Case and Research Letters
Solitary Glomus Tumor Presenting as a Telangiectatic Macule
Mácula telangiectásica como forma de presentación de un tumor glómico solitario
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Allegue, D. González-Vilas, C. Fachal, A. Zulaica" "autores" => array:4 [ 0 => array:4 [ "nombre" => "F." "apellidos" => "Allegue" "email" => array:1 [ 0 => "fallegue@mundo-r.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "D." "apellidos" => "González-Vilas" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "C." "apellidos" => "Fachal" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "A." "apellidos" => "Zulaica" "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 do Meixoeiro, EOXI Vigo, Vigo, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Anatomía Patológica, Hospital Álvaro Cunqueiro, EOXI Vigo, Vigo, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Mácula telangiectásica como forma de presentación de un tumor glómico solitario" ] ] "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" => 755 "Tamanyo" => 75460 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Erythematous-pink macule surrounded by a pale halo.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Glomus tumors of the skin are neoplasms derived from dermal glomus cells. These are modified smooth muscle cells and form part of the glomus body, a neuromyoarterial structure that serves to regulate blood flow to the skin.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">1</span></a> These tumors are usually classified according to the predominant cell type or vessels. Thus, tumors are denoted glomus tumors when there is high cellularity, the most frequent variety; glomangiomas when a vascular pattern predominates; and glomangiomyoma if abundant smooth muscle is present.<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">2,3</span></a> Generally, solid glomus tumors are painful acquired nodules, whereas glomangiomas usually present as multiple lesions and are congenital or onset occurs at a young age. Not infrequently, they may be hereditary, and so are considered as a different entity. They are denoted glomuvenous malformations and a relation to a mutation on chromosome 1p21-22 has been found,<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">4</span></a> in a gene that encodes glomulin.<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">5</span></a> We report the case of a solitary glomus tumor with a distinctive clinical presentation, in form of a telangiectatic macule.</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 60-year-old woman with no medical history of interest was assessed for a painful lesion on the internal face of the left thigh. The lesion had been present for 2 years and had appeared spontaneously, with no apparent relationship with prior trauma. The patient reported pain on touch and pressure, but not spontaneously or with changes in temperature. She did not report any similar lesions in family members. Examination showed a macule with telangiectatic appearance, measuring 1.5<span class="elsevierStyleHsp" style=""></span>cm in diameter, which was painful to palpation, and no other indications of dermal or epidermal involvement (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). Dermatoscopy showed a small blue-grey poorly delimited structure immersed in an extensive fine network with a reticular configuration (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). The lesion was resected, and histopathological study showed a solid, well-delimited nodule, measuring 6<span class="elsevierStyleHsp" style=""></span>mm, located in the hypodermis, formed of monomorphic, rounded cells with oval nuclei, without atypia, that surrounded dilated vessels with thin walls. The cells expressed actin in smooth muscle and endothelial markers in vessels. In the papillary dermis, vessels were observed with dilated lumens, but without glomus cells nearby (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). The diagnosis was of a solitary glomus tumor.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Solitary glomus tumor, given its origin in the glomus body, can appear anywhere on the skin, but it is much more frequent at sites where these structures are more numerous, that is, fingers and particularly the subungual region. They have also been reported ectopically in internal organs, where normally these structures do not appear. Solitary glomus tumors are thus divided into digital and extradigital.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">1,3</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">The characteristic symptomatic triad of these tumors is spontaneous pain, painful hypersensitivity to touch or pressure, and pain triggered by cold; these symptoms therefore enable rapid diagnostic suspicion in the event of pain in the fingers or subungual region.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">1,3</span></a> However, extradigital solitary glomus tumors do not usually present with the full range of symptoms, particularly pain triggered by cold,<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">1,3</span></a> and so diagnosis at these sites is usually delayed 6 or 7 years, with multiple visits to the clinic and even psychological repercussion.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">1,3</span></a> Extradigital solitary glomus tumors usually present as erythematoviolaceous or bluish macules or papules, subcutaneous nodules, or even without clinical findings. In our patient, we could not palpate the nodule, although this was in principle accessible, because of the intense pain with the merest of brushes of the area with the telangiectatic macule. Imaging studies, such as magnetic resonance imaging<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">1</span></a> and high-frequency ultrasonography,<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">6</span></a> can help with diagnosis prior to histopathology. In ultrasound imaging, solitary glomus tumors appear as a well-delimited, hypoechoic oval or rounded lesion in the dermis, occasionally known as the <span class="elsevierStyleItalic">stalk sign</span> (presence of a hypoechoic vascularized prolongation that connects the lesion to adjacent tissues).