Case and Research Letters
Topical Rapamycin: An Additional Therapeutic Option for Tufted Angioma in Adults
Rapamicina tópica: otra opción terapéutica del angioma en penacho en el adulto
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Se observa una placa eritematosa de aspecto vascular en la región anterior del cuello; b: imagen dermatoscópica donde se aprecia un retículo eritematoso; c: varios lóbulos formados por agrupación de vasos y distribuidos en perdigonada en la dermis reticular (H/E<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>2); d: tras 5 meses de tratamiento con rapamicina tópica menos eritema y menos infiltración de la lesión.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "M.M. Escudero-Góngora, O. Corral-Magaña, C. Gómez, A. Martín-Santiago" "autores" => array:4 [ 0 => array:2 [ "nombre" => "M.M." "apellidos" => "Escudero-Góngora" ] 1 => array:2 [ "nombre" => "O." "apellidos" => "Corral-Magaña" ] 2 => array:2 [ "nombre" => "C." "apellidos" => "Gómez" ] 3 => array:2 [ "nombre" => "A." 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Hematoxylin-eosin.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "S. Requena López, C. Maldonado Seral, B. Vivanco Allende" "autores" => array:3 [ 0 => array:2 [ "nombre" => "S." "apellidos" => "Requena López" ] 1 => array:2 [ "nombre" => "C." "apellidos" => "Maldonado Seral" ] 2 => array:2 [ "nombre" => "B." 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Escudero-Góngora, O. Corral-Magaña, C. Gómez, A. Martín-Santiago" "autores" => array:4 [ 0 => array:4 [ "nombre" => "M.M." "apellidos" => "Escudero-Góngora" "email" => array:1 [ 0 => "mmarescudero88@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" => "O." "apellidos" => "Corral-Magaña" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "C." "apellidos" => "Gómez" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "A." "apellidos" => "Martín-Santiago" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicio de Dermatología, Hospital Universitario Son Espases, Palma de Mallorca, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Anatomía Patológica, Hospital Universitario Son Espases, Palma de Mallorca, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Rapamicina tópica: otra opción terapéutica del angioma en penacho en el adulto" ] ] "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" => 1761 "Ancho" => 2917 "Tamanyo" => 338375 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A, Erythematous-violaceous macular lesion on the left breast. B, Dermoscopic image showing a pattern of white lines on a pink background. C, Histological image showing endothelial cells grouped into several lobules, which protrude into dilated, thin-walled channels forming slit-like spaces (hematoxylin-eosin, original magnification ×10).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Tufted angiomas are relatively rare benign vascular proliferations, which were previously referred to as <span class="elsevierStyleItalic">progressive capilliary hemangiomas</span> or <span class="elsevierStyleItalic">angioblastomas</span>.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">1</span></a> They appear mainly in patients under 5 years of age, although adult-onset cases have been reported. They present as erythematous-violaceous plaques or nodules affecting the upper trunk and limbs or the neck. Histologic examination is necessary to establish a definitive diagnosis.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">2</span></a> We report the cases of 3 adults with tufted angiomas diagnosed in our hospital in the past 20 years, highlighting the usefulness of topical rapamycin in one case.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Case Reports</span><p id="par0010" class="elsevierStylePara elsevierViewall">The first patient was a 51-year-old woman with no past medical history of interest, who presented with a lesion that was vascular in appearance. She reported onset some 9 years earlier. Initially the lesion had been asymptomatic, but in the 6 months before the consultation it had grown and become painful. The clinical presentation was a mottled, erythematous lesion on the front and right side of the neck, characterized by superficial telangiectasias. The slightly indurated lesion was about 7<span class="elsevierStyleHsp" style=""></span>cm wide and we detected no thrill or increase in temperature (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A). Dermoscopy revealed an erythematous reticular pattern (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>B). Histologic examination showed a proliferation of vessels grouped into lobules distributed throughout the reticular dermis in a cannonball pattern (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>C). These findings led to a diagnosis of tufted angioma. As the lesion was painful, treatment with rapamycin 0.4% was started using a topical preparation comprising rapamycin powder in a cream base. After 5 months of treatment, the lesion was no longer palpable, the coloration had diminished (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>D), and the patient reported no pain.