What is your diagnosis?
Case for diagnosis. Radiotherapy-induced pemphigus vegetans
Hiram Larangeira de Almeida Jra,
, Antônia Larangeira de Almeidab, Pedro Henrique Evangelista Martinezb, Ana Letícia Boffc
Corresponding author
a Pós-Graduação em Saúde e Comportamento, Universidade Católica de Pelotas, Departamento de Dermatologia, Universidade Federal de Pelotas, Pelotas, RS, Brazil
b Liga de Dermatologia, Universidade Federal de Pelotas, Pelotas, RS, Brazil
c Laboratório Dermapat and Santa Casa de Misericórdia de Porto Alegre, Porto Alegre, RS, Brazil
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He was treated with two cycles of 5-fluorouracil and cisplatin and concomitant radiotherapy (6 MeV linear photon accelerator, total dose of 54 Gy), with complete remission. The control colonoscopy and endoscopy after six months of treatment were normal. Forty-five days before coming to the clinic again, he had a painful perianal and intergluteal erosion with circinate pustular borders and some isolated pustules, with surrounding erythema (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). He was hospitalized and received systemic antibiotics (ceftriaxone and clindamycin) and famciclovir. A computed tomography on admission ruled out tumor recurrence, showing a normal anal canal. An incisional biopsy was performed, which showed pseudoepitheliomatous hyperplasia with microabscesses of inflammatory cells (neutrophils and eosinophils) (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). In some microscopic fields, clefts with isolated keratinocytes next to microabscesses were observed (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">What is your diagnosis?</span><p id="par0010" class="elsevierStylePara elsevierViewall"><ul class="elsevierStyleList" id="lis0005"><li class="elsevierStyleListItem" id="lsti0005"><span class="elsevierStyleLabel">a)</span><p id="par0015" class="elsevierStylePara elsevierViewall">Ulcerated herpes simplex associated with immunosuppression</p></li><li class="elsevierStyleListItem" id="lsti0010"><span class="elsevierStyleLabel">b)</span><p id="par0020" class="elsevierStylePara elsevierViewall">Ulcerated radiodermatitis</p></li><li class="elsevierStyleListItem" id="lsti0015"><span class="elsevierStyleLabel">c)</span><p id="par0025" class="elsevierStylePara elsevierViewall">Pemphigus vegetans</p></li><li class="elsevierStyleListItem" id="lsti0020"><span class="elsevierStyleLabel">d)</span><p id="par0030" class="elsevierStylePara elsevierViewall">Tinea associated with immunosuppression</p></li></ul></p><p id="par0035" class="elsevierStylePara elsevierViewall">The histopathological findings were suggestive of pemphigus vegetans, which may present with acantholytic clefts associated with polymorphonuclear microabscesses,<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> unlike classic pemphigus vulgaris in which the cleavage is accompanied by little inflammation.</p><p id="par0040" class="elsevierStylePara elsevierViewall">There was no therapeutic response to systemic antibiotics and antivirals initiated before the results of the investigation, and there was significant regression with oral prednisone (40 mg) and topical betamethasone. After seven days, the pustular border disappeared, and there was total symptom regression, with the appearance of some hyperkeratotic and verrucous central areas (<a class="elsevierStyleCrossRef" href="#fig0020">Fig. 4</a>). Later, the result of indirect immunofluorescence was positive for IgG, with an intercellular pattern, 1:80 titer.</p><elsevierMultimedia ident="fig0020"></elsevierMultimedia><p id="par0045" class="elsevierStylePara elsevierViewall">There are several reports of radiotherapy-induced pemphigus vulgaris, but not of the vegetans variant. Reported cases are restricted to the irradiated area,<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,3</span></a> (similarly to the case described here) or generalized.<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4,5</span></a> There have also been reports of pemphigus foliaceus induction.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">The mechanism of induction of autoimmune acantholytic diseases by radiotherapy must be complex. One possibility is the antigenic alteration by radiation;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> another possibility would be the alteration of the immune response. Corroborating the hypothesis of antigenic alteration, there are reports of laboratory investigation with immunoblotting demonstrating antibodies against antigens other than desmogleins.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">This case demonstrates the possibility that pemphigus vegetans lesions may occur locally on a previously irradiated area.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect1015">Financial support</span><p id="par0060" class="elsevierStylePara elsevierViewall">None declared.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Authors’ contributions</span><p id="par0065" class="elsevierStylePara elsevierViewall">Hiram Larangeira de Almeida Jr.: Approval of the final version of the manuscript; design and planning of the study; drafting and editing of the manuscript; collection, analysis, and interpretation of data; intellectual participation in the propaedeutic and/or therapeutic conduct of the studied cases; critical review of the literature; critical review of the manuscript.