Case and Research Letters
Acquired Port-Wine Stain (Fegeler Syndrome): A Report of 3 Cases
Malformación capilar adquirida (síndrome de Fegeler): 3 casos
J.F. Millán-Cayetano
, J. del Boz, P. García-Montero, M. de Troya-Martín
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Servicio de Dermatología, Hospital Costa del Sol, Marbella, Málaga, Spain
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Caso 1: mácula rojiza que afecta a la primera y segunda rama del trigémino (punteado) con zona periocular de tonalidad más violácea (flecha). B. Caso 2: máculas rosadas parcheadas (punteado). C. Aspecto de las lesiones anteriores tras realizar la paciente ejercicio físico, volviéndose más evidentes. D. Caso 3: mácula única de tonalidad rojiza (punteado). E. Estado de la lesión previa tras realizar el paciente esfuerzo físico percibiéndose más intensas.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "J.F. Millán-Cayetano, J. del Boz, P. García-Montero, M. de Troya-Martín" "autores" => array:4 [ 0 => array:2 [ "nombre" => "J.F." "apellidos" => "Millán-Cayetano" ] 1 => array:2 [ "nombre" => "J." "apellidos" => "del Boz" ] 2 => array:2 [ "nombre" => "P." "apellidos" => "García-Montero" ] 3 => array:2 [ "nombre" => "M." 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Millán-Cayetano, J. del Boz, P. García-Montero, M. de Troya-Martín" "autores" => array:4 [ 0 => array:4 [ "nombre" => "J.F." "apellidos" => "Millán-Cayetano" "email" => array:1 [ 0 => "jf.millancayetano@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "J." "apellidos" => "del Boz" ] 2 => array:2 [ "nombre" => "P." "apellidos" => "García-Montero" ] 3 => array:2 [ "nombre" => "M." "apellidos" => "de Troya-Martín" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Servicio de Dermatología, Hospital Costa del Sol, Marbella, Málaga, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Malformación capilar adquirida (síndrome de Fegeler): 3 casos" ] ] "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" => 1586 "Ancho" => 3417 "Tamanyo" => 449888 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A, Patient<span class="elsevierStyleHsp" style=""></span>1. A reddish macule affecting the first and second branches of the trigeminal nerve (dotted line), with a more violaceous color in the periocular region (arrow). B, Patient<span class="elsevierStyleHsp" style=""></span>2. Patchy pink macules (dotted line). C, More intense appearance of the lesions in B after the patient performed physical exercise. D, Patient<span class="elsevierStyleHsp" style=""></span>3. Single macule of reddish color (dotted line). E, More intense appearance of the lesion in D after the patient performed physical exercise.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Capillary malformation is a condition included in the group of vascular malformations.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> It is one of the most common vascular abnormalities and can affect up to 0.3% of newborns.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2,3</span></a> It presents clinically as a homogeneous erythematous macule with well-defined borders. It is typically unilateral and is usually present at birth. However, cases of acquired capillary malformation have been reported in patients with no previous lesions of this type.</p><p id="par0010" class="elsevierStylePara elsevierViewall">Patient 1 was a 9-year-old girl followed-up from her first year of life for mastocytosis presenting as urticaria pigmentosa and treated with sodium cromoglycate and cetirizine. When she was 7 years old, a well-defined pink macule started to become visible, affecting segmentally the area of the first branch of the trigeminal nerve, and the superior part of the area of the second branch, with a more violaceous appearance in the periocular region. She presented no other lesions and there was no family history of interest. Skin biopsy revealed a capillary proliferation, negative for GLUT-1, consistent with a diagnosis of capillary malformation.