Case for Diagnosis
Pitting Edema of the Face, An Important Differential Diagnosis
Edema facial con fóvea, un importante diagnóstico diferencial
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Chalela, M.I. González, M.F. Ordóñez Rubiano" "autores" => array:3 [ 0 => array:3 [ "nombre" => "J.G." "apellidos" => "Chalela" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 1 => array:3 [ "nombre" => "M.I." "apellidos" => "González" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:4 [ "nombre" => "M.F." "apellidos" => "Ordóñez Rubiano" "email" => array:1 [ 0 => "Mfordonezr@Gmail.Com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] ] "afiliaciones" => array:3 [ 0 => array:3 [ "entidad" => "Departamento de Dermatología, Hospital Universitario Fundación Santa Fe, Bogotá, Colombia" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Departamento de Anatomía Patológica, Universidad Militar Nueva Granada, Bogotá, Colombia" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Departamento de Dermatología, Universidad Militar Nueva Granada, Bogotá, Colombia" "etiqueta" => "c" "identificador" => "aff0015" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Edema facial con fóvea, un importante diagnóstico diferencial" ] ] "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" => 755 "Ancho" => 506 "Tamanyo" => 67906 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Facial pitting erythematous edema, predominantly on the left side.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">CASE PRESENTATION</span><p id="par0005" class="elsevierStylePara elsevierViewall">A man in his 80s presented with a 7-month history of progressive facial swelling that started in the cheeks and extended to the eyelids and then to the whole face, predominantly on the left side. He had not received treatment for his current complaint. The patient had a history of type 2 diabetes controlled with metformin, and a slightly elevated prostate-specific antigen, with no history of other dermatological conditions.</p><p id="par0010" class="elsevierStylePara elsevierViewall">On physical examination, solid, pitting edema involving his entire face (including eyelids and partially the scalp) associated with erythema was observed (<a class="elsevierStyleCrossRef" href="#fig0005">Figure 1</a>). There were no other lesions in the total skin examination and the rest of the physical exam was unremarkable. A biopsy specimen from his cheek was obtained where a poorly circumscribed dermal tumor extending into the subcutaneous tissue composed of thin vessels lined by prominent endothelial cells exhibiting cellular atypia and proliferation on the luminal side. The dilated vessels extended between dermal collagen bundles and were accompanied by inflammatory cells, predominantly small lymphocytes and plasmocytes.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Immunohistochemistry markers were positive for CD31, FLI1, CD34 and D240 on the lining endothelial cells of the dilated vessels, with a proliferation index (Ki67) of 70%. HHV-8 was negative. (<a class="elsevierStyleCrossRef" href="#fig0010">Figure 2</a>).</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">DIAGNOSIS</span><p id="par0020" class="elsevierStylePara elsevierViewall">Idiopathic angiosarcoma</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">DISCUSSION</span><p id="par0025" class="elsevierStylePara elsevierViewall">Idiopathic angiosarcoma of the head and neck (IAHN) is one of the three main clinicopathological subtypes (angiosarcoma complicating lymphedema and postirradiation angiosarcoma) described in 1987.<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">1,2</span></a> This type of angiosarcoma commonly involves the upper part of the face or the scalp of elderly men.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">3</span></a> The lesions may be single or multifocal, violaceous nodules, plaques or macules representing infiltrating areas, usually they become markedly more violaceous and bloated when the head-tilt maneuver is performed (placing the head below the level of the heart).<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">1,4</span></a> The size usually ranges between 0.8 – 8<span class="elsevierStyleHsp" style=""></span>cms but larger tumors have been recorded.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">4</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">There have been other clinical presentations of IAHN including recurrent angioedema of the face, a rosacea-like eruption, rhinophyma-like eruption or an inflammatory process.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">4</span></a> IAHN presenting as persistent swelling of the face has been reported and,<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">5–7</span></a> even thought it is rare, it is an important differential diagnosis of this acquired syndrome that includes infectious (leprosy, tuberculosis, lymphedema secondary to herpes infection), inflammatory (rosacea, Morbihan disease, acne, Melkersson-Rosenthal syndrome, angioedema), neoplastic (cutaneous lymphomas, metastases) and miscellaneous disorders (amyloidosis, scleromyxedema, Wells syndrome, pseudolymphoma, post-operative and post-radiotherapy states).<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">8</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Clinical suspicion must be confirmed with histopathology and immunohistochemistry.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">3</span></a> Histopathology is similar in all the types of angiosarcoma, and is highly variable depending on its differentiation.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">1</span></a> They are characterized by irregular, meshwork of anastomosing dilated vessels that dissect dermal collagen bundles and skin appendages usually involving the deep subcutaneous tissue.