<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">6,7</span></a> In addition to these findings in B mode, Doppler mode provides additional signs: lesions generally well vascularized, with prominent vessels within, but without forming true <span class="elsevierStyleItalic">vascular lakes</span>, and predominantly arterial flows in pulsed Doppler studies, without clearly visualizing an arteriovenous shunt.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">7</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">This form of presentation as a telangiectatic macule is exceptional and is not clearly reflected in the literature of solid solitary glomus tumors. Schiefer et al.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">1</span></a> mentioned a small, painful, telangiectatic lesion that served as a guide to detect a solitary glomus tumor measuring 6<span class="elsevierStyleHsp" style=""></span>mm on the subcutaneous cell tissue. There is also a rare clinical-pathological variant of glomangioma with a certain resemblance. In 1998, Requena et al.<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">8</span></a> reported a plaque-like telangiectatic glomangioma as a new entity based on the telangiectatic appearance and its acquired nature. In 2013, Farias et al.<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">9</span></a> reported another case of plaque-like telangiectatic plaque glomangioma. Apart from the clinical appearance of telangiectatic macula, they also have other features in common with those of our patient's lesion: they are solitary, acquired, painful lesions that occur in women. In 2007, Monteagudo et al.<a class="elsevierStyleCrossRef" href="#bib0105"><span class="elsevierStyleSup">10</span></a> published another case of solitary glomangioma as a telangiectatic plaque in a 41-year-old women. The lesion was very similar but for the fact that it was congenital and asymptomatic. Unlike our case, these 3 lesions were larger and, histopathologically, they were glomangiomas.</p><p id="par0030" class="elsevierStylePara elsevierViewall">We are unsure of the etiopathogenesis of these telangiectasias. We do not know whether they are induced by the tumor or are a reactive phenomenon, but in all cases, we believe that this is not a coincidence. Differential diagnosis could be considered with those processes that can present a telangiectatic appearance, such as mastocytosis; elastotic hemangioma; post-traumatic or drug-induced telangiectasias; angioma serpiginosum; collagen vasculopathy; generalized essential telangiectasia; acquired capillary malformation (Fegeler syndrome); those processes considered as reactive cutaneous angiomatoses, such as reactive angioendotheliomatosis, diffuse dermal angiomatosis, acroangiodermatitis, intravascular reactive histiocytosis, glomeruloid reactive angioendotheliomatosis, and angipericytomatosis<a class="elsevierStyleCrossRef" href="#bib0110"><span class="elsevierStyleSup">11</span></a>; as well as Kaposi sarcoma. However, the clinical presentation of a single painful lesion and the histopathology enable diagnosis to be established beyond doubt.</p><p id="par0035" class="elsevierStylePara elsevierViewall">We describe an unusual presentation for solitary glomus tumor, with the appearance of a telangiectatic macule. It would be important to take into account such a presentation when assessing local and unexplained pain in extradigital regions so as not to delay diagnosis and to avoid the associated physical and psychological discomfort.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of Interest</span><p id="par0040" 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: Allegue F, González-Vilas D, Fachal C, Zulaica A. Mácula telangiectásica como forma de presentación de un tumor glómico solitario. Actas Dermosifiliogr. 2020;111:434–436.</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" => 755 "Ancho" => 755 "Tamanyo" => 75460 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Erythematous-pink macule surrounded by a pale halo.</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" => 707 "Ancho" => 755 "Tamanyo" => 52293 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Dermatoscopy showing a fine telangiectatic network and poorly-defined blue-grey area.</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" => 1005 "Ancho" => 755 "Tamanyo" => 271914 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Vascular dilatations in the dermis and a well-delimited, highly vascularized, nodule located in the hypodermis. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 11 | 17 | 28 |
| 2024 October | 66 | 47 | 113 |
| 2024 September | 86 | 36 | 122 |
| 2024 August | 128 | 59 | 187 |
| 2024 July | 126 | 30 | 156 |
| 2024 June | 105 | 31 | 136 |
| 2024 May | 96 | 34 | 130 |
| 2024 April | 67 | 29 | 96 |
| 2024 March | 100 | 29 | 129 |
| 2024 February | 82 | 34 | 116 |
| 2024 January | 64 | 33 | 97 |
| 2023 December | 70 | 22 | 92 |
| 2023 November | 92 | 50 | 142 |
| 2023 October | 63 | 24 | 87 |
| 2023 September | 68 | 34 | 102 |
| 2023 August | 56 | 19 | 75 |
| 2023 July | 66 | 49 | 115 |
| 2023 June | 59 | 23 | 82 |
| 2023 May | 70 | 28 | 98 |
| 2023 April | 71 | 18 | 89 |
| 2023 March | 84 | 35 | 119 |
| 2023 February | 66 | 23 | 89 |
| 2023 January | 65 | 35 | 100 |
| 2022 December | 108 | 35 | 143 |
| 2022 November | 56 | 35 | 91 |
| 2022 October | 45 | 32 | 77 |
| 2022 September | 62 | 47 | 109 |
| 2022 August | 94 | 44 | 138 |
| 2022 July | 94 | 49 | 143 |
| 2022 June | 51 | 48 | 99 |
| 2022 May | 60 | 35 | 95 |
| 2022 April | 109 | 28 | 137 |
| 2022 March | 99 | 50 | 149 |
| 2022 February | 113 | 26 | 139 |
| 2022 January | 108 | 35 | 143 |
| 2021 December | 71 | 40 | 111 |
| 2021 November | 85 | 38 | 123 |
| 2021 October | 71 | 44 | 115 |
| 2021 September | 50 | 27 | 77 |
| 2021 August | 39 | 29 | 68 |
| 2021 July | 41 | 17 | 58 |
| 2021 June | 42 | 22 | 64 |
| 2021 May | 35 | 32 | 67 |
| 2021 April | 70 | 87 | 157 |
| 2021 March | 73 | 26 | 99 |
| 2021 February | 44 | 34 | 78 |
| 2021 January | 39 | 22 | 61 |
| 2020 December | 40 | 15 | 55 |
| 2020 November | 38 | 12 | 50 |
| 2020 October | 44 | 8 | 52 |
| 2020 September | 59 | 19 | 78 |
| 2020 August | 69 | 17 | 86 |
| 2020 July | 83 | 24 | 107 |
| 2020 June | 17 | 12 | 29 |
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