Case and research letter
Cobblestone mouth: An exuberant oral presentation of pemphigus vegetans
Una boca en empedrado: una presentación oral exuberante de pénfigo vegetante
P. Mendes-Bastos
, C. Amaro, C. Fernandes
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Department of Dermatology and Venereology, Hospital de Curry Cabral, Centro Hospitalar de Lisboa Central, Portugal
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Mendes-Bastos, C. Amaro, C. Fernandes" "autores" => array:3 [ 0 => array:4 [ "nombre" => "P." "apellidos" => "Mendes-Bastos" "email" => array:1 [ 0 => "Pmendesbastos@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "C." "apellidos" => "Amaro" ] 2 => array:2 [ "nombre" => "C." "apellidos" => "Fernandes" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Department of Dermatology and Venereology, Hospital de Curry Cabral, Centro Hospitalar de Lisboa Central, Portugal" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Una boca en empedrado: una presentación oral exuberante de pénfigo vegetante" ] ] "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" => 1500 "Ancho" => 602 "Tamanyo" => 187184 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A, B) Painful white verrucous papules, fissures, erosions, and crusts on the vermillion border of the lips and the hard palate (cobblestone pattern); sulci and gyri on the dorsum of the tongue (cerebriform tongue). © Oozing, raised erythematous plaques with a verrucous surface on the inguinal folds.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">A 45-year-old woman presented with a 4-month history of painful erosions on the oral mucosa and lips, and reported a weight loss of 12<span class="elsevierStyleHsp" style=""></span>kg due to difficulties in eating. Over the past 2 months she had noticed painful exudative erosions on the inguinal folds and perineum as well as erythematous conjunctiva and easy tearing. She had been prescribed several systemic and topical medications, but her condition had progressively worsened.</p><p id="par0010" class="elsevierStylePara elsevierViewall">On physical examination, the surfaces of the vermillion border of the lips, the gums, and the hard palate had multiple white verrucous papules, fissures, erosions, and crusts forming a cobblestone pattern. The dorsum of the tongue had sulci and gyri that gave it a cerebriform appearance (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A and B). On the perineum, a well-demarcated erosion with a whitish, exudative surface was seen, in addition to painful, oozing, raised erythematous plaques with a verrucous surface on the inguinal folds (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>C) and right axilla. The complete skin examination also revealed two small flaccid blisters and an erosion on the back and a superficial erosion on the left lower tarsal conjunctiva.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Histological examination of one of the flaccid blisters on the back revealed an intraepidermal blister and suprabasal acantholysis (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>A), as well as a dense epidermal and dermal inflammatory infiltrate, rich in neutrophils and eosinophils (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>B). Histological examination of the left inguinal vegetating plaque showed identical findings, in addition to pseudoepitheliomatous epidermal hyperplasia (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>C). Direct immunofluorescence of perilesional skin showed intercellular staining of the epidermis by immunoglobulin (Ig) G deposits (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>D).</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">A diagnosis of pemphigus vegetans was established. The patient was treated with systemic prednisolone 60<span class="elsevierStyleHsp" style=""></span>mg/d (1 mg/kg/day) with a slow taper, azathioprine 150<span class="elsevierStyleHsp" style=""></span>mg/d, and topical tacrolimus 1<span class="elsevierStyleHsp" style=""></span>mg/g ointment on the lips. The cutaneous and mucosal lesions regressed completely in two months and the patient was kept on azathioprine 150<span class="elsevierStyleHsp" style=""></span>mg/d.