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">The second patient was a 24-year-old woman with a history of familial Mediterranean fever, who presented with an erythematous-violaceous macular lesion 2<span class="elsevierStyleHsp" style=""></span>cm in diameter, which had first appeared 11 years earlier (<a class="elsevierStyleCrossRef" href="#fig0010">Figs. 2</a>A and B). The asymptomatic lesion was located on the left breast. Histologic examination showed clusters of endothelial cells in the reticular dermis, forming lobules that protruded into dilated, thin-walled vascular channels, forming slit-like spaces (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>C). These findings confirmed the diagnosis of tufted angioma. Given the absence of symptoms, no treatment was prescribed.</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">The third patient was a 66-year-old man who presented 17 years ago with a verrucous lesion measuring approximately 2<span class="elsevierStyleHsp" style=""></span>cm in diameter on the anterior face of his right hand. The lesion had appeared some months earlier and bled frequently. Complete surgical excision of the tumor was performed. The diagnosis of tufted angioma was reached on the basis of the symptoms and the histologic findings.</p><p id="par0025" class="elsevierStylePara elsevierViewall">Cases of tufted angioma in adults are very rare, with fewer than 50 cases reported in the literature, of which only one was associated with Kassabach-Merritt phenomenon.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">3</span></a> In general, these tumors are characterized by slow and gradual growth followed by stabilization. Spontaneous regression has also been reported. No differences have been reported between adults and children in the presentation. The morbidity of these lesions is mainly related to the pain caused by the tumor, as was the case in our first patient.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Tufted angioma and kaposiform hemangioendothelioma share certain clinical and histopathologic features and it is thought that the two entities may represent the opposite poles of a spectrum of vascular lesions.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">4</span></a> They also share a common complication known as Kasabach-Merritt syndrome: consumptive coagulopathy with severe thrombocytopenia. This syndrome is rare in adults. The characteristic histologic feature of tufted angioma is the presence of lobular clusters of capillaries forming tufts within the dermis. The differential diagnosis includes several other vascular anomalies.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">1</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Complicated tufted angioma in childhood is treated systemically with vincristine, ticlopidine, and acetylsalicylic acid<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">5</span></a>; recently, rapamycin has emerged as a very effective alternative.<a class="elsevierStyleCrossRefs" href="#bib0075"><span class="elsevierStyleSup">6,7</span></a> In uncomplicated cases, Javvaji et al.<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">8</span></a> report good results with low dose aspirin. Clinical management in adults is not well established, and the evidence does not support any definite conclusions as to what the first-line therapy should be. The treatment options described in the literature, with variable results, include pulsed dye laser, topical corticosteroids, intralesional interferon-alfa, surgery, and a watch-and-wait approach.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">3</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Of our 3 cases, one was resolved with surgery, another is being followed up, and the third—a painful neck lesion—improved after topical treatment with 0.4% rapamycin ointment.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Oral rapamycin has become the treatment of choice for complicated tufted angioma in children.