Case and Research Letter
Pemphigus Herpetiformis With Progression to Pemphigus Foliaceus: A Case Report
Pénfigo herpetiforme con evolución a pénfigo foliáceo. Descripción de un caso
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Descripción de un caso" "tienePdf" => "es" "tieneTextoCompleto" => "es" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "526" "paginaFinal" => "528" ] ] "titulosAlternativos" => array:1 [ "en" => array:1 [ "titulo" => "Pemphigus Herpetiformis With Progression To Pemphigus Foliaceus: A Case Report" ] ] "contieneTextoCompleto" => array:1 [ "es" => true ] "contienePdf" => array:1 [ "es" => true ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0015" "etiqueta" => "Figura 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 831 "Ancho" => 1429 "Tamanyo" => 208685 ] ] "descripcion" => array:1 [ "es" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">A. Placa eritematosa descamativa localizada en la zona retroauricular. B. 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Fuentes-Finkelstein, M. Barnadas, C. Gelpi, L. Puig" "autores" => array:4 [ 0 => array:4 [ "nombre" => "P." "apellidos" => "Fuentes-Finkelstein" "email" => array:1 [ 0 => "pfuentesf@santpau.cat" ] "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" => "M." "apellidos" => "Barnadas" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "C." "apellidos" => "Gelpi" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "L." "apellidos" => "Puig" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicio Dermatología, Hospital de la Santa Creu i Sant Pau, Barcelona, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Inmunología, Hospital de la Santa Creu i Sant Pau, Barcelona, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Pénfigo herpetiforme con evolución a pénfigo foliáceo. Descripción de un caso" ] ] "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" => 752 "Ancho" => 1293 "Tamanyo" => 188891 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">A, Erythematous scaling plaque in the retroauricular area. B, Acantholytic subcorneal vesicle (hematoxylin eosin, original magnification ×400).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Pemphigus herpetiformis (PH) is an uncommon variant of pemphigus that accounts for an estimated 6% to 7.2% of all cases of this skin disorder.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a> The term <span class="elsevierStyleItalic">pemphigus herpetiformis</span> was coined by Jablonska et al.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> in 1975 to describe a entity that was clinically similar to dermatitis herpetiformis, showed acantholysis on biopsy, and responded to sulfapyridine. The authors considered that PH was a variant of pemphigus based on the direct immunofluorescence findings. PH typically presents with annular erythematous plaques, peripheral vesicles, and on occasions intense pruritus. Histologic features are highly variable and depend on the stage of the lesion. There have been reports of PH preceding or developing concurrently with pemphigus foliaceous (PF) or pemphigus vulgaris (PV).<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3–5</span></a> We present a case of PH that progressed to PF.</p><p id="par0010" class="elsevierStylePara elsevierViewall">The patient, a 34-year-old woman with no relevant past history, consulted for erythematous papules and plaques with peripheral vesicles and blisters on the lower extremities, the abdomen, and the scalp (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). The lesions had been present for 3 months. There was no mucosal involvement. Two biopsies revealed different degrees of neutrophilic and eosinophilic spongiosis (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>A) and acantholysis, with the formation of intraepidermal vesicles (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>B). Several eosinophils were also observed in the papillary dermis. Direct immunofluorescence showed intercellular immunoglobulin (Ig) G and C3 deposits, predominantly in the suprabasal layers of the epidermis. Anti-epithelial antibodies (titer, 1:40) and anti-desmoglein 1 (Dsg1) antibodies were positive (175 IU/mL; normal value, <<span class="elsevierStyleHsp" style=""></span>20 IU/mL); the results for anti-Dsg1 antibodies were negative. A diagnosis of PH was made and treatment was started with prednisone 20<span class="elsevierStyleHsp" style=""></span>mg/d and topical clobetasol. The lesions improved, but 3 months later, erythematous