Case and Research Letters
Widespread Cutaneous Necrosis as the First Clinical Manifestation of Secondary Antiphospholipid Syndrome
Necrosis cutánea extensa como la primera manifestación clínica de un síndrome antifosfolípidico
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Perelló-Alzamora, J.C. Santos-Duran, A. Santos-Briz, P. Unamuno" "autores" => array:4 [ 0 => array:4 [ "nombre" => "M.R." "apellidos" => "Perelló-Alzamora" "email" => array:1 [ 0 => "mariarosaperello@yahoo.es" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">¿</span>" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "J.C." "apellidos" => "Santos-Duran" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "A." "apellidos" => "Santos-Briz" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "P." "apellidos" => "Unamuno" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Department of Dermatology, University Hospital of Salamanca, Salamanca, Spain" "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Department of Pathology, University Hospital of Salamanca, Salamanca, Spain" "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Necrosis cutánea extensa como la primera manifestación clínica de un síndrome antifosfolípidico" ] ] "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" => 715 "Ancho" => 950 "Tamanyo" => 84175 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Cutaneous lesions on admission. Bullous erythematous violaceous plaques on the right leg.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Antiphospholipid syndrome (APS) is an acquired prothrombotic state characterized by recurrent thromboses, pregnancy loss, thrombocytopenia, and the presence of antiphospholipid antibodies, anticardiolipin antibodies, lupus anticoagulant and/or anti-β2-glycoprotein antibody.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1–3</span></a> The original clinical and laboratory criteria for APS, called the Sapporo criteria, were first published in 1999.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> These were replaced by the Sydney criteria in 2006,<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> when patients were required to have at least 1 clinical criterion and 1 laboratory criterion for a diagnosis of APS to be made.</p><p id="par0010" class="elsevierStylePara elsevierViewall">A wide variety of dermatologic manifestations have been described in patients with APS,<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> including livedo reticularis, livedoid vasculitis, digital gangrene, erythematous macules,<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> skin ulcerations, and, on rare occasions, extensive cutaneous necrosis.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> We report the case of a patient with widespread cutaneous necrosis as the initial manifestation of APS.</p><p id="par0015" class="elsevierStylePara elsevierViewall">A 48-year-old woman presented at the emergency room with a 2-week history of fever, arthralgia, malaise, and chest pain. On examination she was noted to have 3 bullous erythematous violaceous plaques on her right leg (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). Her medical history was remarkable for hypothyroidism, systemic lupus erythematosus (SLE), and obsessive-compulsive disorder. Medications included thyroxine, olanzapine, risperidone, and sertraline. Laboratory tests revealed thrombocytopenia (platelet count 25,000) and normal prothrombin and partial thromboplastin times. Lupus anticoagulant was present and the antinuclear antibody titer was 1:320. C3 and C4 levels were normal.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">The lesions deteriorated rapidly despite initiation of corticosteroid therapy with prednisolone at a dosage of 1<span class="elsevierStyleHsp" style=""></span>mg/kg/d. The dermatology service was called to evaluate the patient on day 7 of hospitalization. Examination revealed extensive cutaneous necrosis on the right leg (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). Further laboratory data revealed an elevated anticardiolipin antibody immunoglobulin (Ig) G titer (30; normal <23). Anti-β2-glycoprotein antibody IgG levels were also elevated, but cryoglobulins and cryofibrinogens were normal. A 4-mm punch biopsy from the periphery of a lesion revealed hemorrhage throughout the dermis with organized thrombi in many dermal vessels, with no evidence of vasculitis (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). No further lesions developed after the addition of intravenous heparin. At discharge, the heparin was replaced by long-term oral anticoagulation therapy and prednisone was tapered over 8 weeks. At the 3-month follow-up, only residual scarring remained (<a class="elsevierStyleCrossRef" href="#fig0020">Fig. 4</a>). There have been no new thrombotic events in 1 year of follow-up.