</p><p id="par0015" class="elsevierStylePara elsevierViewall">Patient 2 was a girl aged 8 years, with no past history of interest. She was referred to our clinic for macules on the left side of her face. The macules had first been noticed by her parents when the child was 4 years old, and they had grown progressively. Her parents stated that the macules became more evident in hot environments and after physical exertion. On examination, patchy pink macules were observed in a segmental distribution along the left body of the jaw and they were seen to become more intense after the patient performed physical exercise.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Patient 3 was a 7-year-old boy with no past history of interest. He was seen for a lesion in the right jaw region. His parents had first noticed the lesion when the child was about 4 years old. They stated that it had been asymptomatic, although its intensity increased with physical activity and with heat. On examination, a patchy pink macule with superficial telangiectasias was observed. The macule became more intense after the child performed physical exercise.</p><p id="par0025" class="elsevierStylePara elsevierViewall">None of the 3 patients had any family history of interest or presented other lesions. Clinically there was no thrill and the lesions were not detectable on ultrasound. Photos of the children when they were younger were requested to check that the lesions had not previously escaped the parents’ attention. The children were referred to a reference center for laser treatment, which was only rejected by the third patient (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">The immense majority of capillary malformations are present at birth or appear in the first years of life. Cases of acquired capillary malformations are rare and were first described in 1939 in a patient who had suffered spinal cord injury.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a> Ten years later, Fegeler<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> reported a new case of postraumatic capillary malformation, which has been named Fegeler syndrome since that time. The etiological factors that can trigger their de novo appearance have not been identified, though a history of trauma is present in up to a third of cases. Previously it has been proposed that the appearance of congenital capillary malformations could be due to a deficit of sympathetic innervation (responsible for vasoconstriction), leading to vascular ectasia that presents as this vascular malformation.<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a> Acquired cases associated with spinal trauma conform to this hypothesis. Other possible etiologies have also been proposed, such as estrogenic impregnation, either during pregnancy or in the pubertal-adult period.<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">7,8</span></a> In some isolated cases, capillary malformations have been related with drugs, herpes zoster, sun damage, cluster headaches, and acoustic neuroma.<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">7,8</span></a> In a capillary malformation of the port wine stain type, the possibility of a postnatal mutation of the <span class="elsevierStyleItalic">GNAQ</span> gene might also be considered.<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a> Despite these proposals, no triggering event has been discovered in many cases. The age at onset reported in the literature varies between 3 and 69 years, with a mean age of 25 years. Lesions most often arise on the face and upper limbs, and they have not been associated with other malformations or skin lesions. Typical and acquired capillary malformations are histologically and morphologically indistinguishable.</p><p id="par0035" class="elsevierStylePara elsevierViewall">The differential diagnosis based on the type of vascular lesion is extensive. But perhaps it is most important to rule out this lesion as part of a syndromic diagnosis associated with more serious repercussions than the purely cosmetic. These include capillary malformation-arteriovenous malformation syndrome (a family history of similar lesions or of arteriovenous malformation is usually detected, the capillary malformations are multiple, with a browner color, increased local temperature, a whitish peripheral halo, and possibly with arterial flow visible on ultrasound) and Sturge-Weber syndrome (lesions that affect one side of the face, but that are usually present at birth).