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"apellidos" => "Toribio" ] ] ] ] ] "idiomaDefecto" => "en" "Traduccion" => array:1 [ "es" => array:9 [ "pii" => "S0001731011004728" "doi" => "10.1016/j.ad.2011.06.010" "estado" => "S300" "subdocumento" => "" "abierto" => array:3 [ "ES" => true "ES2" => true "LATM" => true ] "gratuito" => true "lecturas" => array:1 [ "total" => 0 ] "idiomaDefecto" => "es" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0001731011004728?idApp=UINPBA000044" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1578219012001473?idApp=UINPBA000044" "url" => "/15782190/0000010300000004/v1_201304241303/S1578219012001473/v1_201304241303/en/main.assets" ] "en" => array:15 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Case and Research Letters</span>" "titulo" => "Encephalocraniocutaneous Lipomatosis and Didymosis Aplasticopsilolipara" "tieneTextoCompleto" => true "saludo" => "<span class="elsevierStyleItalic">To the Editor:</span>" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "341" "paginaFinal" => "342" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "N. Martí, V. Alonso, E. Jordá" "autores" => array:3 [ 0 => array:4 [ "nombre" => "N." "apellidos" => "Martí" "email" => array:1 [ 0 => "nuriamarfa@hotmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">¿</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "V." "apellidos" => "Alonso" ] 2 => array:2 [ "nombre" => "E." "apellidos" => "Jordá" ] ] "afiliaciones" => array:1 [ 0 => array:1 [ "entidad" => "Servicio de Dermatología, Hospital Clínico Universitario de Valencia, Valencia, Spain" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Lipomatosis encefalocraneocutánea y didimosis aplasticopsilolípara" ] ] "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" => 741 "Ancho" => 950 "Tamanyo" => 106664 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Nevus psiloliparus adjacent to an aplasia cutis congenita lesion.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Nevus psiloliparus is clinically observed as a well-defined, oval or round, hairless area with a smooth surface located in the parietal or frontoparietal region. Histological examination shows a skin lesion with rudimentary hair follicles and cords of adipose tissue penetrating the dermis. This nevus is the main hallmark of encephalocraniocutaneous lipomatosis (ECCL), otherwise known as Haberland syndrome<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1-5</span></a>; however, 2 cases of nevus psiloliparus have been described without neurological or ocular involvement.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> ECCL is a very rare form of neurocutaneous syndrome clinically characterized by lipomatous hamartomas on the face and scalp, ocular abnormalities, and ipsilateral malformations of the central nervous system. We report the case of a neonate with nevus psiloliparus and aplasia cutis congenita (ACC) on the scalp—an association known as a didymosis aplasticopsilolipara—in addition to ocular lesions (lipodermoid cysts and coloboma), a right temporal arachnoid cyst, and aortic coarctation.</p><p id="par0010" class="elsevierStylePara elsevierViewall">The patient was a 6-day-old neonate, born at term after an uncomplicated pregnancy and normal vaginal delivery; he was the first child of unrelated healthy parents. Physical examination showed a hairless, well-defined, soft, yellowish area adjacent to a bright red, hairless, circumscribed lesion covered by a thin transparent membrane in the right frontoparietal region (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). In addition, 2 yellow papules were observed on the right upper eyelid (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>); these were diagnosed by the ophthalmologist as lipodermoid cysts associated with an ipsilateral coloboma. Neurological examination was normal. Histopathologic examination of the hairless area of the scalp showed abundant fatty tissue, rudimentary hair follicles, and isolated arrector pili muscles; the findings were compatible with nevus psiloliparus. Brain magnetic resonance imaging (MRI) showed an arachnoid cyst occupying the right temporal fossa (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>) and echocardiography revealed moderate aortic coarctation.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">The clinical, histological, and brain MRI findings led to a diagnosis of ECCL.</p><p id="par0020" class="elsevierStylePara elsevierViewall">ECCL is a neurocutaneous syndrome of unknown etiology. Described in 1970 by Haberland and Perou,<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> it is characterized by unilateral lesions in tissues of ectodermal and mesodermal origin: skin, eye, adipose tissue, and brain. Aortic coarctation, progressive bone cysts, and jaw tumors have also been described in association with this syndrome.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> The differential diagnosis with other neurocutaneous mosaicisms, such as Delleman, Goltz, Goldenhar, or Proteus syndromes, can be very difficult due to the large number of overlapping features.</p><p id="par0025" class="elsevierStylePara elsevierViewall">Nevus psiloliparus in close association with ACC was first described in 2 patients by Torrelo et al.