Case and Research Letter
Linear Multiple Epithelioid Hemangioma Associated With a Quiescent Vascular Malformation
Hemangioma epiteloide múltiple lineal asociado a malformación vascular quiescente
C. Martínez-Peinadoa,
, R. Ruiz-Villaverdeb, M. Galán-Gutierrezc, J.M. Martínez de Victoriad
Corresponding author
a Unidad de Gestión Clínica de CMA-Dermatología Médico Quirúrgica y Venereología, Complejo Hospitalario de Jaén, Jaén, Spain
b Unidad de Gestión Clínica de Dermatología Médico Quirúrgica y Venereología, Complejo Hospitalario de Granada, Granada, Spain
c Servicio de Dermatología, Hospital Universitario Reina Sofía, Córdoba, Spain
d Servicio de Anatomía Patológica, Complejo Hospitalario de Jaén, Jaén, Spain
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"apellidos" => "Martínez de Victoria" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">d</span>" "identificador" => "aff0020" ] ] ] ] "afiliaciones" => array:4 [ 0 => array:3 [ "entidad" => "Unidad de Gestión Clínica de CMA-Dermatología Médico Quirúrgica y Venereología, Complejo Hospitalario de Jaén, Jaén, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Unidad de Gestión Clínica de Dermatología Médico Quirúrgica y Venereología, Complejo Hospitalario de Granada, Granada, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Servicio de Dermatología, Hospital Universitario Reina Sofía, Córdoba, Spain" "etiqueta" => "c" "identificador" => "aff0015" ] 3 => array:3 [ "entidad" => "Servicio de Anatomía Patológica, Complejo Hospitalario de Jaén, Jaén, Spain" "etiqueta" => "d" "identificador" => "aff0020" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Hemangioma epiteloide múltiple lineal asociado a malformación vascular quiescente" ] ] "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" => 830 "Ancho" => 1534 "Tamanyo" => 258283 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A, Histology showing the vascular proliferation of epithelioid endothelial cells with a solid pattern lining vascular spaces. Hematoxylin and eosin, original magnification ×40. B, Magnetic resonance angiography image of the arteriovenous malformation in the left upper limb, showing occlusion of the proximal humeral artery.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Epithelioid hemangioma (EH) is a rare, benign, idiopathic vascular proliferation first described by Wells and Whimster in 1969.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">1</span></a> It typically arises in the skin or subcutaneous cellular tissue of the head and neck and, more rarely, has been reported on the trunk and limbs. Trauma, infection, and an underlying vascular malformation have been proposed as possible causes. We present the case of an adolescent male diagnosed with multiple EHs on the left upper limb. Imaging studies revealed a large asymptomatic aneurysmal lesion that required surgery in order to bring about resolution of the EH lesions.</p><p id="par0010" class="elsevierStylePara elsevierViewall">A boy aged 14 years with no past history of interest consulted for the progressive appearance over the previous 2 years of a number of violaceous tumors on his left upper limb. He described the lesions as slow growing and asymptomatic, though some of the lesions had bled on minimal trauma in recent months. Examination revealed the presence of multiple erythematous-violaceous cutaneous and subcutaneous papules and tumors in a linear distribution along the left upper limb (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>); lesions in the distal region of the second, third, and fourth fingers produced clubbing, and some had superficial hemorrhagic scabs. Biopsy was performed on a suspicion of lesions of vascular origin, and histology revealed a proliferation of endothelial cells lining large vascular spaces, with a lymphocytic inflammatory infiltrate and abundant eosinophils in the dermis (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>A). Immunohistochemistry was positive for CD31 and CD34. Based on these findings, we made a diagnosis of EH. Complete blood count, routine biochemistry, and coagulation studies were within normal limits except for the presence of a moderate peripheral eosinophilia. Results of the imaging studies, including magnetic resonance imaging (MRI) and arteriography of the affected limb, suggested the existence