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What is Lichen planus pemphigoides? A highlight of three cases with discussion of differential diagnosis and suggestion of simple classification guidelines
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(D) Deposição linear de C3 ao longo da zona da membrana basal epidérmica.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Reed Maggard, Donna A. Culton, Amy Blake, Paul Googe, Jayson Miedema" "autores" => array:5 [ 0 => array:2 [ "nombre" => "Reed" "apellidos" => "Maggard" ] 1 => array:2 [ "nombre" => "Donna A." 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A highlight of three cases with discussion of differential diagnosis and suggestion of simple classification guidelines" "tieneTextoCompleto" => true "saludo" => "Dear Editor," "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "378" "paginaFinal" => "381" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Reed Maggard, Donna A. Culton, Amy Blake, Paul Googe, Jayson Miedema" "autores" => array:5 [ 0 => array:3 [ "nombre" => "Reed" "apellidos" => "Maggard" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 1 => array:3 [ "nombre" => "Donna A." "apellidos" => "Culton" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "Amy" "apellidos" => "Blake" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "Paul" "apellidos" => "Googe" "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] ] 4 => array:4 [ "nombre" => "Jayson" "apellidos" => "Miedema" "email" => array:1 [ 0 => "jayson_miedema@med.unc.edu" ] "referencia" => array:3 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] 2 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] ] "afiliaciones" => array:3 [ 0 => array:3 [ "entidad" => "University of Washington School of Medicine, Seattle, WA, USA" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Department of Dermatology, University of North Carolina at Chapel Hill, Chapel Hill, NC, USA" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Department of Pathology, University of North Carolina at Chapel Hill, Chapel Hill, NC, USA" "etiqueta" => "c" "identificador" => "aff0015" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 1304 "Ancho" => 2508 "Tamanyo" => 435163 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0015" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Case 3. (A) Scaly lichenoid and bullous lesions on wrists (B) abdomen.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Cutaneous autoimmune disorders exist on a biological spectrum. A conceptually challenging condition is <span class="elsevierStyleItalic">Lichen Planus Pemphigoides</span> (LPP), cases of which appear to share features of bullous pemphigoid and lichen planus. Herein we present three recent cases and emphasize classification as LPP can be made using clinical features in conjunction with histological and immunofluorescence findings. More specifically, classification as LPP can be made in the context of 1) Lichenoid lesions clinically and histologically, 2) Linear staining along the basement membrane zone (BMZ) of IgG and/or C3 on immunofluorescence studies, and 3) Lack of evidence to support another specific diagnosis.</p><p id="par0010" class="elsevierStylePara elsevierViewall">Clinical descriptions of LPP commonly include lichen planus-like lesions with the additional finding of tense blisters and bullae.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> The histology is said to be lichen planus-like. Positive immunofluorescence showing deposition along the dermal-epidermal junction is considered a sine qua non-feature. A number of studies have found the autoantigen to be directed against the NC16A subdomain of collagen XVII (BP180).<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> However, significant heterogeneity in specific target antigen(s) has been documented.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3–6</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">Classification criteria are used to help group conditions for the study.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> They are not meant to serve as diagnostic criteria but are often used at a practical level by emphasizing important disease features. Notably, because diagnostic criteria are limited by inherent sensitivity and specificity characteristics, classification criteria are published by the American College of Rheumatology, whereas diagnostic criteria are not. Given the historic controversy associated with LPP, this is a disease for which classification criteria-like guidelines would be clinically useful.<span class="elsevierStyleEnunciation" id="enun0005"><span class="elsevierStyleLabel">Case 1</span><p id="par0020" class="elsevierStylePara elsevierViewall">was a 55-year-old male with untreated colonic adenocarcinoma who presented with a pruritic rash consisting of violaceous scaly papules and plaques involving the extremities and trunk for several months with more recent blistering (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A‒B). Biopsy of a representative lichenoid lesion revealed a brisk lichenoid interface dermatitis histologically consistent with lichen planus (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>C). Perilesional biopsy for Direct Immunofluorescence (DIF) revealed linear C3 deposition without accompanying IgG (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>D), cytoid bodies and shaggy, fibrillar fibrinogen deposition at the BMZ. The patient improved on prednisone, without recurrence after the taper.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia></span><span class="elsevierStyleEnunciation" id="enun0010"><span class="elsevierStyleLabel">Case 2</span><p id="par0025" class="elsevierStylePara elsevierViewall">was a 29-year-old female who presented with a 6-month history of lichenoid papules and plaques, presumptively diagnosed as lichen planus. At presentation, she was also noted to have scattered vesicles and bullae overlying unaffected and lichenoid skin, distributed on the extremities and trunk (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>A‒B). The autoantibody workup was negative, and no systemic symptoms were present. A biopsy revealed a robust interface process with features of lichen planus. However, in contrast to typical LP the epidermis was completely detached from the underlying dermis, forming a blistering space histologically (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>C). DIF showed faint linear IgG, intense linear deposition of C3 (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>D), and fibrinogen along the BMZ with junctional cytoid bodies. She was started on prednisone and methotrexate with good disease control at follow-up.