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Brito Caldeira, M. Pestana, B. Duarte" "autores" => array:3 [ 0 => array:2 [ "nombre" => "M." "apellidos" => "Brito Caldeira" ] 1 => array:2 [ "nombre" => "M." "apellidos" => "Pestana" ] 2 => array:2 [ "nombre" => "B." "apellidos" => "Duarte" ] ] ] ] ] "idiomaDefecto" => "es" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0001731023007482?idApp=UINPBA000044" "url" => "/00017310/0000011400000010/v1_202311020433/S0001731023007482/v1_202311020433/es/main.assets" ] ] "en" => array:15 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Case and Research Letter</span>" "titulo" => "Successful Treatment of Hailey-Hailey Disease With Dupilumab" "tieneTextoCompleto" => true "saludo" => "To the Editor:" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "914" "paginaFinal" => "915" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "M. Brito Caldeira, M. Pestana, B. Duarte" "autores" => array:3 [ 0 => array:4 [ "nombre" => "M." "apellidos" => "Brito Caldeira" "email" => array:1 [ 0 => "margaridabcaldeira@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "M." "apellidos" => "Pestana" ] 2 => array:2 [ "nombre" => "B." "apellidos" => "Duarte" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Dermatology and Venereology Department, Centro Hospitalar Universitário de Lisboa Central, EPE, Lisbon, Portugal" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Tratamiento exitoso de la efermedad de Hailey-Hailey con dupilumab" ] ] "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" => 631 "Ancho" => 1340 "Tamanyo" => 173988 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Clinical pictures of Hailey-Hailey disease lesions on inferior abdominal skin fold at week 0 (a) and week 12 (b).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Hailey-Hailey disease is a genodermatosis of autosomal dominant transmission, caused by loss-of-function mutations in the ATP2C1 gene. Failure of a proper intracellular calcium homeostasis, particularly in the Golgi apparatus, is responsible for a weakened desmosomal adhesion between keratinocytes. Thus, acantholysis of stratum spinosum occurs, causing a chronic clinical appearance of flaccid blisters and malodourous erosions in intertriginous areas. Chronic pruritus is also frequently present and difficult to treat, contributing to the severe burden of the disease.</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 57-year-old woman suffered from Hailey-Hailey disease since early adulthood, which was refractory to several different topical and systemic treatments, including a recently described novel approach with low-dose naltrexone.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">1</span></a> She had malodourous bilateral hyperkeratotic papules and erosions on axillae, inframammary and abdominal folds (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>a). Pruritus was her main complaint, as it was persistent, intense (Numeric Rating Scale itch 8/10) and disruptive of both her sleep quality and social life.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Dupilumab 300<span class="elsevierStyleHsp" style=""></span>mg every other week was started after a 600<span class="elsevierStyleHsp" style=""></span>mg loading dose. Both pruritus and sleep quality drastically improved after only one week of treatment. Additionally, significant healing of the skin lesions was noticeable by week 12, with reduction in size and thickness of previous hyperkeratotic papules and erosions (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>b). Treatment was well tolerated, and the patient is satisfied with the outcome.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Our case reflects a remarkable improvement with dupilumab, a fully human monoclonal antibody against interleukins (IL) 4 and 13, on both subjective and objective findings in Hailey-Hailey disease, thus corroborating the findings by Alzahrani et al.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">2</span></a> This drug is currently approved for moderate-to-severe atopic dermatitis, showing significant reduction of pruritus right after the induction phase. Even though the mechanism of dupilumab on Hailey-Hailey disease is still unknown, it is possible that the inhibition of IL-4 and IL-13 modulates favourably the intracellular calcium signalling,<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">2</span></a> thus compensating its genetic defect. Furthermore, we emphasize the central role of IL-4 as a neuromodulator on chronic pruritus, which is responsible for amplifying responses to minor pruritogenic agents.<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">3,4</span></a> The expression of IL-4 receptors on sensory neurons of the dorsal root ganglion<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">4</span></a> further corroborates the relevance of this molecule in chronic pruritus.</p><p id="par0025" class="elsevierStylePara elsevierViewall">In summary, we highlight dupilumab's innovative role on managing symptoms and objective findings in patients with Hailey-Hailey disease. It is possible that this positive effect could be extrapolated to other dyskeratosis with severe pruritus, but further investigation is needed.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of interest</span><p id="par0030" 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 "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" => 631 "Ancho" => 1340 "Tamanyo" => 173988 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Clinical pictures of Hailey-Hailey disease lesions on inferior abdominal skin fold at week 0 (a) and week 12 (b).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:4 [ 0 => array:3 [ "identificador" => "bib0025" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Treatment of Hailey-Hailey disease with low-dose naltrexone" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "L.N. Albers" 1 => "J.L. Arbiser" 2 => "R.J. Feldman" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1001/jamadermatol.2017.2446" "Revista" => array:5 [ "tituloSerie" => "JAMA Dermatol" "fecha" => "2017" "volumen" => "153" "paginaInicial" => "1018" "paginaFinal" => "1020" ] ] ] ] ] ] 1 => array:3 [ "identificador" => "bib0030" "etiqueta" => "2" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Hailey-Hailey disease treated with dupilumab: a case series" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "N. Alzahrani" 1 => "J. Grossman-Kranseler" 2 => "R. Swali" 3 => "K. Fiumara" 4 => "P. Zancanaro" 5 => "S. Tyring" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1111/bjd.20475" "Revista" => array:6 [ "tituloSerie" => "Br J Dermatol" "fecha" => "2021" "volumen" => "185" "paginaInicial" => "680" "paginaFinal" => "682" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/33971025" "web" => "Medline" ] ] ] ] ] ] ] ] 2 => array:3 [ "identificador" => "bib0035" "etiqueta" => "3" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Neuroimmunology: scratching the surface of chronic itch" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:1 [ 0 => "K. Whalley" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1038/nrn.2017.126" "Revista" => array:5 [ "tituloSerie" => "Nat Rev Neurosci" "fecha" => "2017" "volumen" => "18" "paginaInicial" => "642" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/28978979" "web" => "Medline" ] ] ] ] ] ] ] ] 3 => array:3 [ "identificador" => "bib0040" "etiqueta" => "4" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Interleukin-4 and interleukin-13 evoke scratching behaviour in mice" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:6 [ 0 => "M. Campion" 1 => "L. Smith" 2 => "S. Gatault" 3 => "C. Métais" 4 => "J. Buddenkotte" 5 => "M. Steinhoff" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1111/exd.14034" "Revista" => array:6 [ "tituloSerie" => "Exp Dermatol" "fecha" => "2019" "volumen" => "28" "paginaInicial" => "1501" "paginaFinal" => "1504" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/31505056" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/00017310/0000011400000010/v1_202311020433/S0001731023000777/v1_202311020433/en/main.assets" "Apartado" => array:4 [ "identificador" => "6160" "tipo" => "SECCION" "es" => array:2 [ "titulo" => "Cartas científico-clínicas" "idiomaDefecto" => true ] "idiomaDefecto" => "es" ] "PDF" => "https://static.elsevier.es/multimedia/00017310/0000011400000010/v1_202311020433/S0001731023000777/v1_202311020433/en/main.pdf?idApp=UINPBA000044&text.app=https://actasdermo.org/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0001731023000777?idApp=UINPBA000044" ]
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