Case and Research Letter
Annular Elastolytic Giant Cell Granuloma in Sun-Protected Sites Responds to Dapsone
Granuloma elastolítico anular de células gigantes en área no fotoexpuesta con respuesta a dapsona
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Monserrat García, J.J. Pereyra Rodríguez, T. de Zulueta Dorado, J. Conejo-Mir Sánchez" "autores" => array:4 [ 0 => array:4 [ "nombre" => "M.T." "apellidos" => "Monserrat García" "email" => array:2 [ 0 => "marite_monserrat@hotmail.com" 1 => "maritemonserrat@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" => "J.J." "apellidos" => "Pereyra Rodríguez" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "T." "apellidos" => "de Zulueta Dorado" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "J." "apellidos" => "Conejo-Mir Sánchez" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Unidad de Gestión Clínica de Dermatología, Hospital Universitario Virgen del Rocío, Sevilla, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Unidad de Gestión Clínica de Anatomía Patológica, Hospital Virgen del Rocío, Sevilla, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Granuloma elastolítico anular de células gigantes en área no fotoexpuesta con respuesta a dapsona" ] ] "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" => 687 "Ancho" => 1850 "Tamanyo" => 317300 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A, Granulomatous lesion with multiple multinucleated giant cells with elastophagocytosis. Hematoxylin and eosin, original magnification<span class="elsevierStyleHsp" style=""></span>×20. B, Stain for elastic fibers, original magnification<span class="elsevierStyleHsp" style=""></span>×40.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">We present the case of a woman aged 59 years with a history of left renal agenesis. She came to outpatients with a 2-month history of persistent painful lesions on both hands. She had been treated with oral corticosteroids (deflazacort, 30<span class="elsevierStyleHsp" style=""></span>mg, in a tapering regimen) and topical methylprednisolone aceponate with no improvement. On examination, indurated red-violaceous papulonodular lesions were observed on the dorsum of the fingers and on the palms of both hands (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">Histology revealed a dermal granulomatous lesion with no areas of necrobiosis and with numerous multinucleated giant cells showing elastophagocytosis, suggestive of annular elastolytic giant cell granuloma (AEGCG) (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>, A and B).</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">After making the diagnosis and because of the lack of response to corticosteroid therapy, treatment was started with dapsone, 100<span class="elsevierStyleHsp" style=""></span>mg/d, leading to complete resolution of the lesions after 4 months of treatment, with no recurrence after a year of follow-up (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>B).</p><p id="par0020" class="elsevierStylePara elsevierViewall">AEGCG is a rare granulomatous disease characterized by the appearance of annular lesions with elevated borders formed of erythematous papules that spread centrifugally to leave an atrophic central area. Histologically, dermal granulomas formed of histiocytes and multinucleated giant cells are observed in close association with degenerated elastic fibers and elastophagocytosis. The lesions are usually asymptomatic, although mild pruritus or pain can develop, and they tend to persist for months or years, after which spontaneous remission typically occurs. The first case was described in 1975 by O’Brien,<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> who coined the term actinic granuloma as the lesions arose in sun-exposed areas. In 1979, Hanke et al.