of a complex aneurysmal lesion in the proximal humeral artery, with destructured vessels and arterial occlusion, and repermeabilization distal to the aneurysmal dilatation (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>B). Vascular nests were observed at the elbow and in the hand, supplied mainly by a branch arising from the area of the aneurysm. Venous return was also abnormal. The final diagnosis was arteriovenous malformation in the left upper limb with humeral artery pseudoaneurysm. Treatment involved resection of the lesion with by-pass using basilic vein from the same arm. Three months after the operation we observed a surprising reduction in the size of the EHs, some of which had disappeared completely. A number of residual fibrotic tags were observed on the distal areas of the fingers (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). Six months after surgery the patient continued on prophylactic antiplatelet treatment (acetylsalicylic acid, 100<span class="elsevierStyleHsp" style=""></span>mg/d) and there was no recurrence of the EHs.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">EH occurs mainly in adult patients of all races; it is rare in the pediatric population. Involvement of the trunk and limbs is extremely rare.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">2</span></a> Clinically they present as reddish-brown papules and tumors that are usually asymptomatic, though they may give rise to pruritus, bleeding, ulceration, or pain, due mostly to lesion growth. They may be superficial or deep; few cases with multiple lesions or a zosteriform pattern have been reported.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">3,4</span></a> Peripheral eosinophilia is observed in less than 20% of cases, and a regional lymphadenopathy more rarely still. Histologically, the lesions are characterized by a proliferation of large endothelial cells of epithelioid morphology that obstruct the vessel lumen, associated with a variable lymphoplasmacytic inflammatory component with abundant eosinophils. Immunohistochemistry is positive for endothelial markers CD31 and CD34, and negative for epithelial markers (cytokeratins).<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">5</span></a> In children, the main differential diagnosis is with pyogenic granulomas,<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">6</span></a> whose eruptive presentation has been associated with certain drugs, minor trauma, and Kimura disease (predominantly in oriental patients, associated with peripheral lymphadenopathy, hypereosinophilia, and elevated immunoglobulin E levels).<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">7</span></a> Other conditions with differing degrees of malignancy must also be considered, including hemangioendothelioma and angiosarcoma<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">5</span></a>; the presence of mitoses, cellular atypia, and morphological alterations will orient the diagnosis. A number of theories on the etiology of EHs have been proposed, with debate over whether this is a true neoplasm, a reactive process with a traumatic or infectious origin, or is secondary to an underlying vascular malformation.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">2</span></a> The presence of arteriovenous connections may be observed histologically.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">3</span></a> However, it may require the expertise of an experienced dermatopathologist to identify their presence in venules and capillaries. Imaging studies including MRI or arteriography are recommended to exclude identifiable arteriovenous abnormalities susceptible to treatment, as in our case. Other treatment options described in the literature include surgery, corticosteroids, radiotherapy, intralesional chemotherapeutic agents, cryotherapy, and laser.<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">8</span></a> Recurrences have been reported in around 10% of cases and spontaneous regression has been observed.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">9</span></a></p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of interest</span><p id="par0020" 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: Martínez-Peinado C, Ruiz-Villaverde R, Galán-Gutierrez M, Martínez de Victoria JM. Hemangioma epiteloide múltiple lineal asociado a malformación vascular quiescente. Actas Dermosifiliogr. 