 Octubre | 32 | 17 | 49 |
| 2020 Septiembre | 38 | 16 | 54 |
| 2020 Agosto | 33 | 21 | 54 |
| 2020 Julio | 15 | 11 | 26 |
| 2020 Junio | 26 | 34 | 60 |
| 2020 Mayo | 15 | 18 | 33 |
| 2020 Abril | 17 | 16 | 33 |
| 2020 Marzo | 13 | 10 | 23 |
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