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> proposed the term annular elastolytic giant cell granuloma to describe a case of lesions of identical characteristics localized in areas not exposed to the sun; this is now the most widely accepted name for the condition.</p><p id="par0025" class="elsevierStylePara elsevierViewall">The etiology is unknown, although, based on the preference for sun-exposed areas or areas with a history of burns, and no involvement of areas in which elastic fibers are absent, such as scars and striae distensae,<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> the most widely held hypothesis is that UV radiation, the increased temperature, and other still-unknown factors can modify the antigenicity of elastic fibers, giving rise to an immune response with the formation of a granulomatous infiltrate.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">The number of lesions varies from a single lesion to widespread forms with systemic involvement.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> An association has been reported with diabetes mellitus and with solid and hematologic tumors, and some authors recommend screening for occult malignancy in these patients. Variants of the disease exist, including the papular,<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a> reticular, and exanthematous forms.<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">The differential diagnosis includes other granulomatous diseases, both infectious, due to fungi and mycobacteria, and inflammatory, such as granuloma annulare, necrobiosis lipoidica, sarcoidosis, and annular lichen planus. AEGCG can be clinically indistinguishable from any of these diseases, and histology is the gold standard for confirming the diagnosis.</p><p id="par0040" class="elsevierStylePara elsevierViewall">Little scientific evidence is available on the treatment of AEGCG, and the self-limiting nature of the disease, with a tendency to spontaneous disappearance of the lesions, makes it difficult to establish the efficacy of the different therapeutic regimens. Recommendations are based on isolated case reports or case series in which different therapeutic agents have been used with variable efficacy.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a> These agents include topical or systemic corticosteroids, topical calcineurin inhibitors, phototherapy, cryotherapy, antimalarial drugs, retinoids, ciclosporin A, tranilast, methotrexate, fumaric acid esters, pentoxifylline, clofazimine, and dapsone. We have only found 1 case report, dating from 1997, of a patient treated with dapsone with a good response.<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Dapsone (4,4-diaminodiphenyl sulfone) is a synthetic sulfone with a dual antimicrobial–anti-inflammatory mechanism of action. Since its incorporation into dermatology in the mid-twentieth century, its therapeutic indications have broadened. It is used mainly in the inflammatory neutrophilic and eosinophilic dermatoses, though there are also anecdotal reports of therapeutic success in many other entities, including AEGCG.<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a> The starting dose in adults is 50 to 100<span class="elsevierStyleHsp" style=""></span>mg/d; this can be increased to 300<span class="elsevierStyleHsp" style=""></span>mg/d to achieve the therapeutic objective, afterwards returning to the minimal effective dose.</p><p id="par0050" class="elsevierStylePara elsevierViewall">In conclusion, we have presented a case that is interesting for 2 reasons: the atypical site of the lesions in an area not exposed to the sun, and being the second reported case with an excellent response to dapsone.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of Interest</span><p id="par0055" 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: García MTM, Rodríguez JJP, de Zulueta Dorado T, Sánchez JC-M. Granuloma elastolítico anular de células gigantes en área no fotoexpuesta con respuesta a dapsona. Actas Dermosifiliogr. 