<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">3,10</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">In summary, acquired capillary malformation may be considered simply to be a late-onset capillary malformation with a variable latency period.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of Interest</span><p id="par0045" 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: Millán-Cayetano JF, del Boz J, García-Montero P, de Troya-Martín M. Malformación capilar adquirida (síndrome de Fegeler): 3 casos. Actas Dermosifiliogr. 2017;108:954–955.</p>" ] ] "multimedia" => array:1 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1586 "Ancho" => 3417 "Tamanyo" => 449888 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A, Patient<span class="elsevierStyleHsp" style=""></span>1. A reddish macule affecting the first and second branches of the trigeminal nerve (dotted line), with a more violaceous color in the periocular region (arrow). B, Patient<span class="elsevierStyleHsp" style=""></span>2. Patchy pink macules (dotted line). C, More intense appearance of the lesions in B after the patient performed physical exercise. D, Patient<span class="elsevierStyleHsp" style=""></span>3. Single macule of reddish color (dotted line). E, More intense appearance of the lesion in D after the patient performed physical exercise.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0055" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:1 [ "referenciaCompleta" => "ISSVA Classification of Vascular Anomalies ©2014 International Society for the Study of Vascular Anomalies. Available at issva.org/classification. Accesed April 2014." ] ] ] 1 => array:3 [ "identificador" => "bib0060" "etiqueta" => "2" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Vascular anomalies-A practical approach" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "M. Theiler" 1 => "R. Wälchli" 2 => "L. 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| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 14 | 9 | 23 |
| 2024 Octubre | 129 | 46 | 175 |
| 2024 Septiembre | 124 | 37 | 161 |
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| 2024 Febrero | 104 | 28 | 132 |
| 2024 Enero | 75 | 33 | 108 |
| 2023 Diciembre | 79 | 21 | 100 |
| 2023 Noviembre | 94 | 28 | 122 |
| 2023 Octubre | 119 | 20 | 139 |
| 2023 Septiembre | 105 | 39 | 144 |
| 2023 Agosto | 68 | 10 | 78 |
| 2023 Julio | 91 | 32 | 123 |
| 2023 Junio | 69 | 19 | 88 |
| 2023 Mayo | 107 | 27 | 134 |
| 2023 Abril | 97 | 26 | 123 |
| 2023 Marzo | 109 | 28 | 137 |
| 2023 Febrero | 84 | 25 | 109 |
| 2023 Enero | 84 | 25 | 109 |
| 2022 Diciembre | 76 | 44 | 120 |
| 2022 Noviembre | 79 | 28 | 107 |
| 2022 Octubre | 46 | 24 | 70 |
| 2022 Septiembre | 58 | 40 | 98 |
| 2022 Agosto | 54 | 38 | 92 |
| 2022 Julio | 42 | 32 | 74 |
| 2022 Junio | 50 | 31 | 81 |
| 2022 Mayo | 75 | 56 | 131 |
| 2022 Abril | 105 | 46 | 151 |
| 2022 Marzo | 103 | 72 | 175 |
| 2022 Febrero | 104 | 33 | 137 |
| 2022 Enero | 106 | 44 | 150 |
| 2021 Diciembre | 168 | 42 | 210 |
| 2021 Noviembre | 92 | 50 | 142 |
| 2021 Octubre | 89 | 70 | 159 |
| 2021 Septiembre | 68 | 44 | 112 |
| 2021 Agosto | 82 | 50 | 132 |
| 2021 Julio | 75 | 21 | 96 |
| 2021 Junio | 68 | 45 | 113 |
| 2021 Mayo | 62 | 48 | 110 |
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| 2021 Enero | 52 | 16 | 68 |
| 2020 Diciembre | 31 | 18 | 49 |
| 2020 Noviembre | 36 | 24 | 60 |
| 2020 Octubre | 39 | 15 | 54 |
| 2020 Septiembre | 45 | 14 | 59 |
| 2020 Agosto | 39 | 22 | 61 |
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| 2020 Junio | 27 | 30 | 57 |
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| 2020 Marzo | 26 | 11 | 37 |
| 2020 Febrero | 5 | 2 | 7 |
| 2020 Enero | 2 | 0 | 2 |
| 2019 Diciembre | 4 | 0 | 4 |
| 2019 Noviembre | 2 | 0 | 2 |
| 2019 Septiembre | 4 | 0 | 4 |
| 2019 Agosto | 4 | 0 | 4 |
| 2019 Julio | 4 | 0 | 4 |
| 2019 Junio | 4 | 0 | 4 |
| 2019 Mayo | 4 | 0 | 4 |
| 2019 Abril | 3 | 0 | 3 |
| 2019 Marzo | 3 | 0 | 3 |
| 2019 Febrero | 2 | 0 | 2 |
| 2018 Diciembre | 4 | 0 | 4 |
| 2018 Noviembre | 2 | 0 | 2 |
| 2018 Octubre | 3 | 0 | 3 |
| 2018 Septiembre | 4 | 0 | 4 |
| 2018 Junio | 1 | 0 | 1 |
| 2018 Abril | 2 | 0 | 2 |
| 2018 Febrero | 39 | 7 | 46 |
| 2018 Enero | 218 | 13 | 231 |
| 2017 Diciembre | 69 | 43 | 112 |
| 2017 Noviembre | 9 | 11 | 20 |
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