Case and Research Letters
Good Response to Tofacitinib in Refractory Amyopathic Dermatomyositis
Dermatomiositis amiopática refractaria con buena respuesta a tofacitinib
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Crespo Cruz, J. del Boz, C. Romero Gómez" "autores" => array:3 [ 0 => array:3 [ "nombre" => "A." "apellidos" => "Crespo Cruz" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 1 => array:4 [ "nombre" => "J." "apellidos" => "del Boz" "email" => array:1 [ 0 => "javierdelboz@yahoo.es" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] 1 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 2 => array:3 [ "nombre" => "C." "apellidos" => "Romero Gómez" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] ] ] "afiliaciones" => array:3 [ 0 => array:3 [ "entidad" => "Servicio de Dermatología, Hospital Universitario Nuestra Señora de Valme, Sevilla, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Dermatología, Hospital Costa del Sol, Marbella, Málaga, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Servicio de Medicina Interna, Hospital Costa del Sol, Marbella, Málaga, Spain" "etiqueta" => "c" "identificador" => "aff0015" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Dermatomiositis amiopática refractaria con buena respuesta a tofacitinib" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1125 "Ancho" => 1500 "Tamanyo" => 262423 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Skin lesions prior to beginning tofacitinib therapy. A, Gottron papules, periungual erythema, and hemorrhagic crusting on the hands. B, Facial erythema and scaling and heliotrope erythema of the eyelids. C, V sign on the chest.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Dermatomyositis (DM) is an idiopathic inflammatory myopathy characterized by muscle inflammation, skin involvement, and systemic manifestations that are often a very broad spectrum in nature.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Amyopathic DM, also known as DM sine myositis, affects a subset of DM patients and presents with cutaneous symptoms but without myopathy.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">Our patient was a 49-year-old white woman with a history of thalassemia, for which she had been in follow-up for 7 years. She presented with asthenia, itching, and skin lesions located predominantly in photo-exposed areas. Physical examination revealed heliotrope erythema, Gottron papules, Gottron sign, shawl sign, V sign, and Holster sign. A detailed medical history was taken and a systemic review performed, as well as multiple additional tests, including nuclear magnetic resonance imaging of the pelvic girdle, an electromyogram, a muscle biopsy, and determination of muscle enzymes. All results were normal, and the patient was diagnosed with amyopathic DM. Screening for antinuclear, anti-synthetase, anti-TIF-1γ (transcription intermediary factor 1 γ), and anti-MDA-5 (melanoma differentiation-associated protein 5) antibodies and occult tumors was negative throughout the follow-up period.</p><p id="par0015" class="elsevierStylePara elsevierViewall">Owing to poor disease control, the patient had undergone multiple treatments to which she responded poorly, including topical and oral corticosteroids (maximum dose, 1 mg/kg/d), azathioprine, methotrexate, hydroxychloroquine, mepacrine, rituximab, oral tacrolimus, intravenous immunoglobulins, and mycophenolate mofetil. She was being simultaneously treated with oral prednisone, hydroxychloroquine, mepacrine, and mycophenolate mofetil up until 7 months before the consultation. Given the progressive worsening of her condition and the severity of the skin lesions (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>) and pruritus, only treatment with low-dose prednisone (2.5 mg/d) was maintained, and tofacitinib (TOF) treatment (5 mg/12 h) was started.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Clinical improvement was observed 2 weeks later and, at the time of writing, all skin lesions have improved significantly (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>) and the pruritus has disappeared.</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">DM is a difficult-to-manage multisystemic disease for which no standardized treatment algorithms have been developed owing to the scarcity of well-designed clinical trials. Furthermore, it is not uncommon for DM to overlap with other autoimmune diseases, posing both diagnostic and therapeutic challenges to physicians.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">TOF is an inhibitor of the Janus kinases JAK3 and JAK1 and attenuates signal transduction activated by interleukin (IL) -2, IL-4, IL-6, IL-7, IL-9, IL-15, IL-21, and type I and II interferons. Because the interferon signaling pathway is overexpressed in DM, TOF may be useful in the treatment of this disease.