Case and Research Letters
Papular Epidermal Nevus With “Skyline” Basal Cell Layer: A Report of 3 New Cases
Papular epidermal nevus with «skyline»: 3 nuevos casos
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Pardo Domínguez, J. del Boz González, R. Fúnez Liébana" "autores" => array:3 [ 0 => array:4 [ "nombre" => "C." "apellidos" => "Pardo Domínguez" "email" => array:1 [ 0 => "c.pardo.d90@gmail.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" => "J." "apellidos" => "del Boz González" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "R." "apellidos" => "Fúnez Liébana" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] ] ] "afiliaciones" => array:3 [ 0 => array:3 [ "entidad" => "Área de Pediatría, Hospital Costa del Sol, Málaga, Marbella, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Área de Dermatología, Hospital Costa del Sol, Málaga, Marbella, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Área de Anatomía Patológica, Hospital Costa del Sol, Málaga, Marbella, Spain" "etiqueta" => "c" "identificador" => "aff0015" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Papular epidermal nevus with «skyline»: 3 nuevos casos" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 852 "Ancho" => 1500 "Tamanyo" => 158043 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A, Skin lesions in Case 1. B, Skin lesions in Case 2. C, Skin lesions in Case 3. D, Detail of skin lesion in Case 2.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">To the Editor:</p><p id="par0010" class="elsevierStylePara elsevierViewall">Papular epidermal nevus with “skyline” basal cell layer (PENS) is a variant of epidermal nevus that presents as isolated keratotic papules of variable number and morphology. Histology reveals the following characteristic features: hyperkeratosis; rectangular acanthosis; a palisade basal cell layer; and a wide supranuclear cytoplasmic region that resembles the characteristic “skyline sign” described in Bowen disease. The presence of a wide acellular region just above the nuclei of the basal cell layer is typical.</p><p id="par0015" class="elsevierStylePara elsevierViewall">In 2004 Tadini reported 2 cases of patients with papular epithelial hamartomas with associated neurological abnormalities. These cases were subsequently included in the <span class="elsevierStyleItalic">Atlas of Genodermatosis</span> as new neurocutaneous syndromes.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> However, it was not until 2011 that the acronym PENS was proposed by Torrelo et al. in their series of 5 patients.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">Eight further articles have since have been published, bringing the total number of reported PENS cases to 21, 10 of which involve associated neurological abnormalities. This association of PENS with extracutaneous abnormalities is known as PENS syndrome.</p><p id="par0025" class="elsevierStylePara elsevierViewall">We present 3 new cases of PENS, at least one of which corresponds to PENS syndrome.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Case Descriptions</span><p id="par0030" class="elsevierStylePara elsevierViewall">Case 1: The patient was a 1-year-old child, with no medical history of interest, who was seen for skin lesions on the limbs that had been present since birth and had remained stable. The lesions were flat, barely noticeable, minimally keratotic papules on the left leg and on both arms (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). Magnetic resonance imaging (MRI) revealed expansion of the hydrophilic cavity at the level of the C5–T1 vertebrae. The patient had left torticollis and neurodevelopmental delay predominantly affecting motor, cognitive, and language function (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0035" class="elsevierStylePara elsevierViewall">Case 2: The patient was a 1-year-old girl with a history of right renal aplasia. She was seen for skin lesions on one arm, the right side, and both legs that her parents had noticed at 1 month of age. The lesions were skin-colored papules without evident associated hyperkeratosis (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). Currently, the patient’s psychomotor development is normal.</p><p id="par0040" class="elsevierStylePara elsevierViewall">Case 3: A 9-year-old girl was referred to our department with multiple stable and asymptomatic skin lesions on the trunk and limbs that had been present since birth. The lesions were skin-colored or brownish keratotic papules, many of which were linear, located on the right armpit, right interscapular area, left retroauricular area, left arm and forearm, the back of the left thigh, right pretibial area, and the back of the shoulder (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). No other symptoms were reported, apart from tiptoeing in early childhood.</p><p id="par0045" class="elsevierStylePara elsevierViewall">In all 3 cases, a skin biopsy revealed the aforementioned findings characteristic of PENS.</p></span><span id="sec9005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect8005">Discussion</span><p id="par0055" class="elsevierStylePara elsevierViewall">We report 3 new cases of PENS, a recently described entity. Familiarity with PENS is important given the possible association with other anomalies, particularly those affecting neurological function.</p><p id="par0060" class="elsevierStylePara elsevierViewall">In the 3 cases presented here the patients had multiple, widely distributed skin lesions. In 2 of the patients the lesions had been present since birth, as reported in the majority of published cases. Although a likely familial association is described in 2 cases in the literature,<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,4</span></a> none of the relatives of our 3 patients presented similar lesions.