scaling plaques started to reappear in the presternal, dorsal, and retroauricular areas and on the scalp (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>A). Biopsy of one of these plaques showed a subcorneal acantholytic vesicle (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>B). The anti-epithelial antibody titer was 1:80 and anti-Dsg1 antibody levels remained high at 161<span class="elsevierStyleHsp" style=""></span>IU/mL. Given the persistence of the lesions, treatment was started with dapsone 50<span class="elsevierStyleHsp" style=""></span>mg/d, with dose increments up to 100<span class="elsevierStyleHsp" style=""></span>mg/d. The clinical response was favorable and the dose was progressively reduced to 12.5<span class="elsevierStyleHsp" style=""></span>mg every second day. Occasional flares consisting of minimal papules with scaling on the neckline, back, and scalp were observed.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">PH is a variant of pemphigus that generally has a good prognosis, and most patients respond to sulfones.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> PH has a wide spectrum of clinical and histologic findings and accordingly numerous autoimmune blistering disorders must be considered in the differential diagnosis, namely, dermatitis herpetiformis, linear IgA bullous dermatosis, PF, PV, and bullous pemphigoid.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> Histologic findings can vary according to the stage of the disease and the type of lesion biopsied. Varying degrees of neutrophilic and/or eosinophilic spongiosis, with or without acantholysis in the middle and/or subcorneal layer, are observed. Kuhn et al.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> found that the inflammatory infiltrate in patients with PH was 68% eosinophil-dominant, 16% neutrophil-dominant, and 16% mixed eosinophil/neutrophil. We would like to stress the importance of performing direct immunofluorescence to test for the presence of an autoimmune blistering disorder when histology reveals neutrophilic and/or eosinophilic spongiosis.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Anti-Dsg1 and/or anti-Dsg3 antibodies have been described in most cases of pH,<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5,8</span></a> but there have been isolated reports of negative results for both antibodies.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">We have described an atypical course of PH that has been previously reported by Maciejowska et al.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> in 2 of 15 patients and by Santi et al.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> in 1 of 7 patients. There have also been reports of cases of PH developing after or at the same time as PF or PV.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,5</span></a> This possibility, together with the fact that anti-Dsg1 antibodies are detected in PH, has led to the hypothesis that PH and PF might be connected and that PH might actually be a variant of PF.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">It is not known why patients with PH, despite having anti-Dsg1 antibodies, have different clinical manifestations and histological findings to those with PF. Several hypotheses have been proposed. One is that in PH, IgG might cause keratinocytes to induce interleukin 8, whose chemotactic activity would explain the presence of a neutrophilic infiltrate.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> Another is that patients might develop antibodies that, despite their minimum acantholytic activity, could activate eosinophils and neutrophils through the Fc portion of IgG.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Finally, it is possible that antibodies targeting epidermal antigens other than desmogleins or different epitopes might be responsible for the different phenotypic expression of pemphigus seen in PH.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Fuentes-Finkelstein P, Barnadas M, Gelpi C, Puig L. Pénfigo herpetiforme con evolución a pénfigo foliáceo. Descripción de un caso. Actas Dermosifiliogr. 2014;105:533–535.</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" => 1004 "Ancho" => 754 "Tamanyo" => 106826 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Erythematous papules and plaques with circinate borders and peripheral vesicles.