</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia></span><span class="elsevierStyleEnunciation" id="enun0015"><span class="elsevierStyleLabel">Case 3</span><p id="par0030" class="elsevierStylePara elsevierViewall">was a 68-year-old female who presented with a 2-month history of a scaly pruritic rash. Outside biopsy at the onset of her rash revealed a band-like infiltrate of lymphocytes in the papillary dermis with vacuolar change and necrotic keratinocytes, histologically consistent with lichen planus. One week prior to their presentation in our clinic she additionally developed bullae over the upper extremities and trunk (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>A‒B). DIF revealed linear C3 deposition at the BMZ without IgG. Indirect immunofluorescence was positive for linear IgG staining localizing to the epidermal side of salt-split skin at a titer of 1:1280. She was treated with triamcinolone wraps, prednisone, and intravenous immunoglobulin with improvement.</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia></span></p><p id="par0035" class="elsevierStylePara elsevierViewall">In all three cases, we were able to exclude traditional lichen planus because of the immunofluorescence findings. Traditional bullous pemphigoid was excluded because of lichenoid histological and clinical findings. None of the cases had other evidence of lupus or bullous lupus erythematosus.</p><p id="par0040" class="elsevierStylePara elsevierViewall">Autoimmune disorders exist on a biological spectrum and often no solitary test has absolute specificity. In the past, authors have noted cases that share features of both lichen planus and bullous pemphigoid and have termed these cases <span class="elsevierStyleItalic">lichen planus pemphigoid</span>.</p><p id="par0045" class="elsevierStylePara elsevierViewall">In accordance with our concept of the disease and review of the literature, we emphasize the following classification guidelines: 1) Lichenoid lesions clinically and histologically, precluding classification as routine lichen planus or bullous pemphigoid; 2) Linear staining along the BMZ of IgG and/or C3 on immunofluorescence studies; 3) Lack of diagnostic findings for a separate specific diagnosis, such as lupus erythematosus.</p><p id="par0050" class="elsevierStylePara elsevierViewall">Notably, the above omits the need for more exotic ancillary testing, such as enzyme-linked immunosorbent assay or antigen subtyping.</p><p id="par0055" class="elsevierStylePara elsevierViewall">We choose the term “classification guidelines” in an attempt to parallel other conditions in rheumatology. Challenges in classification are reflected in the American College of Rheumatology’s publication of classification criteria, based on the recognition that rheumatic diseases “tend to be heterogenous in their presentation, course, and outcome and not have a single clinical, laboratory, pathologic, or radiologic feature that could serve as a “gold standard….”<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">LPP is not a commonly made diagnosis. Zaraa et al. published the largest case review.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> They conceptualized the cases as a combination between lichen planus and bullous pemphigoid and emphasize the importance of clinical and histological correlation. Hubner et al. also underscored the importance of careful clinicopathological correlation, especially to exclude other entities.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">When presented with similar cases, some clinicians may experience confusion in nosological classification. However, we feel the emphasis of these criteria may help clinicians feel confident in the appropriateness of a diagnosis of lichen planus pemphigoid, when these minimum criteria are satisfied.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Financial support</span><p id="par0070" class="elsevierStylePara elsevierViewall">None declared.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Authors’ contributions</span><p id="par0075" class="elsevierStylePara elsevierViewall">Reed Maggard: Approval of the final manuscript version; critical literature review; manuscript critical review; preparation and writing of manuscript.</p><p id="par0080" class="elsevierStylePara elsevierViewall">Donna A. Culton: Approval of the final manuscript version; critical literature review; data collection, analysis and interpretation; intellection participation and/or therapeutic management of studied cases; manuscript critical review; study concept and planning.</p><p id="par0085" class="elsevierStylePara elsevierViewall">Amy Blake: Approval of final version of the manuscript; intellection participation and/or therapeutic management of studied cases; data collection, analysis and interpretation.</p><p id="par0090" class="elsevierStylePara elsevierViewall">Paul Googe: Approval of the final version of the manuscript; data collection analysis and interpretation; intellectual participation and/or therapeutic management of studied cases.</p><p id="par0095" class="elsevierStylePara elsevierViewall">Jayson Miedema: Approval of the final version of the manuscript; critical literature review; intellectual participation and/or therapeutic management of studied cases; preparation and writing of manuscript; manuscript critical review.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflicts of interest</span><p id="par0100" class="elsevierStylePara elsevierViewall">None declared.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:4 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Financial support" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Authors’ contributions" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Conflicts of interest" ] 3 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2022-07-11" "fechaAceptado" => "2022-08-05" "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "⋆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Study conducted at the University of North Carolina at Chapel Hill; Chapel Hill, North Carolina, United States of America.</p>" ] ] "multimedia" => array:3 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 2150 "Ancho" => 2508 "Tamanyo" => 691053 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Case 1. (A) Scaly papules and plaques on abdomen. (B) Blistering. (C) Brisk lichenoid interface dermatitis. (D) Linear C3 deposition at the BMZ.</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" => 2296 "Ancho" => 2508 "Tamanyo" => 756149 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Case 2. (A) Lichenoid lesions with scattered vesicles on hands and (B) thighs. (C) An interface process histologically. (D) Intense linear deposition of C3 along BMZ.</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" => 1304 "Ancho" => 2508 "Tamanyo" => 435163 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0015" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Case 3. 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