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> in 2005; a third case was reported by Stieler et al.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> in 2008. This association, known as didymosis aplasticopsilolipara, has been described as an example of nonallelic twin spotting.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2,8</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Our case is the fourth report of ACC and nevus psiloliparus in association with ECCL. The fact that this association has been described in isolated cases only suggests that it is purely coincidental; however, we consider that if new cases of ECCL with this characteristic clinical presentation are reported, we may need to consider the possibility that ACC and nevus psiloliparus form part of the clinical spectrum of this neurocutaneous syndrome.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara">Please cite this article as: Martí N, Alonso V, Jordá E. Lipomatosis encefalocraneocutánea y didimosis aplasticopsilolípara. Actas Dermosifiliogr.2012;103:341-342.</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" => 741 "Ancho" => 950 "Tamanyo" => 106664 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Nevus psiloliparus adjacent to an aplasia cutis congenita lesion.</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" => 539 "Ancho" => 950 "Tamanyo" => 77454 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Two lipodermoid cysts on the right upper eyelid.</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" => 984 "Ancho" => 1500 "Tamanyo" => 226871 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Arachnoid cyst occupying the right temporal fossa. A, brain axial <span class="elsevierStyleItalic">T</span><span class="elsevierStyleInf"><span class="elsevierStyleItalic">1</span></span>-weighted magnetic resonance imaging; B, brain axial <span class="elsevierStyleItalic">T</span><span class="elsevierStyleInf"><span class="elsevierStyleItalic">2</span></span>-weighted magnetic resonance imaging.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:8 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Nevus psiloliparus and aplasia cutis: A further possible example of didymosis" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "A. Torrelo" 1 => "M.C. Boente" 2 => "O. Nieto" 3 => "R. Asial" 4 => "I. Colmenero" 5 => "B. 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2021 Mayo | 72 | 46 | 118 |
2021 Abril | 174 | 53 | 227 |
2021 Marzo | 88 | 23 | 111 |
2021 Febrero | 63 | 41 | 104 |
2021 Enero | 38 | 24 | 62 |
2020 Diciembre | 40 | 16 | 56 |
2020 Noviembre | 35 | 23 | 58 |
2020 Octubre | 23 | 8 | 31 |
2020 Septiembre | 23 | 17 | 40 |
2020 Agosto | 30 | 24 | 54 |
2020 Julio | 30 | 21 | 51 |
2020 Junio | 33 | 34 | 67 |
2020 Mayo | 26 | 17 | 43 |
2020 Abril | 36 | 24 | 60 |
2020 Marzo | 24 | 23 | 47 |
2020 Febrero | 4 | 0 | 4 |
2020 Enero | 4 | 0 | 4 |
2019 Diciembre | 8 | 5 | 13 |
2019 Noviembre | 4 | 1 | 5 |
2019 Septiembre | 8 | 2 | 10 |
2019 Agosto | 4 | 4 | 8 |
2019 Julio | 4 | 3 | 7 |
2019 Junio | 3 | 0 | 3 |
2019 Mayo | 6 | 21 | 27 |
2019 Abril | 2 | 1 | 3 |
2019 Marzo | 2 | 9 | 11 |
2019 Febrero | 4 | 0 | 4 |
2018 Diciembre | 4 | 0 | 4 |
2018 Noviembre | 1 | 0 | 1 |
2018 Octubre | 3 | 1 | 4 |
2018 Septiembre | 3 | 0 | 3 |
2018 Junio | 0 | 1 | 1 |
2018 Mayo | 0 | 7 | 7 |
2018 Marzo | 0 | 1 | 1 |
2018 Febrero | 36 | 3 | 39 |
2018 Enero | 62 | 9 | 71 |
2017 Diciembre | 59 | 10 | 69 |
2017 Noviembre | 38 | 13 | 51 |
2017 Octubre | 40 | 13 | 53 |
2017 Septiembre | 53 | 10 | 63 |
2017 Agosto | 95 | 25 | 120 |
2017 Julio | 64 | 4 | 68 |
2017 Junio | 68 | 37 | 105 |
2017 Mayo | 74 | 6 | 80 |
2017 Abril | 139 | 10 | 149 |
2017 Marzo | 92 | 28 | 120 |
2017 Febrero | 71 | 9 | 80 |
2017 Enero | 67 | 8 | 75 |
2016 Diciembre | 62 | 11 | 73 |
2016 Noviembre | 104 | 8 | 112 |
2016 Octubre | 92 | 18 | 110 |
2016 Septiembre | 100 | 11 | 111 |
2016 Agosto | 49 | 9 | 58 |
2016 Julio | 34 | 17 | 51 |
2016 Junio | 10 | 23 | 33 |
2016 Mayo | 5 | 7 | 12 |
2016 Abril | 4 | 6 | 10 |
2016 Marzo | 5 | 1 | 6 |
2016 Febrero | 2 | 10 | 12 |
2016 Enero | 12 | 13 | 25 |
2015 Diciembre | 15 | 2 | 17 |
2015 Noviembre | 11 | 17 | 28 |
2015 Octubre | 14 | 7 | 21 |
2015 Septiembre | 16 | 6 | 22 |
2015 Agosto | 19 | 7 | 26 |
2015 Julio | 120 | 15 | 135 |
2015 Junio | 76 | 18 | 94 |
2015 Mayo | 100 | 20 | 120 |
2015 Abril | 81 | 13 | 94 |
2015 Marzo | 124 | 15 | 139 |
2015 Febrero | 111 | 18 | 129 |
2015 Enero | 61 | 23 | 84 |
2014 Diciembre | 77 | 12 | 89 |
2014 Noviembre | 67 | 14 | 81 |
2014 Octubre | 101 | 22 | 123 |
2014 Septiembre | 69 | 25 | 94 |
2014 Agosto | 89 | 18 | 107 |
2014 Julio | 100 | 15 | 115 |
2014 Junio | 75 | 14 | 89 |
2014 Mayo | 84 | 20 | 104 |
2014 Abril | 77 | 6 | 83 |
2014 Marzo | 57 | 5 | 62 |
2014 Febrero | 37 | 8 | 45 |
2014 Enero | 55 | 7 | 62 |
2013 Diciembre | 41 | 10 | 51 |
2013 Noviembre | 45 | 5 | 50 |
2013 Octubre | 30 | 6 | 36 |
2013 Septiembre | 31 | 8 | 39 |
2013 Agosto | 43 | 13 | 56 |
2013 Julio | 14 | 24 | 38 |
2013 Junio | 11 | 8 | 19 |
2013 Mayo | 12 | 6 | 18 |
2013 Abril | 15 | 19 | 34 |
2013 Marzo | 17 | 8 | 25 |
2013 Febrero | 39 | 4 | 43 |
2013 Enero | 61 | 6 | 67 |
2012 Diciembre | 28 | 5 | 33 |
2012 Septiembre | 2 | 0 | 2 |