</p><p id="par0025" class="elsevierStylePara elsevierViewall">Pemphigus vegetans is a rare variant of pemphigus vulgaris, and is thought to represent a reactive response of the skin to the autoimmune insult of pemphigus vulgaris.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> It is characterized by flaccid blisters that erupt into erosions and eventually form papillomatous vegetations, especially in intertriginous areas and on the scalp or face.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a> Involvement of the vermilion border of the lips is a clinical hallmark of oral involvement.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> The tongue may show cerebriform-like changes.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> The exuberant cobblestone pattern of the lips and oral mucosa associated with marked weight loss in the current case highlights the importance of oral manifestations of pemphigus vegetans.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Suprabasal acantholysis is an early histopathological finding in pemphigus vulgaris and pemphigus vegetans. With time, the lesions acquire a verrucous and hyperkeratotic appearance and biopsy samples from pemphigus vegetans may exhibit epidermal hyperplasia, papillomatosis, and intraepidermal eosinophilic abscesses.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,4</span></a> Presence of an eosinophilic response, formation of microabscesses, and extent of vesiculation have been proposed as possible histopathological features for distinguishing pemphigus vegetans from pemphigus vulgaris.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Direct immunofluorescence usually shows deposits of IgG and possibly C3 on the surface of keratinocytes. Circulating intercellular antibodies against desmogleins, generally of the IgG class, are detected in most patients by indirect immunofluorescence.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> The patient described in this report had elevated titers of both anti-desmoglein 3 (>200<span class="elsevierStyleHsp" style=""></span>RU/mL) and anti-desmoglein 1 antibodies (142<span class="elsevierStyleHsp" style=""></span>RU/mL).</p><p id="par0040" class="elsevierStylePara elsevierViewall">The clinical differential diagnosis should include Hayley–Hayley disease, IgA pemphigus, and paraneoplastic pemphigus,<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> while the histological differential diagnosis should include pemphigus vulgaris, Hayley–Hayley disease, Darier disease, and Grover disease. Pyodermatitis-pyostomatitis vegetans can clinically and histopathologically resemble pemphigus vegetans and only indirect and direct immunofluorescence studies can accurately distinguish between these two entities.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "multimedia" => array:2 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1500 "Ancho" => 602 "Tamanyo" => 187184 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A, B) Painful white verrucous papules, fissures, erosions, and crusts on the vermillion border of the lips and the hard palate (cobblestone pattern); sulci and gyri on the dorsum of the tongue (cerebriform tongue). © Oozing, raised erythematous plaques with a verrucous surface on the inguinal folds.</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" => 1927 "Ancho" => 750 "Tamanyo" => 389519 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">(A) Intraepidermal blister and a dense epidermal and dermal inflammatory infiltrate (hematoxylin–eosin, original magnification, ×10). (B) Suprabasal acantholysis and inflammatory infiltrate, rich in neutrophils and eosinophils (hematoxylin–eosin, original magnification, ×40). (C) Pseudoepitheliomatous epidermal hyperplasia and dense inflammatory dermal infiltrate (hematoxylin–eosin, original magnification, ×10). (D) Direct immunofluorescence showing immunoglobulin G intercellular epidermal deposits.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:6 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Pemphigus" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:1 [ 0 => "M. Amagai" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "LibroEditado" => array:5 [ "titulo" => "Dermatology" "paginaInicial" => "461" "paginaFinal" => "474" "edicion" => "3rd ed." 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| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 8 | 12 | 20 |