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">3</span></a> The lining endothelial cells appear hyperchromatic and pleomorphic that usually protrudes into vascular lumina. Immunohistochemical markers are of vascular origin, cytoplasmic CD31 and CD34 are the most frequently used for diagnosis, for a more sensitive diagnosis other nuclear inmunohistochemical markers such as ERG and FLI1 may be used.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">1</span></a> Additionally, D2-40 and VEGFR-3, lymphatic markers are expressed.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">1</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Currently, surgery (3<span class="elsevierStyleHsp" style=""></span>cm margins) with subsequent radiotherapy is the mainstay of treatment.<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">1,3</span></a> Regarding chemotherapy, studies are inconsistent, paclitaxel and doxorubicin-based regimens are considered the best choice.<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">1,4</span></a> New targeted cancer therapies (tyrosine kinase-pazopanib, sorafenib- and VEGF inhibitors-Bevacizumab-) have been used for angiosarcoma but the studies have shown conflicting results.<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">1,4</span></a> Treatment of facial swelling IAHN is extrapolated from studies of other frequent variants, but surgery is not an option given the difficulty of the approach.</p><p id="par0045" class="elsevierStylePara elsevierViewall">It is important to consider IAHN as a differential diagnosis of solid progressive pitting edema of the face as it alters the patient's life prognosis due to ineffective and poorly studied treatment options. Our patient is currently treated with radiotherapy, with no progression of the disease after 6 months. Upcoming studies regarding new targeted cancer therapies may change its prognosis, even in extensive and highly proliferative disease such as in the case of our patient.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:4 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "CASE PRESENTATION" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "DIAGNOSIS" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "DISCUSSION" ] 3 => array:1 [ "titulo" => "Références" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2018-02-14" "fechaAceptado" => "2018-04-16" "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Chalela JG, González MI, Ordóñez Rubiano MF. Edema facial con fóvea, un importante diagnóstico diferencial. Actas Dermosifiliogr. 2020;111:65–66.</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" => 755 "Ancho" => 506 "Tamanyo" => 67906 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Facial pitting erythematous edema, predominantly on the left side.</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" => 733 "Ancho" => 756 "Tamanyo" => 214821 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A. Panoramic view of the tumor extending through the dermis. B. The tumor composed of thin vessels lined by prominent endothelial cells exhibiting cellular atypia on the luminal side. C-D Immunohistochemistry showing vascular and endothelial differentiation, also high proliferation index.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "Références" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:8 [ 0 => array:3 [ "identificador" => "bib0045" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Cutaneous angiosarcoma: A current update" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "E. Shustef" 1 => "V. Kazlouskaya" 2 => "V.G. Prieto" 3 => "D. Ivan" 4 => "P.P. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 37 | 3 | 40 |
| 2024 October | 340 | 58 | 398 |
| 2024 September | 368 | 28 | 396 |
| 2024 August | 405 | 66 | 471 |
| 2024 July | 514 | 43 | 557 |
| 2024 June | 387 | 43 | 430 |
| 2024 May | 312 | 33 | 345 |
| 2024 April | 318 | 33 | 351 |
| 2024 March | 391 | 35 | 426 |
| 2024 February | 394 | 44 | 438 |
| 2024 January | 413 | 36 | 449 |
| 2023 December | 419 | 30 | 449 |
| 2023 November | 576 | 33 | 609 |
| 2023 October | 569 | 18 | 587 |
| 2023 September | 474 | 30 | 504 |
| 2023 August | 437 | 21 | 458 |
| 2023 July | 519 | 44 | 563 |
| 2023 June | 500 | 32 | 532 |
| 2023 May | 667 | 42 | 709 |
| 2023 April | 523 | 23 | 546 |
| 2023 March | 587 | 34 | 621 |
| 2023 February | 402 | 34 | 436 |
| 2023 January | 447 | 39 | 486 |
| 2022 December | 320 | 45 | 365 |
| 2022 November | 324 | 33 | 357 |
| 2022 October | 269 | 25 | 294 |
| 2022 September | 235 | 35 | 270 |
| 2022 August | 278 | 36 | 314 |
| 2022 July | 211 | 44 | 255 |
| 2022 June | 99 | 43 | 142 |
| 2022 May | 307 | 56 | 363 |
| 2022 April | 397 | 36 | 433 |
| 2022 March | 417 | 62 | 479 |
| 2022 February | 477 | 48 | 525 |
| 2022 January | 491 | 60 | 551 |
| 2021 December | 393 | 52 | 445 |
| 2021 November | 350 | 38 | 388 |
| 2021 October | 415 | 56 | 471 |
| 2021 September | 319 | 38 | 357 |
| 2021 August | 346 | 29 | 375 |
| 2021 July | 362 | 25 | 387 |
| 2021 June | 668 | 64 | 732 |
| 2021 May | 424 | 55 | 479 |
| 2021 April | 880 | 113 | 993 |
| 2021 March | 530 | 37 | 567 |
| 2021 February | 278 | 33 | 311 |
| 2021 January | 246 | 17 | 263 |
| 2020 December | 229 | 20 | 249 |
| 2020 November | 146 | 13 | 159 |
| 2020 October | 120 | 15 | 135 |
| 2020 September | 79 | 20 | 99 |
| 2020 August | 84 | 17 | 101 |
| 2020 July | 63 | 10 | 73 |
| 2020 June | 50 | 20 | 70 |
| 2020 May | 61 | 21 | 82 |
| 2020 April | 56 | 19 | 75 |
| 2020 March | 26 | 12 | 38 |
| 2020 February | 2 | 2 | 4 |
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