Dermatopathology
Intravascular histiocytosis: case report of a rare disease probably associated with silicone breast implant
Yasmin Gama Abuawad
, Ticiana de Andrade Castelo Branco Diniz, Priscila Kakizaki, Neusa Yuriko Sakai Valente
Corresponding author
Department of Dermatology, Hospital do Servidor Público Estadual de São Paulo, São Paulo, SP, Brazil
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implant" "tieneTextoCompleto" => true "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "347" "paginaFinal" => "350" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Yasmin Gama Abuawad, Ticiana de Andrade Castelo Branco Diniz, Priscila Kakizaki, Neusa Yuriko Sakai Valente" "autores" => array:4 [ 0 => array:4 [ "nombre" => "Yasmin Gama" "apellidos" => "Abuawad" "email" => array:1 [ 0 => "yasmin_gama@yahoo.com.br" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "Ticiana de Andrade Castelo Branco" "apellidos" => "Diniz" ] 2 => array:2 [ "nombre" => "Priscila" "apellidos" => "Kakizaki" ] 3 => array:2 [ "nombre" => "Neusa Yuriko Sakai" "apellidos" => "Valente" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Department of Dermatology, Hospital do Servidor Público Estadual de São Paulo, São Paulo, SP, Brazil" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "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" => 542 "Ancho" => 1305 "Tamanyo" => 195127 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">(A) Ectasia, thrombosis and recanalization of upper dermal vessels and the presence of histiocytoid cells inside of those vessels, (Hematoxylin & eosin, ×100). (B) Detail of the histiocytoid cells, (Hematoxylin & eosin, ×400).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Intravascular histiocytosis is a rare condition characterized by the presence of histiocytes within the dilated lymphatic vessels of the dermis. It can be primary or secondary to systemic diseases such as rheumatoid arthritis and metallic prosthesis. The pathogenesis of the disease is still unclear.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Case report</span><p id="par0010" class="elsevierStylePara elsevierViewall">A 55 year-old female patient referred the onset of assymptomatic erythematous/purple patches in the breasts with progression to the back and proximal limbs one year and 6 months ago (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). The patient presented with infiltrative ductal breast cancer in the right breast treated in 2006 and proceeded to the placement of silicone implant in 2009. The main clinical hypothesis were telangiectatic metastatic breast carcinoma and three cutaneous biopsies were performed in the left breast and one in the right thigh. The histopathology revealed ectasia, thrombosis and recanalization of upper dermal vessels and the presence of histiocytoid cells inside of those vessels (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). The intravascular cells were negative for estrogen and progesterone receptors, CK7, EMA and AE1/AE3 in the immnunohistochemistry, which excluded the hypothesis of telangiectatic metastatic breast carcinoma. The endotelial cells were CD34 positive (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). The CD68 antibody was then utilized and was positive in the intravascular cells, confirming the histiocytoid origin and leading to the diagnosis of intravascular histiocytosis (<a class="elsevierStyleCrossRef" href="#fig0020">Fig. 4</a>).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><elsevierMultimedia ident="fig0020"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Discussion</span><p id="par0015" class="elsevierStylePara elsevierViewall">Intravascular histiocytosis is a rare condition first described in 1994. It is characterized by the presence of histiocytes within the dilated lymphatic vessels of the papillary and reticular dermis.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,2</span></a> Since 2003, the disease has been included on the spectrum of reactive cutaneous angiomatosis, as well as reactive angioendotheliomatosis. Intravascular histiocytosis may be primary or secondary mainly to rheumathoid arthritis, but also to other inflamatory diseases such as Crohn's disease, dermatomyositis and monoclonal gammopathy, metallic prosthesis, chronic infections and neoplasms, specially breast cancer. However, there are cases showing association with colorectal cancer, melanoma, vulvar necrosis and osteoarthrosis.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,3</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">The disease affects mostly women with 17–87 years old (65 years, mean age). The cutaneous lesions are most commonly limited and presented as erythematous/purple macules or pacthes or with livedo reticular like lesions in the limbs or trunck. They are rarely disseminated. In cases associated with rheumathoid arthritis, metallic prosthesis and osteoarthrosis, the lesion appeared near the joint and in cases associated with breast cancer, they appeared on the scar of the mastectomy.