</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" => 867 "Ancho" => 1301 "Tamanyo" => 276426 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A, Neutrophilic spongiosis in the epidermis with isolated eosinophils (hematoxylin eosin, original magnification ×400). B, Acantholytic vesicle in the middle layer of the epidermis accompanied by neutrophils and eosinophils.</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" => 752 "Ancho" => 1293 "Tamanyo" => 188891 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">A, Erythematous scaling plaque in the retroauricular area. B, Acantholytic subcorneal vesicle (hematoxylin eosin, original magnification ×400).</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" => "Is pemphigus herpetiformis an entity?" 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 14 | 15 | 29 |
| 2024 October | 139 | 59 | 198 |
| 2024 September | 151 | 28 | 179 |
| 2024 August | 162 | 73 | 235 |
| 2024 July | 192 | 40 | 232 |
| 2024 June | 116 | 38 | 154 |
| 2024 May | 130 | 37 | 167 |
| 2024 April | 100 | 31 | 131 |
| 2024 March | 122 | 32 | 154 |
| 2024 February | 102 | 39 | 141 |
| 2024 January | 98 | 35 | 133 |
| 2023 December | 105 | 31 | 136 |
| 2023 November | 106 | 33 | 139 |
| 2023 October | 84 | 46 | 130 |
| 2023 September | 109 | 42 | 151 |
| 2023 August | 70 | 20 | 90 |
| 2023 July | 94 | 36 | 130 |
| 2023 June | 83 | 28 | 111 |
| 2023 May | 117 | 30 | 147 |
| 2023 April | 82 | 23 | 105 |
| 2023 March | 69 | 26 | 95 |
| 2023 February | 77 | 19 | 96 |
| 2023 January | 64 | 20 | 84 |
| 2022 December | 75 | 42 | 117 |
| 2022 November | 60 | 27 | 87 |
| 2022 October | 41 | 24 | 65 |
| 2022 September | 44 | 49 | 93 |
| 2022 August | 70 | 33 | 103 |
| 2022 July | 34 | 27 | 61 |
| 2022 June | 55 | 30 | 85 |
| 2022 May | 97 | 43 | 140 |
| 2022 April | 100 | 35 | 135 |
| 2022 March | 117 | 72 | 189 |
| 2022 February | 92 | 24 | 116 |
| 2022 January | 74 | 40 | 114 |
| 2021 December | 58 | 42 | 100 |
| 2021 November | 105 | 53 | 158 |
| 2021 October | 95 | 42 | 137 |
| 2021 September | 115 | 30 | 145 |
| 2021 August | 145 | 40 | 185 |
| 2021 July | 147 | 31 | 178 |
| 2021 June | 84 | 24 | 108 |
| 2021 May | 80 | 25 | 105 |
| 2021 April | 205 | 104 | 309 |
| 2021 March | 111 | 35 | 146 |
| 2021 February | 69 | 27 | 96 |
| 2021 January | 33 | 19 | 52 |
| 2020 December | 53 | 12 | 65 |
| 2020 November | 47 | 6 | 53 |
| 2020 October | 62 | 9 | 71 |
| 2020 September | 80 | 10 | 90 |
| 2020 August | 63 | 19 | 82 |
| 2020 July | 46 | 25 | 71 |
| 2020 June | 44 | 21 | 65 |
| 2020 May | 32 | 16 | 48 |
| 2020 April | 29 | 15 | 44 |
| 2020 March | 31 | 12 | 43 |
| 2020 February | 3 | 3 | 6 |
| 2020 January | 0 | 5 | 5 |
| 2019 December | 4 | 5 | 9 |
| 2019 November | 0 | 2 | 2 |
| 2019 October | 0 | 3 | 3 |
| 2019 September | 4 | 5 | 9 |
| 2019 August | 2 | 2 | 4 |
| 2019 July | 0 | 4 | 4 |
| 2019 June | 3 | 7 | 10 |
| 2019 May | 1 | 29 | 30 |
| 2019 April | 14 | 1 | 15 |
| 2019 March | 2 | 8 | 10 |
| 2019 February | 1 | 0 | 1 |
| 2019 January | 2 | 1 | 3 |
| 2018 December | 1 | 0 | 1 |
| 2018 November | 2 | 0 | 2 |
| 2018 October | 3 | 0 | 3 |
| 2018 September | 5 | 0 | 5 |
| 2018 April | 0 | 1 | 1 |
| 2018 March | 3 | 4 | 7 |
| 2018 February | 30 | 2 | 32 |
| 2018 January | 48 | 5 | 53 |
| 2017 December | 40 | 5 | 45 |
| 2017 November | 32 | 4 | 36 |
| 2017 October | 37 | 5 | 42 |
| 2017 September | 42 | 12 | 54 |
| 2017 August | 58 | 6 | 64 |
| 2017 July | 63 | 10 | 73 |
| 2017 June | 67 | 44 | 111 |
| 2017 May | 50 | 15 | 65 |
| 2017 April | 53 | 13 | 66 |
| 2017 March | 43 | 29 | 72 |
| 2017 February | 39 | 5 | 44 |
| 2017 January | 25 | 6 | 31 |
| 2016 December | 41 | 13 | 54 |
| 2016 November | 55 | 13 | 68 |
| 2016 October | 83 | 7 | 90 |
| 2016 September | 108 | 9 | 117 |
| 2016 August | 46 | 2 | 48 |
| 2016 July | 41 | 8 | 49 |
| 2016 June | 5 | 1 | 6 |
| 2016 May | 2 | 4 | 6 |
| 2016 April | 4 | 8 | 12 |
| 2016 March | 5 | 7 | 12 |
| 2016 February | 4 | 11 | 15 |
| 2016 January | 14 | 1 | 15 |
| 2015 December | 9 | 5 | 14 |
| 2015 November | 3 | 5 | 8 |
| 2015 October | 4 | 1 | 5 |
| 2015 September | 2 | 5 | 7 |
| 2015 August | 5 | 3 | 8 |
| 2015 July | 42 | 8 | 50 |
| 2015 June | 35 | 6 | 41 |
| 2015 May | 46 | 12 | 58 |
| 2015 April | 40 | 8 | 48 |
| 2015 March | 39 | 8 | 47 |
| 2015 February | 42 | 7 | 49 |
| 2015 January | 29 | 5 | 34 |
| 2014 December | 45 | 5 | 50 |
| 2014 November | 40 | 9 | 49 |
| 2014 October | 27 | 3 | 30 |
| 2014 September | 16 | 4 | 20 |
| 2014 August | 15 | 4 | 19 |
| 2014 July | 9 | 7 | 16 |
| 2014 June | 6 | 5 | 11 |
| 2014 May | 2 | 1 | 3 |
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