Case and Research Letter
Herpetiforme Pemphigus: Report of a Pediatric Case
Pénfigo herpetiforme: revisión de un caso en edad pediátrica
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Taliercio, B. Ferrari, M.E. Abad, M. Larralde" "autores" => array:4 [ 0 => array:4 [ "nombre" => "V.L." "apellidos" => "Taliercio" "email" => array:2 [ 0 => "vanitali@gmail.com" 1 => "brunoferraripiel@gmail.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" => "B." "apellidos" => "Ferrari" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "M.E." "apellidos" => "Abad" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 3 => array:3 [ "nombre" => "M." "apellidos" => "Larralde" "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Sección de Dermatología Pediátrica, Hospital Ramos Mejía, Buenos Aires, Argentina" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Departamento de Dermatología, Hospital Alemán, Buenos Aires, Argentina" "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: revisión de un caso en edad pediátrica" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1350 "Ancho" => 1800 "Tamanyo" => 433297 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Branching vesicles on healthy and erythematous skin. Crusts and erosions on the right leg.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Pemphigus herpetiformis (PH) is an uncommon and atypical variant of pemphigus.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> It appears mainly between ages 30 and 80 years<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> and is exceptional in children and adolescents.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> Clinically, it is characterized by polymorphic lesions, with erythematous-edematous plaques of different sizes, erosions, excoriations resulting from scratching, vesicles arranged in branches, and blisters containing serum, blood and serum, or purulent material.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> Diagnosis is based on histopathology and confirmed by direct immunofluorescence.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a></p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Case Description</span><p id="par0010" class="elsevierStylePara elsevierViewall">The patient was a 14-year-old girl with no personal history of interest who had had generalized pruriginous dermatosis for the previous 2 years.</p><p id="par0015" class="elsevierStylePara elsevierViewall">Physical examination revealed blisters containing serum arranged in rosette formation on erythematous plaques and healthy skin, vesicles grouped together in branches (<a class="elsevierStyleCrossRefs" href="#fig0005">Figs. 1 and 2</a>), erosions, crusts, and excoriations resulting from scratching because of intense pruritus. The lesions were located on the trunk, upper and lower limbs, and gluteal area. The oral and genital mucosa and scalp were spared, and the Nikolsky sign was negative. Her general status was normal.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Histopathology of a blister revealed acantholysis with intermediate and focally suprabasal cleavage compatible with pemphigus. Clusters of neutrophils and occasional eosinophils were observed between the acantholytic keratinocytes in the stratum spinosum (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>).</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">Direct immunofluorescence revealed deposits of intradermal and intracellular immunoglobulin (Ig) G and absence of IgA, thus enabling us to diagnose PH.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Treatment was started with methylprednisolone at 1<span class="elsevierStyleHsp" style=""></span>mg/kg/d, which was subsequently complemented with mycophenolate mofetil. Corticosteroids were tapered until discontinuation. Since the active lesions had resolved after 2 years, treatment was discontinued.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Discussion</span><p id="par0035" class="elsevierStylePara elsevierViewall">PH is not a classic form of pemphigus. It was first reported in 1955, although the term PH was first used in 1975, since the condition combined clinical characteristics that mimicked dermatitis herpetiformis with the typical immunohistologic characteristics of pemphigus.