Case and Research Letters
Papular Acantholytic Dyskeratosis of the Vulva in a Woman With Benign Familial Pemphigus
Disqueratosis acantolítica papular de la vulva en una mujer con pénfigo benigno familiar
Servicio de Dermatología, Hospital Clínic, Universitat de Barcelona, Barcelona, Spain
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Baliu-Piqué, P. Iranzo" "autores" => array:2 [ 0 => array:4 [ "nombre" => "C." "apellidos" => "Baliu-Piqué" "email" => array:1 [ 0 => "carola.baliu.pique@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "P." "apellidos" => "Iranzo" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Servicio de Dermatología, Hospital Clínic, Universitat de Barcelona, Barcelona, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Disqueratosis acantolítica papular de la vulva en una mujer con pénfigo benigno familiar" ] ] "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" => 1067 "Ancho" => 800 "Tamanyo" => 249742 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Punch biopsy of a vulvar papule showing an acantholytic intraepidermal papule with dyskeratosis. Hematoxylin and eosin, original magnification ×20.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">We describe the case of a woman aged 50 years, with a history of systemic hypertension on treatment and benign familial pemphigus (BFP) that had arisen more than 10 years earlier. During those years, she had presented recurrent outbreaks of erosive lesions in the groin, perianal region, and submammary folds (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>), often with bacterial superinfection. She had received topical therapy with corticosteroids, antibiotics, tacrolimus, and diclofenac, with a partial response. She had also received systemic acitretin at a dose of 25<span class="elsevierStyleHsp" style=""></span>mg/d on several occasions. During a follow-up visit, perianal lesions suggestive of PBF were observed, and asymptomatic shiny whitish papules measuring 2 to 3<span class="elsevierStyleHsp" style=""></span>mm in diameter with a cobblestone appearance on both labia majora (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). Biopsy of 1 of these vulvar lesions revealed hyperkeratosis with irregular acanthosis and suprabasal acantholysis with dyskeratosis (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>); direct immunofluorescence was negative.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">Papular acantholytic dyskeratosis (PAD) of the vulva, first described in 1984 by Chorzelski et al.,<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">1</span></a> mainly affects young women, typically on the labia majora, although it has also been described on the penis in men, in the anal canal, and in the inguinal folds.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">2,3</span></a> It usually presents as multiple whitish papules that may coalesce to form plaques; solitary papules are rare. Most commonly it is asymptomatic, although intense pruritus can occur. Histopathology reveals acantholysis with varying degrees of dyskeratosis, and direct and indirect immunofluorescence are negative in almost all cases studied.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">4</span></a> Though initially considered an independent entity, its relationship with Darier disease (DD) and with BFP has been under discussion due to the histologic similarity between the diseases.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">5</span></a> The etiology of PAD is unclear. The majority of cases are sporadic, but there have been reports in recent years of cases of DAP due to mutations in genes <span class="elsevierStyleItalic">ATP2A2</span><a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">6</span></a> and <span class="elsevierStyleItalic">ATP2C1</span>,<a class="elsevierStyleCrossRefs" href="#bib0080"><span class="elsevierStyleSup">7–9</span></a> which are mutations known in DD and BFP, respectively. Pernet et al.<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">8</span></a> reported a familial case of PAD in which the same mutation in <span class="elsevierStyleItalic">ATP2C1</span> was detected in the mother and daughter, suggesting that PAD is allelic with BFP. Yu et al.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">9</span></a> published the case of a woman with PAD and a family history of BFP; those authors suggested the possibility that a patient with PAD may subsequently develop lesions of BFP and that the 2 diseases belonged to a common spectrum of genetically linked acantholytic dyskeratotic dermatoses. Genetic analysis remains to be performed in our patient, but the coexistence of PAD and BFP in the same patient supports this hypothesis.