Case Letter
Therapeutic approach of Lyell syndrome with infliximab and dexamethasone pulse: report of a clinical case
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Dara Jácome Pacheco, Maria Rodrigues Viegas Ribeiro, Catarina Sousa Duque Soares Queirós, Maria Fátima Cameira Martins Xambre" "autores" => array:4 [ 0 => array:4 [ "nombre" => "Jânia Dara Jácome" "apellidos" => "Pacheco" "email" => array:1 [ 0 => "jania_pacheco@hotmail.com" ] "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" => "Maria Rodrigues Viegas" "apellidos" => "Ribeiro" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "Catarina Sousa Duque Soares" "apellidos" => "Queirós" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "Maria Fátima Cameira Martins" "apellidos" => "Xambre" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Department of Anaesthesiology, Hospital de Santa Maria, Centro Hospitalar Universitário Lisboa Norte, Lisboa, Portugal" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Department of Dermatology, Hospital de Santa Maria, Centro Hospitalar Universitário Lisboa Norte, Lisboa, Portugal" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 3114 "Ancho" => 1341 "Tamanyo" => 180818 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Detachment of the epidermis.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Toxic Epidermal Necrolysis (TEN) is a rare, life-threatening mucocutaneous disease.</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 23-year-old female presented with TEN after treatment with diclofenac, metamizole and etoricoxib. She was febrile, with oral, ocular, and perineal mucositis and a maculopapular rash involving an estimated body surface area of 20%. She was admitted to the dermatology department, skin biopsies were collected, and Intravenous Immunoglobulin (IVIg) was started. However, her clinical condition worsened, so she was transferred to the burn unit.</p><p id="par0015" class="elsevierStylePara elsevierViewall">Upon admission, the patient had an extensive maculopapular rash. There were also positive Nikolsky signs in her face, back, palms and feet soles, and involvement of the oral, ocular, vaginal and perineal mucous membranes (<a class="elsevierStyleCrossRefs" href="#fig0005">Figs. 1 and 2</a>). Additionally, given the upper airway edema, she was orotracheal intubated.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Under immunoglobulin therapy, there was a clinical worsening, with an increase in the body surface involvement, from 85% to 100%. In addition, she developed multiorgan failure with hematological, cardiovascular, hepatic, gastrointestinal and renal dysfunction.</p><p id="par0025" class="elsevierStylePara elsevierViewall">On the eighth day of hospitalization, the patient received a single infliximab dose (5 mg/kg, EV) and was started on a pulse of dexamethasone (100 mg, EV × 1/day) for three days. After administration of infliximab and dexamethasone pulse, the patient evolved favorably with the progressive resolution of multiorgan dysfunction, and on the nineteenth day, the patient presented epithelialization of the entire body surface.</p><p id="par0030" class="elsevierStylePara elsevierViewall">According to literature, TEN is a severe idiosyncratic reaction, mostly drug-induced. It is characterized by a detachment of the skin and mucous membranes at the level of the dermis-epidermis junction as a result of the necrosis of keratinocytes.</p><p id="par0035" class="elsevierStylePara elsevierViewall">The diagnosis of TEN is clinical. It is characterized by a prodromal phase with flu-like symptoms that precede the appearance of mucocutaneous lesions. The confirmation of the diagnosis involves a skin biopsy.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">In patients with probable drug-induced TEN, early diagnosis and immediate removal of the drug are essential to improve the prognosis.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Apart from supportive care, there is no established treatment. However, several immunosuppressants and immunomodulators have been used, and although there are no adequate randomized studies, some drugs have shown promise.</p><p id="par0050" class="elsevierStylePara elsevierViewall">Corticosteroids were among the first drugs used; however, their use is currently controversial. Although some studies demonstrate a worse prognosis,<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> there are others that show benefits with the administration of high doses over a short period of time.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">IVIg used to be the most consensual therapy for the treatment of this syndrome. However, a meta-analysis published in 2012 concluded that this drug is not associated with a better prognosis.