</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><elsevierMultimedia ident="fig0020"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">Only 23 cases of extensive cutaneous necrosis linked to APS have been reported in the literature.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,8</span></a> Most of the patients have been young women and the underlying diseases included SLE (9 cases), lupus-like disease (1 case), urinary tract infection (2 cases), acquired immunodeficiency syndrome (1 case), rheumatoid arthritis (1 case), mycosis fungoides (1 case), and mixed connective tissue disease (7 cases). Seven patients had no underlying disease. All of the patients developed thrombotic complications limited to skin, and, as in our patient, the lower limbs were the most commonly affected site. Skin biopsy revealed the presence of thrombi in dermal venules and capillaries, with no evidence of vasculitis.</p><p id="par0030" class="elsevierStylePara elsevierViewall">The mechanisms of thrombosis associated with antiphospholipid antibodies remain unknown.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> The main entities to take into consideration in the differential diagnosis are catastrophic antiphospholipid antibody syndrome and disseminated intravascular coagulation.</p><p id="par0035" class="elsevierStylePara elsevierViewall">Our patient achieved complete healing with prednisolone at a dose of 1<span class="elsevierStyleHsp" style=""></span>mg/kg/d and heparin, which was replaced by oral anticoagulants at discharge. At the time of writing, after 1 year of follow-up, there have been no further thrombotic episodes.</p><p id="par0040" class="elsevierStylePara elsevierViewall">We conclude that widespread cutaneous necrosis is a rare initial manifestation of APS and should be considered a major thrombotic event.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> It is important to recognize these lesions because early diagnosis enables early treatment, and, possibly, better prognosis.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "multimedia" => array:4 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 715 "Ancho" => 950 "Tamanyo" => 84175 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Cutaneous lesions on admission. Bullous erythematous violaceous plaques on the right leg.</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" => 714 "Ancho" => 950 "Tamanyo" => 118061 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Cutaneous lesions on day 7 of hospitalization. Extensive cutaneous necrosis on the right leg.</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" => 713 "Ancho" => 950 "Tamanyo" => 227776 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Skin biopsy. Extensive epidermal basal vasculopathy, hemorrhage throughout the dermis with organized thrombi in many dermal vessels and no evidence of vasculitis.</p>" ] ] 3 => array:7 [ "identificador" => "fig0020" "etiqueta" => "Figure 4" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr4.jpeg" "Alto" => 714 "Ancho" => 950 "Tamanyo" => 95056 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Cutaneous lesions after 3 months. Residual scarring visible on the leg.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:8 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Extensive cutaneous necrosis associated with anticardiolipin antibodies" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "S. Paira" 1 => "S. Roverano" 2 => "A. Zunino" 3 => "M.E. Oliva" 4 => "M.L. 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| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 5 | 12 | 17 |
| 2024 Octubre | 42 | 59 | 101 |
| 2024 Septiembre | 47 | 48 | 95 |