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">The incidence of PH is very low (6% of all types of pemphigus).<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> It presents between ages 30 and 80 years and is exceptional in children. It affects both sexes equally.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Clinically, it is characterized by the presence of vesicles and blisters arranged in branches or rosettes mixed with erosions and crusts to give a variable appearance.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,4</span></a> The scalp, trunk, and limbs can also be affected; the mucosa are rarely involved.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Histology findings vary with the progress of the lesions and range from subcorneal pustules and intraepidermal vesicles that contain neutrophils and eosinophilic spongiosis to acantholysis.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> The cleavage site varies with the target antigen. In most cases of PH, the target antigen is desmoglein 1,<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> which predominates in the superficial layers of the epidermis (subcorneal location). The histopathology findings are similar to those of pemphigus foliaceus. In contrast, when the target antigen is desmoglein 3, acantholysis can be observed in the suprabasal and intermediate layers of the epidermis, thus pointing to pemphigus vulgaris in histology.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">Direct immunofluorescence demonstrates the presence of intercellular deposits of—mainly—IgG and C3 in the epidermis,<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> as in the case we report. Indirect immunofluorescence reveals circulating IgG antibodies that mainly target desmoglein 1 and, in some cases, desmoglein 3. Antibodies against desmocollin 1 and 3 and an unknown 178–kDa protein were recently reported in patients with PH.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">The differential diagnosis should take into account herpetiform dermatitis, pemphigus foliaceus, pemphigus vulgaris, and linear IgA dermatosis.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">Although the disease is benign with a good response to therapy and remission tends to be complete, treatment-refractory cases have been reported<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a>; hence the association with circulating antibody titers.<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">Although the association with tumors in patients with pemphigus is open to debate, there have been reports of cases of PH that occurred simultaneously with neoplasms, especially those affecting the lung. PH has also been associated with lupus and infectious diseases such as HIV infection.<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">7–9</span></a></p><p id="par0075" class="elsevierStylePara elsevierViewall">The many therapeutic options include systemic corticosteroids (prednisone 0.5-1<span class="elsevierStyleHsp" style=""></span>mg/kg/d), dapsone (100-300<span class="elsevierStyleHsp" style=""></span>mg), or a combination of both.<a class="elsevierStyleCrossRefs" href="#bib0070"><span class="elsevierStyleSup">4,10</span></a> In refractory cases, immunosuppressive drugs are a valid option for inducing remission.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,2,10</span></a></p><p id="par0080" class="elsevierStylePara elsevierViewall">The patient in the present report received prednisone for 1 year. Mycophenolate mofetil was subsequently added. The dose of corticosteroids was gradually reduced, and the patient achieved complete remission. Progress was favorable over 3 years of follow-up.</p><p id="par0085" class="elsevierStylePara elsevierViewall">The interest of this case report lies in the occurrence of an infrequent condition at an unusual age. We stress the favorable response to treatment.