Case and Research Letter
Granulomatous Variant of Pigmented Purpura
Variante granulomatosa de púrpura pigmentada
Department of Dermatology, Shirakawa Kosei General Hospital, and Fukushima Medical University, Fukushima, Japan
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Hakenoshita, T. Yamamoto" "autores" => array:2 [ 0 => array:2 [ "nombre" => "H." "apellidos" => "Hakenoshita" ] 1 => array:4 [ "nombre" => "T." "apellidos" => "Yamamoto" "email" => array:1 [ 0 => "toyamade@fmu.ac.jp" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Department of Dermatology, Shirakawa Kosei General Hospital, and Fukushima Medical University, Fukushima, Japan" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Variante granulomatosa de púrpura pigmentada" ] ] "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" => 964 "Ancho" => 1300 "Tamanyo" => 196035 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Clinical features of purpuric lesions on the right and left shin. (B) Close-up view.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">We report the case of an elderly man with a granulomatous variant of chronic pigmented purpura who presented with papular lesions on the lower legs.</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 78-year-old Japanese man complained of asymptomatic eruptions of 2 years’ duration on his lower legs. He had no history of systemic granulomatous disease and had not taken any drugs. Physical examination showed a number of purpuric papular lesions on both shins (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A and B). The lesions were tiny, ranging from the size of a pinhead to 5<span class="elsevierStyleHsp" style=""></span>mm, and were both isolated and arranged in clusters. Laboratory data, which included blood cell counts, biochemical liver and renal function tests, and lipid, cholesterol, and triglyceride levels were normal. Histologic examination revealed extravasation of red blood cells, hemosiderin deposition, and dense inflammatory cell infiltration in the upper dermis (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>A). The infiltrate was composed of mononuclear cells, histiocytes, and giant cells (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>B), and immunohistochemistry revealed a number of CD3<span class="elsevierStyleSup">+</span> T cells, with a predominance of CD4<span class="elsevierStyleSup">+</span> cells. There were also CD8<span class="elsevierStyleSup">+</span> T cells scattered among the granulomatous lesions (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>C–E) and abundant CD68<span class="elsevierStyleSup">+</span> cells at the periphery of the cellular infiltrates (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>F). Vasculitis was not observed. Special stains for microorganisms were all negative. The purpuric lesions were resistant to treatment with topical corticosteroids.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Several cases of granulomatous pigmented purpura have been described since the entity was first reported by Saito and Matsuoka.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a> Most of the cases have involved Asian people, but there have been few reports from Japan.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> Although the clinical features described in the original report included lichen aureus-like brown coalesced hemorrhagic papules on the dorsum of the feet,<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a> additional features such as macules, papules, plaques, and pigmentation have been reported. Apart from the typical clinical features of chronic pigmented purpura, our patient also had a number of papular lesions on both shins, similarly to a previously reported case.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a> The age of the patients with granulomatous pigmented purpura described in the literature ranges from 22 to 78 years old (present case), with disease duration varying from 2 months to 20 years. In addition to the lower extremities, there have also been rare reports of lesions on the wrists,<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">4</span></a> forearms, and lower back.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">5</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">In our case, histologic features showed dense cellular infiltrates composed of lymphocytes and histiocytes in the upper dermis. CD4<span class="elsevierStyleSup">+</span> cells were predominant among the CD3<span class="elsevierStyleSup">+</span> T cells, C, and CD68<span class="elsevierStyleSup">+</span> histiocytes surrounded the cellular infiltrates in the upper dermis. What triggered the granulomatous reaction is not clear, but the long-term chronic course may have had a role. Hyperlipidemia has been reported in several cases,<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">6</span></a> and other studies have suggested that lipid microdroplets may induce an inflammatory reaction of the microvasculature.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a> Drugs have also been suggested as possible triggers and have been implicated as causative factors of pigmented purpura. In addition, there have been reports of an association with antinuclear antibody and rheumatoid factor positivity, and regulatory T cells were attributed a possible role in the induction of granulomatous changes in pigmented purpura in a patient with Sjögren syndrome.