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,2</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">The pathogenesis remains nuclear,<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a> however the findings suggest that the key to the pathogenesis can be the migration of histiocytes caused by the surrounding inflammatory reaction. Requena et al. postulated that intravascular histiocytosis would be caused by congenital or acquired lymphangiectasy or lymphatic obstruction, by trauma, surgery, radiation or infections.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2,5,6</span></a> Other hypothesis is that chronic inflammation would lead to stasis and more exposition to antigens that would stimulate proliferation and aggregation of histiocytes within lymphatic vessels. The role of TNF is being taking in into account because of the disease's association with rheumathoid arthritis and other disorders associated with histiocytes, such as multicentric reticulohistiocytosis and intersticial granulomatous dermatites.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,3</span></a> The histologic findings consist of CD68+ histiocytes aggregates within the dilated lymphatic vessels (podoplanin+) in the papillary and reticular dermis. Histophatological differential diagnosis with reactive angioendotheliomatosis remains controversial and some authors suggest that they represent different aspects of the same phenomenon in which the presence of histiocytes would lead to formation of microthrombos and endothelial proliferation.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2,7</span></a> Telangiectatic metastatic breast carcinoma is a rare form of cutaneous metastasis of breast cancer that consists in the onset of erythemathous macules with telangiectasias or lymphangioma circumscriptum. Histopathology reveals intravascular proliferation of tumoral cells that can be positive for estrogen and progesterone receptors.<a class="elsevierStyleCrossRefs" href="#bib0090"><span class="elsevierStyleSup">8,9</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">The course of the disease is chronic and there is not a specific treatment. In the secondary cases it is necessary to treat the overlying disease. Some patients evolved into cure after removal of metallic prosthesis.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,2,10</span></a> Therapy with infliximab, methotrexate, pentoxifiline, radiotherapy, corticosteroids and topic tracolimus have variable response and frequent relapses.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,2</span></a> Our case presented an exuberant disseminated disease and possible association with silicone breast implant, for its beginning in the breasts, which association has never been described in the literature.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Financial support</span><p id="par0035" class="elsevierStylePara elsevierViewall">None declared.</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Authors' contributions</span><p id="par0040" class="elsevierStylePara elsevierViewall">Yasmin Gama Abuawad: Conception and planning of the study; elaboration and writing of the manuscript; obtaining, analysis, and interpretation of the data; critical review of the literature.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Ticiana de Andrade Castelo Branco Diniz: Conception and planning of the study; obtaining, analysis, and interpretation of the data; critical review of the literature.</p><p id="par0050" class="elsevierStylePara elsevierViewall">Priscila Kakizaki: Approval of the final version of the manuscript; conception and planning of the study; obtaining, analysis, and interpretation of the data; effective participation in research orientation; 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="par0055" class="elsevierStylePara elsevierViewall">Neusa Yuriko Sakai Valente: Approval of the final version of the manuscript; conception and planning of the study; obtaining, analysis, and interpretation of the data; effective participation in research orientation; intellectual participation in the propaedeutic and/or therapeutic conduct of the studied cases; critical review of the literature; critical review of the manuscript.</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conflicts of interest</span><p id="par0060" class="elsevierStylePara elsevierViewall">None declared.