| 2024 Agosto | 57 | 67 | 124 |
| 2024 Julio | 41 | 62 | 103 |
| 2024 Junio | 66 | 50 | 116 |
| 2024 Mayo | 62 | 47 | 109 |
| 2024 Abril | 64 | 22 | 86 |
| 2024 Marzo | 70 | 28 | 98 |
| 2024 Febrero | 60 | 27 | 87 |
| 2024 Enero | 57 | 29 | 86 |
| 2023 Diciembre | 52 | 16 | 68 |
| 2023 Noviembre | 81 | 35 | 116 |
| 2023 Octubre | 58 | 33 | 91 |
| 2023 Septiembre | 71 | 31 | 102 |
| 2023 Agosto | 41 | 15 | 56 |
| 2023 Julio | 97 | 34 | 131 |
| 2023 Junio | 47 | 29 | 76 |
| 2023 Mayo | 24 | 13 | 37 |
| 2023 Abril | 34 | 25 | 59 |
| 2023 Marzo | 79 | 27 | 106 |
| 2023 Febrero | 48 | 34 | 82 |
| 2023 Enero | 34 | 31 | 65 |
| 2022 Diciembre | 52 | 40 | 92 |
| 2022 Noviembre | 51 | 29 | 80 |
| 2022 Octubre | 36 | 39 | 75 |
| 2022 Septiembre | 34 | 53 | 87 |
| 2022 Agosto | 43 | 41 | 84 |
| 2022 Julio | 36 | 45 | 81 |
| 2022 Junio | 32 | 26 | 58 |
| 2022 Mayo | 44 | 32 | 76 |
| 2022 Abril | 48 | 44 | 92 |
| 2022 Marzo | 36 | 54 | 90 |
| 2022 Febrero | 38 | 35 | 73 |
| 2022 Enero | 50 | 47 | 97 |
| 2021 Diciembre | 28 | 44 | 72 |
| 2021 Noviembre | 57 | 52 | 109 |
| 2021 Octubre | 61 | 54 | 115 |
| 2021 Septiembre | 36 | 44 | 80 |
| 2021 Agosto | 52 | 29 | 81 |
| 2021 Julio | 41 | 34 | 75 |
| 2021 Junio | 32 | 27 | 59 |
| 2021 Mayo | 39 | 30 | 69 |
| 2021 Abril | 66 | 43 | 109 |
| 2021 Marzo | 54 | 30 | 84 |
| 2021 Febrero | 43 | 23 | 66 |
| 2021 Enero | 64 | 22 | 86 |
| 2020 Diciembre | 34 | 9 | 43 |
| 2020 Noviembre | 15 | 18 | 33 |
| 2020 Octubre | 34 | 7 | 41 |
| 2020 Septiembre | 20 | 13 | 33 |
| 2020 Agosto | 29 | 20 | 49 |
| 2020 Julio | 55 | 13 | 68 |
| 2020 Junio | 34 | 22 | 56 |
| 2020 Mayo | 28 | 15 | 43 |
| 2020 Abril | 43 | 15 | 58 |
| 2020 Marzo | 33 | 30 | 63 |
| 2020 Febrero | 4 | 13 | 17 |
| 2020 Enero | 1 | 11 | 12 |
| 2019 Diciembre | 4 | 1 | 5 |
| 2019 Noviembre | 0 | 2 | 2 |
| 2019 Octubre | 0 | 1 | 1 |
| 2019 Septiembre | 4 | 0 | 4 |
| 2019 Julio | 0 | 5 | 5 |
| 2019 Junio | 2 | 6 | 8 |
| 2019 Mayo | 2 | 10 | 12 |
| 2019 Abril | 0 | 5 | 5 |
| 2019 Marzo | 3 | 7 | 10 |
| 2019 Febrero | 3 | 2 | 5 |
| 2019 Enero | 1 | 3 | 4 |
| 2018 Diciembre | 2 | 4 | 6 |
| 2018 Noviembre | 3 | 7 | 10 |
| 2018 Octubre | 3 | 1 | 4 |
| 2018 Septiembre | 3 | 0 | 3 |
| 2018 Abril | 1 | 0 | 1 |
| 2018 Marzo | 1 | 1 | 2 |
| 2018 Febrero | 31 | 1 | 32 |
| 2018 Enero | 36 | 5 | 41 |
| 2017 Diciembre | 51 | 8 | 59 |
| 2017 Noviembre | 40 | 3 | 43 |
| 2017 Octubre | 41 | 4 | 45 |
| 2017 Septiembre | 37 | 10 | 47 |
| 2017 Agosto | 27 | 9 | 36 |
| 2017 Julio | 41 | 5 | 46 |
| 2017 Junio | 57 | 15 | 72 |
| 2017 Mayo | 47 | 14 | 61 |
| 2017 Abril | 35 | 9 | 44 |
| 2017 Marzo | 30 | 8 | 38 |
| 2017 Febrero | 62 | 7 | 69 |
| 2017 Enero | 39 | 5 | 44 |
| 2016 Diciembre | 44 | 10 | 54 |
| 2016 Noviembre | 59 | 8 | 67 |
| 2016 Octubre | 66 | 9 | 75 |
| 2016 Septiembre | 100 | 12 | 112 |
| 2016 Agosto | 34 | 8 | 42 |
| 2016 Julio | 32 | 7 | 39 |
| 2016 Junio | 9 | 0 | 9 |
| 2016 Mayo | 7 | 9 | 16 |
| 2016 Abril | 9 | 2 | 11 |
| 2016 Marzo | 7 | 0 | 7 |
| 2016 Febrero | 4 | 7 | 11 |
| 2016 Enero | 4 | 41 | 45 |
| 2015 Diciembre | 6 | 0 | 6 |
| 2015 Noviembre | 20 | 18 | 38 |
| 2015 Octubre | 32 | 19 | 51 |
| 2015 Septiembre | 26 | 10 | 36 |
| 2015 Agosto | 17 | 6 | 23 |
| 2015 Julio | 245 | 2 | 247 |
| 2015 Junio | 113 | 10 | 123 |
| 2015 Mayo | 120 | 8 | 128 |
| 2015 Abril | 109 | 8 | 117 |
| 2015 Marzo | 76 | 4 | 80 |
| 2015 Febrero | 86 | 1 | 87 |
| 2015 Enero | 109 | 0 | 109 |
| 2014 Diciembre | 81 | 1 | 82 |
| 2014 Noviembre | 71 | 0 | 71 |
| 2014 Octubre | 64 | 0 | 64 |
| 2014 Septiembre | 41 | 0 | 41 |
| 2014 Agosto | 46 | 0 | 46 |
| 2014 Julio | 43 | 0 | 43 |
| 2014 Junio | 64 | 0 | 64 |
| 2014 Mayo | 74 | 0 | 74 |
| 2014 Abril | 52 | 0 | 52 |
| 2014 Marzo | 103 | 13 | 116 |
| 2014 Febrero | 100 | 8 | 108 |
| 2014 Enero | 115 | 12 | 127 |
| 2013 Diciembre | 107 | 15 | 122 |
| 2013 Noviembre | 84 | 13 | 97 |
| 2013 Octubre | 76 | 6 | 82 |
| 2013 Septiembre | 50 | 7 | 57 |
| 2013 Agosto | 43 | 8 | 51 |
| 2013 Julio | 52 | 23 | 75 |
| 2013 Junio | 59 | 20 | 79 |
| 2013 Mayo | 60 | 8 | 68 |
| 2013 Abril | 37 | 15 | 52 |
| 2013 Marzo | 46 | 13 | 59 |
| 2013 Febrero | 38 | 11 | 49 |
| 2013 Enero | 41 | 10 | 51 |
| 2012 Diciembre | 35 | 10 | 45 |
| 2012 Noviembre | 6 | 14 | 20 |
| 2012 Octubre | 2 | 5 | 7 |
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