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> In our case, associations with systemic autoimmune diseases, hyperlipidemia, and drug intake were all ruled out. Further studies are necessary to investigate the triggering events and etiology of granulomatous reactions in pigmented purpura.</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" => 964 "Ancho" => 1300 "Tamanyo" => 196035 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Clinical features of purpuric lesions on the right and left shin. (B) Close-up view.</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" => 1439 "Ancho" => 1950 "Tamanyo" => 559753 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">(A) Histological features showing focal dense cellular infiltrates in the upper dermis. (B) Higher magnification showed lymphohistiocytic infiltration with giant cells. The immunohistochemistry study revealed a number of CD3<span class="elsevierStyleSup">+</span> (C), CD4<span class="elsevierStyleSup">+</span> (D), CD8<span class="elsevierStyleSup">+</span> (E), and CD68<span class="elsevierStyleSup">+</span> (F) cells.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:6 [ 0 => array:3 [ "identificador" => "bib0035" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Granulomatous pigmented purpuric dermatosis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "R. Saito" 1 => "Y. Matsuoka" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:6 [ "tituloSerie" => "J Dermatol" "fecha" => "1996" "volumen" => "23" "paginaInicial" => "551" "paginaFinal" => "555" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/8854588" "web" => "Medline" ] ] ] ] ] ] ] ] 1 => array:3 [ "identificador" => "bib0040" "etiqueta" => "2" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Granulomatous pigmented purpuric dermatitis associated with primary Sjögren's syndrome" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "C. Wakusawa" 1 => "T. Fujimura" 2 => "T. Haga" 3 => "S. Aiba" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.2340/00015555-1380" "Revista" => array:6 [ "tituloSerie" => "Acta Derm Venereol" "fecha" => "2013" "volumen" => "93" "paginaInicial" => "95" "paginaFinal" => "96" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/22717995" "web" => "Medline" ] ] ] ] ] ] ] ] 2 => array:3 [ "identificador" => "bib0045" "etiqueta" => "3" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Granulomatous pigmented purpura: an unusual histological variant" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "M.J.J. Kerns" 1 => "B.D. Mallatt" 2 => "H.N. Shamma" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1097/DAD.0b013e31817e23c9" "Revista" => array:6 [ "tituloSerie" => "Am J Dermatopathol" "fecha" => "2009" "volumen" => "31" "paginaInicial" => "77" "paginaFinal" => "80" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/19155731" "web" => "Medline" ] ] ] ] ] ] ] ] 3 => array:3 [ "identificador" => "bib0050" "etiqueta" => "4" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Granulomatous variant of chronic pigmented purpuric dermatoses: report of four new cases and an association with hyperlipidaemia" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:6 [ 0 => "W.L. Lin" 1 => "T.T. Kuo" 2 => "P.Y. Shih" 3 => "W.C. Lin" 4 => "W.R. Wong" 5 => "H.S. Hong" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1111/j.1365-2230.2007.02462.x" "Revista" => array:6 [ "tituloSerie" => "Clin Exp Dermatol" "fecha" => "2007" "volumen" => "32" "paginaInicial" => "513" "paginaFinal" => "515" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/17535280" "web" => "Medline" ] ] ] ] ] ] ] ] 4 => array:3 [ "identificador" => "bib0055" "etiqueta" => "5" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Granulomatous pigmented purpura: report of a case and review of the literature" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "J. Kaplan" 1 => "S. Burgin" 2 => "A. 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Walsh" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1111/j.1600-0560.2011.01793.x" "Revista" => array:6 [ "tituloSerie" => "J Cutan Pathol" "fecha" => "2011" "volumen" => "38" "paginaInicial" => "979" "paginaFinal" => "983" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/21899591" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/00017310/0000010700000001/v1_201512300105/S0001731015003415/v1_201512300105/en/main.assets" "Apartado" => array:4 [ "identificador" => "6160" "tipo" => "SECCION" "es" => array:2 [ "titulo" => "Cartas científico-clínicas" "idiomaDefecto" => true ] "idiomaDefecto" => "es" ] "PDF" => "https://static.elsevier.es/multimedia/00017310/0000010700000001/v1_201512300105/S0001731015003415/v1_201512300105/en/main.pdf?idApp=UINPBA000044&text.app=https://actasdermo.org/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0001731015003415?idApp=UINPBA000044" ]
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| 2017 Octubre | 3 | 9 | 12 |
| 2017 Septiembre | 8 | 13 | 21 |
| 2017 Agosto | 5 | 15 | 20 |
| 2017 Julio | 3 | 8 | 11 |
| 2017 Junio | 7 | 30 | 37 |
| 2017 Mayo | 5 | 23 | 28 |
| 2017 Abril | 10 | 20 | 30 |
| 2017 Marzo | 7 | 41 | 48 |
| 2017 Febrero | 6 | 3 | 9 |
| 2017 Enero | 6 | 8 | 14 |
| 2016 Diciembre | 4 | 28 | 32 |
| 2016 Noviembre | 3 | 14 | 17 |
| 2016 Octubre | 6 | 22 | 28 |
| 2016 Septiembre | 0 | 2 | 2 |
| 2016 Agosto | 1 | 1 | 2 |
| 2016 Julio | 1 | 1 | 2 |
| 2016 Junio | 1 | 1 | 2 |
| 2016 Mayo | 0 | 1 | 1 |
| 2016 Abril | 0 | 1 | 1 |
| 2016 Marzo | 0 | 9 | 9 |
| 2016 Febrero | 0 | 6 | 6 |
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