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">TNF-alpha inhibitors seem promising drugs in the treatment of TEN. There are some case reports where a single dose of infliximab or etanercept interrupted the progression of the disease and induced regression of the skin lesions.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">In fact, the case we reported is in line with these findings since the patient did not show any improvement after IVIg administration but had a great response to infliximab and high-dose dexamethasone.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Financial support</span><p id="par0070" class="elsevierStylePara elsevierViewall">None declared.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Authors' contributions</span><p id="par0075" class="elsevierStylePara elsevierViewall">Jânia Dara Jácome Pacheco: Collection, analysis, and interpretation of data; effective participation in research orientation; intellectual participation in the propaedeutic and/or therapeutic conduct of the studied cases; management of case studies; critical review of the manuscript; drafting and editing of the manuscript; critical review of the literature.</p><p id="par0080" class="elsevierStylePara elsevierViewall">Maria Rodrigues Viegas Ribeiro: Critical review of the literature; collection, analysis, and interpretation of data; drafting and editing of the manuscript.</p><p id="par0085" class="elsevierStylePara elsevierViewall">Catarina Sousa Duque Soares Queirós: Critical review of the manuscript.</p><p id="par0090" class="elsevierStylePara elsevierViewall">Maria Fátima Cameira Martins Xambre: Approval of the final version of the manuscript.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflicts of interest</span><p id="par0095" class="elsevierStylePara elsevierViewall">None declared.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:4 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Financial support" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Authors' contributions" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Conflicts of interest" ] 3 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2020-07-30" "fechaAceptado" => "2020-10-13" "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Study conducted at the Hospital de Santa Maria, Centro Hospitalar Universitário Lisboa Norte, Lisboa, Portugal.</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" => 928 "Ancho" => 1675 "Tamanyo" => 118454 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Blisters, vesicles, and detachment of the epidermis.</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" => 3114 "Ancho" => 1341 "Tamanyo" => 180818 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Detachment of the epidermis.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Toxic epidermal necrolysis (Lyell syndrome): a pathology for burn units" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "L. 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"fecha" => "2007" "volumen" => "87" "paginaInicial" => "144" "paginaFinal" => "148" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/17340021" "web" => "Medline" ] ] ] ] ] ] ] ] 3 => array:3 [ "identificador" => "bib0020" "etiqueta" => "4" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "The efficacy of intravenous immunoglobulin for the treatment of toxic epidermal necrolysis: a systematic review and meta-analysis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "Y.C. Huang" 1 => "Y.C. Li" 2 => "T.J. Chen" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:5 [ "tituloSerie" => "Br J Dermatol." 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 14 | 10 | 24 |
| 2024 October | 121 | 59 | 180 |
| 2024 September | 119 | 72 | 191 |
| 2024 August | 163 | 108 | 271 |
| 2024 July | 152 | 109 | 261 |
| 2024 June | 117 | 61 | 178 |
| 2024 May | 97 | 66 | 163 |
| 2024 April | 133 | 71 | 204 |
| 2024 March | 95 | 59 | 154 |
| 2024 February | 86 | 91 | 177 |
| 2024 January | 73 | 49 | 122 |
| 2023 December | 63 | 60 | 123 |
| 2023 November | 82 | 99 | 181 |
| 2023 October | 59 | 86 | 145 |
| 2023 September | 93 | 70 | 163 |
| 2023 August | 56 | 35 | 91 |
| 2023 July | 63 | 28 | 91 |
| 2023 June | 63 | 36 | 99 |
| 2023 May | 64 | 31 | 95 |
| 2023 April | 47 | 17 | 64 |
| 2023 March | 85 | 45 | 130 |
| 2023 February | 60 | 33 | 93 |
| 2023 January | 52 | 34 | 86 |
| 2022 December | 71 | 30 | 101 |
| 2022 November | 96 | 57 | 153 |
| 2022 October | 112 | 46 | 158 |
| 2022 September | 52 | 64 | 116 |
| 2022 August | 79 | 44 | 123 |
| 2022 July | 89 | 62 | 151 |
| 2022 June | 91 | 79 | 170 |
| 2022 May | 82 | 73 | 155 |
| 2022 April | 65 | 56 | 121 |
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