| 2024 Octubre | 65 | 68 | 133 |
| 2024 Septiembre | 63 | 56 | 119 |
| 2024 Agosto | 75 | 81 | 156 |
| 2024 Julio | 69 | 65 | 134 |
| 2024 Junio | 70 | 58 | 128 |
| 2024 Mayo | 99 | 65 | 164 |
| 2024 Abril | 67 | 41 | 108 |
| 2024 Marzo | 59 | 40 | 99 |
| 2024 Febrero | 59 | 50 | 109 |
| 2024 Enero | 61 | 42 | 103 |
| 2023 Diciembre | 67 | 48 | 115 |
| 2023 Noviembre | 68 | 40 | 108 |
| 2023 Octubre | 68 | 35 | 103 |
| 2023 Septiembre | 56 | 49 | 105 |
| 2023 Agosto | 53 | 35 | 88 |
| 2023 Julio | 78 | 62 | 140 |
| 2023 Junio | 54 | 54 | 108 |
| 2023 Mayo | 78 | 60 | 138 |
| 2023 Abril | 81 | 33 | 114 |
| 2023 Marzo | 63 | 40 | 103 |
| 2023 Febrero | 69 | 24 | 93 |
| 2023 Enero | 65 | 39 | 104 |
| 2022 Diciembre | 90 | 40 | 130 |
| 2022 Noviembre | 87 | 38 | 125 |
| 2022 Octubre | 73 | 52 | 125 |
| 2022 Septiembre | 56 | 56 | 112 |
| 2022 Agosto | 51 | 55 | 106 |
| 2022 Julio | 36 | 52 | 88 |
| 2022 Junio | 41 | 43 | 84 |
| 2022 Mayo | 94 | 35 | 129 |
| 2022 Abril | 90 | 55 | 145 |
| 2022 Marzo | 93 | 56 | 149 |
| 2022 Febrero | 91 | 36 | 127 |
| 2022 Enero | 107 | 67 | 174 |
| 2021 Diciembre | 85 | 43 | 128 |
| 2021 Noviembre | 60 | 53 | 113 |
| 2021 Octubre | 111 | 68 | 179 |
| 2021 Septiembre | 77 | 47 | 124 |
| 2021 Agosto | 107 | 36 | 143 |
| 2021 Julio | 114 | 40 | 154 |
| 2021 Junio | 79 | 35 | 114 |
| 2021 Mayo | 61 | 36 | 97 |
| 2021 Abril | 155 | 77 | 232 |
| 2021 Marzo | 113 | 23 | 136 |
| 2021 Febrero | 93 | 34 | 127 |
| 2021 Enero | 95 | 20 | 115 |
| 2020 Diciembre | 103 | 24 | 127 |
| 2020 Noviembre | 62 | 14 | 76 |
| 2020 Octubre | 53 | 8 | 61 |
| 2020 Septiembre | 77 | 14 | 91 |
| 2020 Agosto | 65 | 27 | 92 |
| 2020 Julio | 51 | 16 | 67 |
| 2020 Junio | 71 | 25 | 96 |
| 2020 Mayo | 42 | 26 | 68 |
| 2020 Abril | 33 | 25 | 58 |
| 2020 Marzo | 28 | 27 | 55 |
| 2020 Febrero | 3 | 11 | 14 |
| 2020 Enero | 0 | 12 | 12 |
| 2019 Diciembre | 4 | 16 | 20 |
| 2019 Noviembre | 0 | 17 | 17 |
| 2019 Octubre | 0 | 17 | 17 |
| 2019 Septiembre | 0 | 15 | 15 |
| 2019 Agosto | 0 | 10 | 10 |
| 2019 Julio | 4 | 34 | 38 |
| 2019 Junio | 0 | 23 | 23 |
| 2019 Mayo | 3 | 36 | 39 |
| 2019 Abril | 2 | 36 | 38 |
| 2019 Marzo | 0 | 5 | 5 |
| 2019 Febrero | 2 | 10 | 12 |
| 2019 Enero | 1 | 19 | 20 |
| 2018 Diciembre | 0 | 15 | 15 |
| 2018 Noviembre | 3 | 3 | 6 |
| 2018 Octubre | 0 | 1 | 1 |
| 2018 Septiembre | 4 | 4 | 8 |
| 2018 Agosto | 0 | 17 | 17 |
| 2018 Julio | 0 | 10 | 10 |
| 2018 Junio | 0 | 11 | 11 |
| 2018 Mayo | 1 | 12 | 13 |
| 2018 Abril | 0 | 6 | 6 |
| 2018 Marzo | 5 | 5 | 10 |
| 2018 Febrero | 77 | 3 | 80 |
| 2018 Enero | 100 | 8 | 108 |
| 2017 Diciembre | 62 | 9 | 71 |
| 2017 Noviembre | 41 | 11 | 52 |
| 2017 Octubre | 27 | 7 | 34 |
| 2017 Septiembre | 26 | 14 | 40 |
| 2017 Agosto | 28 | 18 | 46 |
| 2017 Julio | 25 | 13 | 38 |
| 2017 Junio | 37 | 24 | 61 |
| 2017 Mayo | 27 | 17 | 44 |
| 2017 Abril | 27 | 8 | 35 |
| 2017 Marzo | 16 | 35 | 51 |
| 2017 Febrero | 16 | 12 | 28 |
| 2017 Enero | 19 | 14 | 33 |
| 2016 Diciembre | 7 | 14 | 21 |
| 2016 Noviembre | 11 | 19 | 30 |
| 2016 Octubre | 11 | 14 | 25 |
| 2016 Septiembre | 0 | 12 | 12 |
| 2016 Agosto | 0 | 10 | 10 |
| 2016 Julio | 4 | 3 | 7 |
| 2016 Junio | 15 | 1 | 16 |
| 2016 Mayo | 11 | 2 | 13 |
| 2016 Abril | 18 | 4 | 22 |
| 2016 Marzo | 18 | 3 | 21 |
| 2016 Febrero | 18 | 3 | 21 |
| 2016 Enero | 17 | 2 | 19 |
| 2015 Diciembre | 12 | 13 | 25 |
| 2015 Noviembre | 18 | 1 | 19 |
| 2015 Octubre | 21 | 3 | 24 |
| 2015 Septiembre | 15 | 1 | 16 |
| 2015 Agosto | 12 | 5 | 17 |
| 2015 Julio | 139 | 0 | 139 |
| 2015 Junio | 51 | 0 | 51 |
| 2015 Mayo | 30 | 4 | 34 |
| 2015 Abril | 64 | 5 | 69 |
| 2015 Marzo | 27 | 3 | 30 |
| 2015 Febrero | 37 | 4 | 41 |
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