</p><p id="par0070" class="elsevierStylePara elsevierViewall">Antônia Larangeira de Almeida: Approval of the final version of the manuscript; design and planning of the study; drafting and editing of the manuscript; collection, analysis, and interpretation of data; intellectual participation in the propaedeutic and/or therapeutic conduct of the studied cases; critical review of the literature; critical review of the manuscript.</p><p id="par0075" class="elsevierStylePara elsevierViewall">Pedro Henrique Evangelista Martinez: Approval of the final version of the manuscript; design and planning of the study; drafting and editing of the manuscript; collection, analysis, and interpretation of data; intellectual participation in propaedeutic and/or therapeutic conduct of the studied cases; critical review of the literature; critical review of the manuscript.</p><p id="par0080" class="elsevierStylePara elsevierViewall">Ana Letícia Boff: Approval of the final version of the manuscript; design and planning of the study; drafting and editing of the manuscript; collection, analysis, and interpretation of data; critical review of the literature; critical review of the manuscript.</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Conflicts of interest</span><p id="par0085" class="elsevierStylePara elsevierViewall">None declared.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:8 [ 0 => array:3 [ "identificador" => "xres1711211" "titulo" => "Abstract" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0005" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec1513461" "titulo" => "Keywords" ] 2 => array:2 [ "identificador" => "sec0005" "titulo" => "Case report" ] 3 => array:2 [ "identificador" => "sec0010" "titulo" => "What is your diagnosis?" ] 4 => array:2 [ "identificador" => "sec0015" "titulo" => "Financial support" ] 5 => array:2 [ "identificador" => "sec0020" "titulo" => "Authors’ contributions" ] 6 => array:2 [ "identificador" => "sec0025" "titulo" => "Conflicts of interest" ] 7 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2021-07-16" "fechaAceptado" => "2021-09-13" "PalabrasClave" => array:1 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec1513461" "palabras" => array:2 [ 0 => "Radiotherapy" 1 => "Pemphigus vegetans" ] ] ] ] "tieneResumen" => true "resumen" => array:1 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar1030" class="elsevierStyleSimplePara elsevierViewall">A 73-year-old male patient developed a poorly differentiated squamous cell carcinoma in the anal canal nine months ago. He was treated with two cycles of 5-fluorouracil and cisplatin and concomitant radiotherapy (6 MeV linear photon accelerator, total dose of 54 Gy), with complete remission. Since forty-five days he presentes a painful perianal and intergluteal erosion with circinate pustular borders. Light microscopy showed pseudoepitheliomatous hyperplasia of the epidermis with microabscesses of inflammatory cells (neutrophils and eosinophils) and acantholytic keratinocytes . Indirect immunofluorescence was positive for IgG, with an intercellular pattern, 1:80 titer. The diagnosis of radiotherapy-induced pemphigus vegetans was established and there was significant regression with oral prednisone (40 mg) and topical betamethasone.</p></span>" ] ] "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Study conducted at the Liga de Dermatologia da Universidade Federal de Pelotas, Pelotas, RS, Brazil.</p>" ] ] "multimedia" => array:4 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 611 "Ancho" => 905 "Tamanyo" => 70089 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Initial clinical aspect with perianal and intergluteal erosion with circinate pustular edges and some isolated pustules.</p>" ] ] 1 => array:8 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1606 "Ancho" => 1508 "Tamanyo" => 721987 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Light microscopy showing clefts and polymorphonuclear microabscess (Hematoxylin & eosin, ×150).</p>" ] ] 2 => array:8 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 788 "Ancho" => 905 "Tamanyo" => 251024 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0015" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Light microscopy detailing the clefts and polymorphonuclear microabscess with suprabasal cleavage (arrows) and acantholytic keratinocytes (arrowheads); (Hematoxylin & eosin, ×400).</p>" ] ] 3 => array:8 [ "identificador" => "fig0020" "etiqueta" => "Figure 4" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr4.jpeg" "Alto" => 669 "Ancho" => 905 "Tamanyo" => 117476 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0020" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Clinical aspect with significant improvement after seven days of treatment, without the circinate pustular borders and mild verrucosity in the center.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:7 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Pemphigus vegetans associated with verrucous lesions: expanding a phenotype" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "H.L. 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