2016;107:531–533.</p>" ] ] "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" => 1207 "Ancho" => 1650 "Tamanyo" => 431394 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A, Indurated, papular erythematous-violaceous lesions on the palms and volar surface of the fingers. B, Complete resolution of the lesions after treatment.</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" => 687 "Ancho" => 1850 "Tamanyo" => 317300 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A, Granulomatous lesion with multiple multinucleated giant cells with elastophagocytosis. Hematoxylin and eosin, original magnification<span class="elsevierStyleHsp" style=""></span>×20. B, Stain for elastic fibers, original magnification<span class="elsevierStyleHsp" style=""></span>×40.</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" => "Actinic granuloma" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:1 [ 0 => "J.P. 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| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 6 | 7 | 13 |
| 2024 Octubre | 83 | 37 | 120 |
| 2024 Septiembre | 98 | 24 | 122 |
| 2024 Agosto | 115 | 46 | 161 |
| 2024 Julio | 130 | 26 | 156 |
| 2024 Junio | 136 | 22 | 158 |
| 2024 Mayo | 109 | 37 | 146 |
| 2024 Abril | 98 | 28 | 126 |
| 2024 Marzo | 97 | 24 | 121 |
| 2024 Febrero | 64 | 28 | 92 |
| 2024 Enero | 94 | 32 | 126 |
| 2023 Diciembre | 68 | 24 | 92 |
| 2023 Noviembre | 74 | 26 | 100 |
| 2023 Octubre | 97 | 21 | 118 |
| 2023 Septiembre | 78 | 32 | 110 |
| 2023 Agosto | 65 | 15 | 80 |
| 2023 Julio | 89 | 36 | 125 |
| 2023 Junio | 82 | 17 | 99 |
| 2023 Mayo | 83 | 24 | 107 |
| 2023 Abril | 85 | 20 | 105 |
| 2023 Marzo | 79 | 29 | 108 |
| 2023 Febrero | 67 | 25 | 92 |
| 2023 Enero | 50 | 47 | 97 |
| 2022 Diciembre | 48 | 28 | 76 |
| 2022 Noviembre | 38 | 23 | 61 |
| 2022 Octubre | 29 | 19 | 48 |
| 2022 Septiembre | 34 | 35 | 69 |
| 2022 Agosto | 25 | 41 | 66 |
| 2022 Julio | 24 | 23 | 47 |
| 2022 Junio | 38 | 17 | 55 |
| 2022 Mayo | 80 | 35 | 115 |
| 2022 Abril | 89 | 27 | 116 |
| 2022 Marzo | 82 | 38 | 120 |
| 2022 Febrero | 77 | 26 | 103 |
| 2022 Enero | 86 | 68 | 154 |
| 2021 Diciembre | 49 | 41 | 90 |
| 2021 Noviembre | 71 | 53 | 124 |
| 2021 Octubre | 60 | 55 | 115 |
| 2021 Septiembre | 68 | 75 | 143 |
| 2021 Agosto | 72 | 23 | 95 |
| 2021 Julio | 50 | 21 | 71 |
| 2021 Junio | 46 | 32 | 78 |
| 2021 Mayo | 41 | 35 | 76 |
| 2021 Abril | 76 | 45 | 121 |
| 2021 Marzo | 58 | 25 | 83 |
| 2021 Febrero | 39 | 31 | 70 |
| 2021 Enero | 28 | 20 | 48 |
| 2020 Diciembre | 16 | 16 | 32 |
| 2020 Noviembre | 20 | 22 | 42 |
| 2020 Octubre | 21 | 14 | 35 |
| 2020 Septiembre | 19 | 8 | 27 |
| 2020 Agosto | 25 | 17 | 42 |
| 2020 Julio | 25 | 12 | 37 |
| 2020 Junio | 24 | 31 | 55 |
| 2020 Mayo | 18 | 19 | 37 |
| 2020 Abril | 22 | 16 | 38 |
| 2020 Marzo | 23 | 20 | 43 |
| 2020 Febrero | 4 | 0 | 4 |
| 2019 Diciembre | 4 | 0 | 4 |
| 2019 Septiembre | 4 | 0 | 4 |
| 2019 Mayo | 2 | 2 | 4 |
| 2019 Abril | 0 | 1 | 1 |
| 2019 Marzo | 2 | 4 | 6 |
| 2019 Febrero | 2 | 0 | 2 |
| 2018 Diciembre | 3 | 0 | 3 |
| 2018 Octubre | 3 | 0 | 3 |
| 2018 Septiembre | 4 | 0 | 4 |
| 2018 Febrero | 17 | 4 | 21 |
| 2018 Enero | 49 | 5 | 54 |
| 2017 Diciembre | 43 | 13 | 56 |
| 2017 Noviembre | 33 | 4 | 37 |
| 2017 Octubre | 36 | 6 | 42 |
| 2017 Septiembre | 43 | 5 | 48 |
| 2017 Agosto | 35 | 4 | 39 |
| 2017 Julio | 37 | 7 | 44 |
| 2017 Junio | 38 | 9 | 47 |
| 2017 Mayo | 39 | 6 | 45 |
| 2017 Abril | 30 | 9 | 39 |
| 2017 Marzo | 18 | 16 | 34 |
| 2017 Febrero | 16 | 4 | 20 |
| 2017 Enero | 12 | 4 | 16 |
| 2016 Diciembre | 35 | 19 | 54 |
| 2016 Noviembre | 30 | 18 | 48 |
| 2016 Octubre | 25 | 15 | 40 |
| 2016 Julio | 3 | 6 | 9 |
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