</p><p id="par0015" class="elsevierStylePara elsevierViewall">We have described a patient with the simultaneous presence of lesions clinically and histologically typical of BFP and PAD; the study of mutations in both lesions remains to be performed. To date, we have found no case reports in the literature describing the coexistence of BFP and PAD in a single patient.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of Interest</span><p id="par0020" 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 "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Baliu-Piqué C, Iranzo P. Disqueratosis acantolítica papular de la vulva en una mujer con pénfigo benigno familiar. Actas Dermosifiliogr. 2017;108:78–79.</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" => 1030 "Ancho" => 1583 "Tamanyo" => 238952 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Erosive plaque in the submammary region, compatible with benign familial pemphigus.</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" => 1777 "Ancho" => 1333 "Tamanyo" => 583932 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Multiple shiny whitish papules measuring 2 to 3<span class="elsevierStyleHsp" style=""></span>mm in diameter with a cobblestone appearance on the labia majora.</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" => 1067 "Ancho" => 800 "Tamanyo" => 249742 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Punch biopsy of a vulvar papule showing an acantholytic intraepidermal papule with dyskeratosis. Hematoxylin and eosin, original magnification ×20.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:9 [ 0 => array:3 [ "identificador" => "bib0050" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Papular acantholytic dyskeratosis of the vulva a new entity?" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "T.P. Chorzelski" 1 => "J. Kudejko" 2 => "S. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 14 | 13 | 27 |
| 2024 October | 83 | 43 | 126 |
| 2024 September | 90 | 37 | 127 |
| 2024 August | 112 | 54 | 166 |
| 2024 July | 114 | 43 | 157 |
| 2024 June | 91 | 32 | 123 |
| 2024 May | 83 | 46 | 129 |
| 2024 April | 88 | 33 | 121 |
| 2024 March | 104 | 43 | 147 |
| 2024 February | 82 | 34 | 116 |
| 2024 January | 65 | 42 | 107 |
| 2023 December | 77 | 23 | 100 |
| 2023 November | 80 | 32 | 112 |
| 2023 October | 83 | 31 | 114 |
| 2023 September | 73 | 37 | 110 |
| 2023 August | 53 | 27 | 80 |
| 2023 July | 66 | 47 | 113 |
| 2023 June | 59 | 28 | 87 |
| 2023 May | 56 | 32 | 88 |
| 2023 April | 65 | 33 | 98 |
| 2023 March | 67 | 47 | 114 |
| 2023 February | 58 | 33 | 91 |
| 2023 January | 48 | 35 | 83 |
| 2022 December | 65 | 37 | 102 |
| 2022 November | 37 | 30 | 67 |
| 2022 October | 36 | 29 | 65 |
| 2022 September | 33 | 41 | 74 |
| 2022 August | 27 | 28 | 55 |
| 2022 July | 26 | 35 | 61 |
| 2022 June | 23 | 18 | 41 |
| 2022 May | 62 | 33 | 95 |
| 2022 April | 63 | 36 | 99 |
| 2022 March | 61 | 53 | 114 |
| 2022 February | 47 | 24 | 71 |
| 2022 January | 72 | 43 | 115 |
| 2021 December | 41 | 50 | 91 |
| 2021 November | 69 | 48 | 117 |
| 2021 October | 48 | 50 | 98 |
| 2021 September | 46 | 40 | 86 |
| 2021 August | 56 | 41 | 97 |
| 2021 July | 33 | 38 | 71 |
| 2021 June | 44 | 35 | 79 |
| 2021 May | 48 | 45 | 93 |
| 2021 April | 77 | 58 | 135 |
| 2021 March | 75 | 37 | 112 |
| 2021 February | 93 | 39 | 132 |
| 2021 January | 59 | 28 | 87 |
| 2020 December | 43 | 30 | 73 |
| 2020 November | 26 | 14 | 40 |
| 2020 October | 23 | 16 | 39 |
| 2020 September | 41 | 22 | 63 |
| 2020 August | 34 | 22 | 56 |
| 2020 July | 28 | 23 | 51 |
| 2020 June | 35 | 40 | 75 |
| 2020 May | 22 | 11 | 33 |
| 2020 April | 33 | 16 | 49 |
| 2020 March | 26 | 14 | 40 |
| 2020 February | 6 | 1 | 7 |
| 2020 January | 4 | 0 | 4 |
| 2019 December | 8 | 0 | 8 |
| 2019 November | 3 | 0 | 3 |
| 2019 September | 8 | 0 | 8 |
| 2019 August | 2 | 0 | 2 |
| 2019 July | 6 | 0 | 6 |
| 2019 June | 6 | 0 | 6 |
| 2019 May | 4 | 2 | 6 |
| 2019 April | 2 | 4 | 6 |
| 2019 March | 4 | 0 | 4 |
| 2019 February | 4 | 0 | 4 |
| 2019 January | 3 | 0 | 3 |
| 2018 December | 3 | 0 | 3 |
| 2018 November | 10 | 0 | 10 |
| 2018 October | 8 | 0 | 8 |
| 2018 September | 2 | 0 | 2 |
| 2018 February | 17 | 5 | 22 |
| 2018 January | 23 | 7 | 30 |
| 2017 December | 36 | 13 | 49 |
| 2017 November | 23 | 4 | 27 |
| 2017 October | 15 | 7 | 22 |
| 2017 September | 17 | 8 | 25 |
| 2017 August | 23 | 4 | 27 |
| 2017 July | 17 | 7 | 24 |
| 2017 June | 35 | 20 | 55 |
| 2017 May | 27 | 10 | 37 |
| 2017 April | 29 | 16 | 45 |
| 2017 March | 38 | 18 | 56 |
| 2017 February | 56 | 38 | 94 |
| 2017 January | 61 | 34 | 95 |
| 2016 December | 9 | 13 | 22 |
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