2016;107:862–864.</p>" ] ] "multimedia" => array:3 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1188 "Ancho" => 1583 "Tamanyo" => 343628 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Multiple erythematous-violaceous tumors on the left upper arm and forearm in a linear, zosteriform pattern.</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" => 830 "Ancho" => 1534 "Tamanyo" => 258283 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A, Histology showing the vascular proliferation of epithelioid endothelial cells with a solid pattern lining vascular spaces. Hematoxylin and eosin, original magnification ×40. B, Magnetic resonance angiography image of the arteriovenous malformation in the left upper limb, showing occlusion of the proximal humeral artery.</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" => 732 "Ancho" => 900 "Tamanyo" => 108141 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Regression of the distal digital tumors to residual fibrotic tags after the operation on the humeral arteriovenous malformation and basilic-vein by-pass.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:9 [ 0 => array:3 [ "identificador" => "bib0050" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Subcutaneous angiolymphoid hyperplasia with eosinophilia" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "G.C. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 8 | 10 | 18 |
| 2024 October | 78 | 40 | 118 |
| 2024 September | 85 | 19 | 104 |
| 2024 August | 105 | 43 | 148 |
| 2024 July | 97 | 34 | 131 |
| 2024 June | 116 | 28 | 144 |
| 2024 May | 70 | 30 | 100 |
| 2024 April | 79 | 23 | 102 |
| 2024 March | 103 | 28 | 131 |
| 2024 February | 132 | 32 | 164 |
| 2024 January | 54 | 35 | 89 |
| 2023 December | 60 | 20 | 80 |
| 2023 November | 67 | 50 | 117 |
| 2023 October | 72 | 17 | 89 |
| 2023 September | 88 | 25 | 113 |
| 2023 August | 43 | 16 | 59 |
| 2023 July | 71 | 28 | 99 |
| 2023 June | 71 | 26 | 97 |
| 2023 May | 57 | 31 | 88 |
| 2023 April | 75 | 20 | 95 |
| 2023 March | 78 | 26 | 104 |
| 2023 February | 71 | 33 | 104 |
| 2023 January | 52 | 30 | 82 |
| 2022 December | 63 | 30 | 93 |
| 2022 November | 41 | 26 | 67 |
| 2022 October | 22 | 28 | 50 |
| 2022 September | 34 | 21 | 55 |
| 2022 August | 47 | 43 | 90 |
| 2022 July | 34 | 34 | 68 |
| 2022 June | 28 | 25 | 53 |
| 2022 May | 41 | 37 | 78 |
| 2022 April | 69 | 34 | 103 |
| 2022 March | 46 | 36 | 82 |
| 2022 February | 48 | 33 | 81 |
| 2022 January | 53 | 46 | 99 |
| 2021 December | 43 | 34 | 77 |
| 2021 November | 56 | 47 | 103 |
| 2021 October | 67 | 72 | 139 |
| 2021 September | 49 | 50 | 99 |
| 2021 August | 67 | 32 | 99 |
| 2021 July | 100 | 24 | 124 |
| 2021 June | 39 | 24 | 63 |
| 2021 May | 36 | 41 | 77 |
| 2021 April | 74 | 67 | 141 |
| 2021 March | 95 | 21 | 116 |
| 2021 February | 74 | 28 | 102 |
| 2021 January | 36 | 24 | 60 |
| 2020 December | 35 | 19 | 54 |
| 2020 November | 59 | 25 | 84 |
| 2020 October | 33 | 8 | 41 |
| 2020 September | 43 | 17 | 60 |
| 2020 August | 27 | 17 | 44 |
| 2020 July | 25 | 15 | 40 |
| 2020 June | 58 | 31 | 89 |
| 2020 May | 26 | 11 | 37 |
| 2020 April | 39 | 21 | 60 |
| 2020 March | 51 | 16 | 67 |
| 2020 February | 2 | 0 | 2 |
| 2020 January | 3 | 0 | 3 |
| 2019 December | 8 | 0 | 8 |
| 2019 November | 4 | 0 | 4 |
| 2019 September | 4 | 0 | 4 |
| 2019 August | 4 | 0 | 4 |
| 2019 July | 4 | 0 | 4 |
| 2019 June | 6 | 0 | 6 |
| 2019 May | 5 | 0 | 5 |
| 2019 April | 2 | 5 | 7 |
| 2019 March | 6 | 0 | 6 |
| 2019 February | 1 | 0 | 1 |
| 2019 January | 7 | 0 | 7 |
| 2018 December | 3 | 0 | 3 |
| 2018 November | 5 | 0 | 5 |
| 2018 October | 17 | 0 | 17 |
| 2018 September | 4 | 0 | 4 |
| 2018 February | 25 | 5 | 30 |
| 2018 January | 33 | 9 | 42 |
| 2017 December | 50 | 6 | 56 |
| 2017 November | 29 | 7 | 36 |
| 2017 October | 25 | 5 | 30 |
| 2017 September | 23 | 9 | 32 |
| 2017 August | 27 | 8 | 35 |
| 2017 July | 28 | 8 | 36 |
| 2017 June | 35 | 10 | 45 |
| 2017 May | 29 | 4 | 33 |
| 2017 April | 40 | 15 | 55 |
| 2017 March | 29 | 9 | 38 |
| 2017 February | 25 | 6 | 31 |
| 2017 January | 18 | 9 | 27 |
| 2016 December | 52 | 35 | 87 |
| 2016 November | 19 | 25 | 44 |
| 2016 October | 3 | 4 | 7 |
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