<a class="elsevierStyleCrossRefs" href="#bib0075"><span class="elsevierStyleSup">6,7</span></a> There has been no experience to date of the use of topical rapamycin in the treatment of this type of tumor. However, in view of the demonstrated efficacy of rapamycin in the treatment of facial angiofibromas in tuberous sclerosis,<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">8,9</span></a> and in isolated cases of lymphatic malformation, we suggest that topical rapamycin may be another option for the treatment of symptomatic tufted hemangioma not associated with coagulopathy.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Conflict of interests</span><p id="par0050" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:3 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Case Reports" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Conflict of interests" ] 2 => 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: Escudero-Góngora MM, Corral-Magaña O, Gómez C, Martín-Santiago A. Rapamicina tópica: otra opción terapéutica del angioma en penacho en el adulto. Actas Dermosifiliogr. 2018;109:192–194.</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" => 2188 "Ancho" => 2917 "Tamanyo" => 323285 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A, Clinical image. Erythematous plaque, vascular in appearance, on the anterior region of the neck. B, Dermoscopic image showing an erythematous reticular pattern. C, Groups of vessels forming several lobes distributed in a canonball pattern throughout the reticular dermis (hematoxylin-eosin, original magnification ×2). D, Reduced erythema and infiltration following 5 months of treatment with topical rapamycin.</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" => 1761 "Ancho" => 2917 "Tamanyo" => 338375 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A, Erythematous-violaceous macular lesion on the left breast. B, Dermoscopic image showing a pattern of white lines on a pink background. C, Histological image showing endothelial cells grouped into several lobules, which protrude into dilated, thin-walled channels forming slit-like spaces (hematoxylin-eosin, original magnification ×10).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:9 [ 0 => array:3 [ "identificador" => "bib0050" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Vascular tumours in infants. Part II: Vascular tumours of intermediate malignancy and malignant tumours" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "I. Colmenero" 1 => "P.H. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 11 | 14 | 25 |
| 2024 October | 76 | 49 | 125 |
| 2024 September | 95 | 31 | 126 |
| 2024 August | 112 | 58 | 170 |
| 2024 July | 82 | 35 | 117 |
| 2024 June | 78 | 39 | 117 |
| 2024 May | 79 | 51 | 130 |
| 2024 April | 92 | 25 | 117 |
| 2024 March | 99 | 30 | 129 |
| 2024 February | 102 | 36 | 138 |
| 2024 January | 61 | 40 | 101 |
| 2023 December | 71 | 17 | 88 |
| 2023 November | 80 | 31 | 111 |
| 2023 October | 68 | 26 | 94 |
| 2023 September | 88 | 35 | 123 |
| 2023 August | 54 | 15 | 69 |
| 2023 July | 79 | 19 | 98 |
| 2023 June | 62 | 23 | 85 |
| 2023 May | 65 | 23 | 88 |
| 2023 April | 51 | 22 | 73 |
| 2023 March | 51 | 31 | 82 |
| 2023 February | 52 | 21 | 73 |
| 2023 January | 57 | 29 | 86 |
| 2022 December | 60 | 44 | 104 |
| 2022 November | 28 | 36 | 64 |
| 2022 October | 26 | 19 | 45 |
| 2022 September | 24 | 38 | 62 |
| 2022 August | 25 | 33 | 58 |
| 2022 July | 22 | 36 | 58 |
| 2022 June | 25 | 21 | 46 |
| 2022 May | 44 | 39 | 83 |
| 2022 April | 51 | 20 | 71 |
| 2022 March | 76 | 35 | 111 |
| 2022 February | 50 | 19 | 69 |
| 2022 January | 62 | 31 | 93 |
| 2021 December | 52 | 42 | 94 |
| 2021 November | 49 | 48 | 97 |
| 2021 October | 68 | 56 | 124 |
| 2021 September | 41 | 29 | 70 |
| 2021 August | 43 | 35 | 78 |
| 2021 July | 33 | 28 | 61 |
| 2021 June | 40 | 25 | 65 |
| 2021 May | 46 | 36 | 82 |
| 2021 April | 80 | 44 | 124 |
| 2021 March | 92 | 31 | 123 |
| 2021 February | 66 | 29 | 95 |
| 2021 January | 41 | 14 | 55 |
| 2020 December | 47 | 19 | 66 |
| 2020 November | 38 | 24 | 62 |
| 2020 October | 29 | 16 | 45 |
| 2020 September | 39 | 14 | 53 |
| 2020 August | 31 | 23 | 54 |
| 2020 July | 41 | 20 | 61 |
| 2020 June | 44 | 29 | 73 |
| 2020 May | 15 | 9 | 24 |
| 2020 April | 29 | 16 | 45 |
| 2020 March | 33 | 12 | 45 |
| 2020 February | 4 | 0 | 4 |
| 2020 January | 4 | 0 | 4 |
| 2019 December | 8 | 0 | 8 |
| 2019 November | 4 | 0 | 4 |
| 2019 September | 11 | 0 | 11 |
| 2019 August | 4 | 0 | 4 |
| 2019 July | 4 | 0 | 4 |
| 2019 June | 4 | 0 | 4 |
| 2019 May | 9 | 0 | 9 |
| 2019 April | 2 | 0 | 2 |
| 2019 March | 2 | 0 | 2 |
| 2019 February | 2 | 0 | 2 |
| 2018 December | 5 | 0 | 5 |
| 2018 November | 3 | 0 | 3 |
| 2018 September | 3 | 0 | 3 |
| 2018 February | 3 | 21 | 24 |
| 2018 January | 6 | 12 | 18 |
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