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflicts of Interest</span><p id="par0090" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:4 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Case Description" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Discussion" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Conflicts of Interest" ] 3 => 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: Taliercio VL, Ferrari B, Abad ME, Larralde M. Pénfigo herpetiforme: revisión de un caso en edad pediátrica. Actas Dermosifiliogr. 2017;108:276–277.</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" => 1125 "Ancho" => 1500 "Tamanyo" => 320237 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Blisters containing serum arranged in a rosette pattern with a central crust on erythematous skin.</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" => 1350 "Ancho" => 1800 "Tamanyo" => 433297 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Branching vesicles on healthy and erythematous skin. Crusts and erosions 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" => 1565 "Ancho" => 1417 "Tamanyo" => 315837 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Intraepidermal blister with suprabasal and intermediate layer acantholysis and numerous acantholytic cells in the interior. Also visible are clusters of neutrophils and eosinophils between the acantholytic keratinocytes of the stratum spinosum (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" => "bib0055" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Herpetiform pemphigus, a variable pattern of pemphigus" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "S. Jablonska" 1 => "T.P. Chorzelski" 2 => "E.H. Beutner" 3 => "J. 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| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 8 | 5 | 13 |
| 2024 Octubre | 77 | 49 | 126 |
| 2024 Septiembre | 70 | 29 | 99 |
| 2024 Agosto | 100 | 71 | 171 |
| 2024 Julio | 82 | 34 | 116 |
| 2024 Junio | 110 | 26 | 136 |
| 2024 Mayo | 94 | 33 | 127 |
| 2024 Abril | 101 | 33 | 134 |
| 2024 Marzo | 68 | 22 | 90 |
| 2024 Febrero | 80 | 30 | 110 |
| 2024 Enero | 54 | 31 | 85 |
| 2023 Diciembre | 46 | 15 | 61 |
| 2023 Noviembre | 75 | 24 | 99 |
| 2023 Octubre | 55 | 24 | 79 |
| 2023 Septiembre | 55 | 30 | 85 |
| 2023 Agosto | 42 | 20 | 62 |
| 2023 Julio | 58 | 29 | 87 |
| 2023 Junio | 43 | 24 | 67 |
| 2023 Mayo | 57 | 32 | 89 |
| 2023 Abril | 65 | 24 | 89 |
| 2023 Marzo | 59 | 31 | 90 |
| 2023 Febrero | 50 | 27 | 77 |
| 2023 Enero | 43 | 19 | 62 |
| 2022 Diciembre | 52 | 43 | 95 |
| 2022 Noviembre | 27 | 20 | 47 |
| 2022 Octubre | 24 | 18 | 42 |
| 2022 Septiembre | 23 | 33 | 56 |
| 2022 Agosto | 26 | 26 | 52 |
| 2022 Julio | 22 | 31 | 53 |
| 2022 Junio | 30 | 18 | 48 |
| 2022 Mayo | 36 | 33 | 69 |
| 2022 Abril | 43 | 43 | 86 |
| 2022 Marzo | 52 | 53 | 105 |
| 2022 Febrero | 48 | 33 | 81 |
| 2022 Enero | 43 | 40 | 83 |
| 2021 Diciembre | 53 | 39 | 92 |
| 2021 Noviembre | 44 | 55 | 99 |
| 2021 Octubre | 55 | 60 | 115 |
| 2021 Septiembre | 42 | 39 | 81 |
| 2021 Agosto | 49 | 42 | 91 |
| 2021 Julio | 48 | 35 | 83 |
| 2021 Junio | 35 | 31 | 66 |
| 2021 Mayo | 39 | 57 | 96 |
| 2021 Abril | 82 | 56 | 138 |
| 2021 Marzo | 64 | 46 | 110 |
| 2021 Febrero | 72 | 37 | 109 |
| 2021 Enero | 40 | 27 | 67 |
| 2020 Diciembre | 41 | 24 | 65 |
| 2020 Noviembre | 40 | 21 | 61 |
| 2020 Octubre | 37 | 19 | 56 |
| 2020 Septiembre | 38 | 18 | 56 |
| 2020 Agosto | 24 | 26 | 50 |
| 2020 Julio | 28 | 21 | 49 |
| 2020 Junio | 42 | 37 | 79 |
| 2020 Mayo | 32 | 9 | 41 |
| 2020 Abril | 23 | 13 | 36 |
| 2020 Marzo | 17 | 11 | 28 |
| 2020 Febrero | 5 | 0 | 5 |
| 2019 Diciembre | 5 | 0 | 5 |
| 2019 Septiembre | 1 | 0 | 1 |
| 2019 Julio | 4 | 0 | 4 |
| 2019 Mayo | 2 | 0 | 2 |
| 2019 Abril | 3 | 3 | 6 |
| 2019 Febrero | 2 | 0 | 2 |
| 2019 Enero | 1 | 0 | 1 |
| 2018 Diciembre | 4 | 0 | 4 |
| 2018 Noviembre | 3 | 0 | 3 |
| 2018 Octubre | 1 | 0 | 1 |
| 2018 Septiembre | 6 | 0 | 6 |
| 2018 Abril | 0 | 1 | 1 |
| 2018 Febrero | 13 | 4 | 17 |
| 2018 Enero | 37 | 5 | 42 |
| 2017 Diciembre | 39 | 5 | 44 |
| 2017 Noviembre | 27 | 3 | 30 |
| 2017 Octubre | 23 | 7 | 30 |
| 2017 Septiembre | 21 | 33 | 54 |
| 2017 Agosto | 29 | 28 | 57 |
| 2017 Julio | 21 | 11 | 32 |
| 2017 Junio | 31 | 9 | 40 |
| 2017 Mayo | 28 | 13 | 41 |
| 2017 Abril | 51 | 36 | 87 |
| 2017 Marzo | 21 | 26 | 47 |
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