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,4</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Kurtzman et al<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> reported promising results in 3 patients with recalcitrant cutaneous DM who were treated with TOF. Subsequently, Paik and Christopher-Stine<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> reported a good response to TOF treatment in an adult DM patient who had failed to respond to multiple therapies. In a review by Cinats et al<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> of emerging uses of JAK inhibitors in skin diseases, including psoriasis, atopic dermatitis, alopecia areata, vitiligo, DM, and graft versus host disease, adverse effects reported included infections in TOF-treated patients and anemia and thrombocytopenia in those treated with ruxolitinib.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">In the last year, clinical cases have been published demonstrating a therapeutic effect of TOF not only in skin manifestations, but also interstitial lung disease and severe refractory calcinosis, with no notable side effects<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,3,7–9</span></a> except for transient hypercalcemia and increased body mass index in 2 patients.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">The present case is the sixth published description of amyopathic DM treated with TOF. As in the 5 previously published cases,<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,5,7</span></a> the patient improved rapidly and treatment was well tolerated.</p><p id="par0050" class="elsevierStylePara elsevierViewall">Clinical trials are required to reliably confirm the efficacy and safety profile of this treatment. A clinical trial is currently underway to evaluate the efficacy of TOF treatment of treatment-refractory DM.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a></p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of Interest</span><p id="par0055" 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: Crespo Cruz A, del Boz J, Romero Gómez C. Dermatomiositis amiopática refractaria con buena respuesta a tofacitinib. Actas Dermosifiliogr. 2021;112:374–376.</p>" ] ] "multimedia" => array:2 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1125 "Ancho" => 1500 "Tamanyo" => 262423 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Skin lesions prior to beginning tofacitinib therapy. A, Gottron papules, periungual erythema, and hemorrhagic crusting on the hands. B, Facial erythema and scaling and heliotrope erythema of the eyelids. C, V sign on the chest.</p>" ] ] 1 => array:8 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1125 "Ancho" => 1500 "Tamanyo" => 216258 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Skin lesions after 7 months of tofacitinib therapy. A, Flattening of Gottron papules and disappearance of periungual erythema. B, Discrete heliotrope erythema. C, Poikiloderma on the chest with no evidence of V sign.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Successful treatment of extensive calcifications and acute pulmonary involvement in dermatomyositis with the Janus-Kinase inhibitor tofacitinib - A report of two cases" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "S. Wendel" 1 => "N. Venhoff" 2 => "B.C. Frye" 3 => "A.M. May" 4 => "P. Agarwal" 5 => "M. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 15 | 9 | 24 |
| 2024 October | 87 | 80 | 167 |
| 2024 September | 83 | 52 | 135 |
| 2024 August | 121 | 70 | 191 |
| 2024 July | 97 | 45 | 142 |
| 2024 June | 110 | 47 | 157 |
| 2024 May | 87 | 35 | 122 |
| 2024 April | 79 | 34 | 113 |
| 2024 March | 69 | 29 | 98 |
| 2024 February | 66 | 36 | 102 |
| 2024 January | 89 | 36 | 125 |
| 2023 December | 70 | 21 | 91 |
| 2023 November | 92 | 56 | 148 |
| 2023 October | 83 | 24 | 107 |
| 2023 September | 78 | 36 | 114 |
| 2023 August | 51 | 17 | 68 |
| 2023 July | 82 | 35 | 117 |
| 2023 June | 63 | 25 | 88 |
| 2023 May | 126 | 32 | 158 |
| 2023 April | 70 | 28 | 98 |
| 2023 March | 63 | 43 | 106 |
| 2023 February | 86 | 32 | 118 |
| 2023 January | 66 | 33 | 99 |
| 2022 December | 72 | 57 | 129 |
| 2022 November | 38 | 38 | 76 |
| 2022 October | 39 | 34 | 73 |
| 2022 September | 36 | 53 | 89 |
| 2022 August | 32 | 54 | 86 |
| 2022 July | 36 | 50 | 86 |
| 2022 June | 37 | 34 | 71 |
| 2022 May | 95 | 48 | 143 |
| 2022 April | 137 | 37 | 174 |
| 2022 March | 111 | 77 | 188 |
| 2022 February | 105 | 55 | 160 |
| 2022 January | 141 | 64 | 205 |
| 2021 December | 103 | 62 | 165 |
| 2021 November | 95 | 69 | 164 |
| 2021 October | 90 | 81 | 171 |
| 2021 September | 87 | 63 | 150 |
| 2021 August | 71 | 54 | 125 |
| 2021 July | 68 | 34 | 102 |
| 2021 June | 82 | 37 | 119 |
| 2021 May | 100 | 82 | 182 |
| 2021 April | 238 | 159 | 397 |
| 2021 March | 59 | 43 | 102 |
| 2021 February | 56 | 33 | 89 |
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