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:9 [ 0 => array:3 [ "identificador" => "xres1365101" "titulo" => "Abstract" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0005" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec1254933" "titulo" => "Keywords" ] 2 => array:2 [ "identificador" => "sec0005" "titulo" => "Introduction" ] 3 => array:2 [ "identificador" => "sec0010" "titulo" => "Case report" ] 4 => array:2 [ "identificador" => "sec0015" "titulo" => "Discussion" ] 5 => array:2 [ "identificador" => "sec0020" "titulo" => "Financial support" ] 6 => array:2 [ "identificador" => "sec0025" "titulo" => "Authors' contributions" ] 7 => array:2 [ "identificador" => "sec0030" "titulo" => "Conflicts of interest" ] 8 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2018-11-30" "fechaAceptado" => "2019-04-01" "PalabrasClave" => array:1 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec1254933" "palabras" => array:4 [ 0 => "Cell proliferation" 1 => "Histiocytosis" 2 => "Immunohistochemistry" 3 => "Prostheses and implants" ] ] ] ] "tieneResumen" => true "resumen" => array:1 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Intravascular histiocytosis is a rare condition characterized by the aggregate of histiocytes within dilated dermal vessels. The diagnosis is mainly histophatological and immunohistochemical. We describe a case of a 55 year-old female patient presenting erythematous/purple patches on the breasts, back and limbs. She previously presented ductal carcinoma in the right breast in 2006 which was treated with mastectomy and proceeded to silicone breast implant in 2009. Clinical hypothesis was telangiectatic metastatic carcinoma. Histopathology showed vascular ectasia, thrombosis and recanalization of upper dermis small vessels. On immunohistochemistry, intravascular cells were CD 68+ and negative for estrogen and progesterone receptors, CK7, EMA and AE1/AE3 and endothelial cells were CD64+, leading to the diagnosis of intravascular histiocytosis.</p></span>" ] ] "NotaPie" => array:2 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">How to cite this article: Abuawad YG, Diniz TACB, Kakizaki P, Valente NYS. Intravascular histiocytosis: case report of a rare disease probably associated with silicone breast implant. An Bras Dermatol. 2020;95:344–7.</p>" ] 1 => array:2 [ "etiqueta" => "☆☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0010">Study conducted at the Hospital do Servidor Público Estadual de São Paulo, São Paulo, SP, Brazil.</p>" ] ] "multimedia" => array:4 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 2112 "Ancho" => 2167 "Tamanyo" => 336447 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Erythematous/purple patches on the breasts, back and limbs.</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" => 542 "Ancho" => 1305 "Tamanyo" => 195127 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">(A) Ectasia, thrombosis and recanalization of upper dermal vessels and the presence of histiocytoid cells inside of those vessels, (Hematoxylin & eosin, ×100). (B) Detail of the histiocytoid cells, (Hematoxylin & eosin, ×400).</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" => 680 "Ancho" => 905 "Tamanyo" => 117289 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Immunohistochemistry CD 34 (400×): positive in the endothelial cells.</p>" ] ] 3 => array:7 [ "identificador" => "fig0020" "etiqueta" => "Figure 4" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr4.jpeg" "Alto" => 437 "Ancho" => 905 "Tamanyo" => 110019 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">(A) Immunohistochemistry CD68, 200×: positive in the intravascular cells. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 17 | 5 | 22 |
| 2024 October | 92 | 72 | 164 |
| 2024 September | 129 | 92 | 221 |
| 2024 August | 138 | 127 | 265 |
| 2024 July | 118 | 114 | 232 |
| 2024 June | 116 | 91 | 207 |
| 2024 May | 90 | 55 | 145 |
| 2024 April | 108 | 98 | 206 |
| 2024 March | 110 | 67 | 177 |
| 2024 February | 140 | 82 | 222 |
| 2024 January | 117 | 55 | 172 |
| 2023 December | 109 | 68 | 177 |
| 2023 November | 143 | 78 | 221 |
| 2023 October | 78 | 82 | 160 |
| 2023 September | 65 | 72 | 137 |
| 2023 August | 45 | 41 | 86 |
| 2023 July | 38 | 37 | 75 |
| 2023 June | 38 | 38 | 76 |
| 2023 May | 44 | 22 | 66 |
| 2023 April | 38 | 20 | 58 |
| 2023 March | 70 | 50 | 120 |
| 2023 February | 45 | 40 | 85 |
| 2023 January | 39 | 36 | 75 |
| 2022 December | 40 | 32 | 72 |
| 2022 November | 38 | 55 | 93 |
| 2022 October | 85 | 54 | 139 |
| 2022 September | 60 | 73 | 133 |
| 2022 August | 34 | 54 | 88 |
| 2022 July | 46 | 48 | 94 |
| 2022 June | 39 | 68 | 107 |
| 2022 May | 32 | 58 | 90 |
| 2022 April | 50 | 48 | 98 |
| 2022 March | 43 | 76 | 119 |
| 2022 February | 25 | 35 | 60 |
| 2022 January | 48 | 89 | 137 |
| 2021 December | 47 | 49 | 96 |
| 2021 November | 48 | 70 | 118 |
| 2021 October | 57 | 104 | 161 |
| 2021 September | 34 | 47 | 81 |
| 2021 August | 45 | 64 | 109 |
| 2021 July | 36 | 45 | 81 |
| 2021 June | 31 | 50 | 81 |
| 2021 May | 42 | 76 | 118 |
| 2021 April | 85 | 154 | 239 |
| 2021 March | 83 | 62 | 145 |
| 2021 February | 52 | 11 | 63 |
| 2021 January | 72 | 20 | 92 |
| 2020 December | 44 | 18 | 62 |
| 2020 November | 49 | 15 | 64 |
| 2020 October | 44 | 6 | 50 |
| 2020 September | 21 | 11 | 32 |
| 2020 August | 13 | 8 | 21 |
| 2020 July | 23 | 14 | 37 |
| 2020 June | 19 | 16 | 35 |
| 2020 May | 16 | 9 | 25 |
| 2020 April | 0 | 4 | 4 |
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