</p><p id="par0065" class="elsevierStylePara elsevierViewall">The association of PENS with extracutaneous abnormalities is known as PENS syndrome, a term proposed at the 23rd meeting of the Spanish Pediatric Dermatology Group in 2012. Our first patient clearly fulfils the criteria for PENS syndrome. Psychomotor delay and mental retardation are features of most of the cases described,<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> including those presented here. However, given the young age of our 3 patients, we cannot rule out the potential appearance later in life of other associated neurological conditions such as epilepsy, dyslexia, or attention deficit hyperactivity disorder. An association between PENS and nephrourological malformations (e.g. renal aplasia, as observed in Case 2) had not been previously reported, and it is therefore unclear whether a causal relationship exists. Rodríguez-Díaz et al.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> previously reported an association between PENS and shortening of the Achilles tendon. Although difficult to establish with certainty, this anomaly could account for the tiptoeing in early childhood observed in Case 3.</p><p id="par0070" class="elsevierStylePara elsevierViewall">Because PENS is likely underdiagnosed, knowledge of its presentation and possible associated anomalies is important to ensure correct diagnosis.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Conflicts of Interest</span><p id="par0075" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:4 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Case Descriptions" ] 1 => array:2 [ "identificador" => "sec9005" "titulo" => "Discussion" ] 2 => array:2 [ "identificador" => "sec0010" "titulo" => "Conflicts of Interest" ] 3 => 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: Pardo Domínguez C, et al. Papular epidermal nevus with «skyline»: 3 nuevos casos. Actas Dermosifiliogr. 2020;111:185–187.</p>" ] ] "multimedia" => array:2 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 852 "Ancho" => 1500 "Tamanyo" => 158043 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A, Skin lesions in Case 1. B, Skin lesions in Case 2. C, Skin lesions in Case 3. D, Detail of skin lesion in Case 2.</p>" ] ] 1 => array:8 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 680 "Ancho" => 905 "Tamanyo" => 271009 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Skin biopsy with hyperkeratosis, acanthosis, and a palisade basal cell layer. A wide acellular area is focally evident just above the nuclei of the basal cell layer.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:6 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Papular epitelial hamartomas and neurologic abnormalities syndrome" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "G. Tadini" 1 => "R. Caputo" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "LibroEditado" => array:5 [ "editores" => "G.Tadini, R.Caputo" "titulo" => "Atlas of Genodermatoses" "paginaInicial" => "198" "paginaFinal" => "199" "serieFecha" => "2006" ] ] ] ] ] ] 1 => array:3 [ "identificador" => "bib0010" "etiqueta" => "2" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Papular epidermal nevus with ‘skyline’ basal cell layer (PENS)" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "A. Torrelo" 1 => "I. Colmenero" 2 => "L. Kristal" 3 => "L. Navarro" 4 => "C. Hafner" 5 => "A. 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Tadini" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1111/pde.12251" "Revista" => array:6 [ "tituloSerie" => "Pediatr Dermatol" "fecha" => "2014" "volumen" => "31" "paginaInicial" => "e33" "paginaFinal" => "e35" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/24274825" "web" => "Medline" ] ] ] ] ] ] ] ] 4 => array:3 [ "identificador" => "bib0025" "etiqueta" => "5" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Síndrome PENS. Comunicación XXIII Reunión del Grupo Español de Dermatología Pediátrica" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:6 [ 0 => "A. Nuño" 1 => "E. Rodríguez-Díaz" 2 => "P. Gonzalvo" 3 => "I. Colmenero" 4 => "A. Hernández-Martín" 5 => "A. Torrelo" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Libro" => array:1 [ "fecha" => "2012" ] ] ] ] ] ] 5 => array:3 [ "identificador" => "bib0030" "etiqueta" => "6" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Papular epidermal nevus with “skyline” basal cell layer (PENS) with extracutaneous findings" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:6 [ 0 => "E. Rodríguez-Díaz" 1 => "P. Gonzalvo" 2 => "I. Colmenero" 3 => "L. Requena" 4 => "A. Hernández-Martín" 5 => "A. Torrelo" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1111/j.1525-1470.2012.01830.x" "Revista" => array:6 [ "tituloSerie" => "Pediatr Dermatol" "fecha" => "2013" "volumen" => "30" "paginaInicial" => "e54" "paginaFinal" => "e56" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/22985090" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/15782190/0000011100000002/v1_202004140647/S1578219020300056/v1_202004140647/en/main.assets" "Apartado" => array:4 [ "identificador" => "6157" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Case and Research Letters" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/15782190/0000011100000002/v1_202004140647/S1578219020300056/v1_202004140647/en/main.pdf?idApp=UINPBA000044&text.app=https://actasdermo.org/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1578219020300056?idApp=UINPBA000044" ]
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| 2023 Octubre | 96 | 26 | 122 |
| 2023 Septiembre | 88 | 33 | 121 |
| 2023 Agosto | 86 | 17 | 103 |
| 2023 Julio | 66 | 41 | 107 |
| 2023 Junio | 73 | 28 | 101 |
| 2023 Mayo | 84 | 26 | 110 |
| 2023 Abril | 64 | 22 | 86 |
| 2023 Marzo | 81 | 34 | 115 |
| 2023 Febrero | 45 | 22 | 67 |
| 2023 Enero | 51 | 28 | 79 |
| 2022 Diciembre | 49 | 34 | 83 |
| 2022 Noviembre | 48 | 28 | 76 |
| 2022 Octubre | 40 | 18 | 58 |
| 2022 Septiembre | 61 | 28 | 89 |
| 2022 Agosto | 36 | 36 | 72 |
| 2022 Julio | 44 | 41 | 85 |
| 2022 Junio | 23 | 21 | 44 |
| 2022 Mayo | 73 | 51 | 124 |
| 2022 Abril | 81 | 33 | 114 |
| 2022 Marzo | 63 | 40 | 103 |
| 2022 Febrero | 69 | 27 | 96 |
| 2022 Enero | 82 | 30 | 112 |
| 2021 Diciembre | 71 | 35 | 106 |
| 2021 Noviembre | 79 | 38 | 117 |
| 2021 Octubre | 93 | 48 | 141 |
| 2021 Septiembre | 49 | 39 | 88 |
| 2021 Agosto | 44 | 22 | 66 |
| 2021 Julio | 27 | 19 | 46 |
| 2021 Junio | 41 | 14 | 55 |
| 2021 Mayo | 45 | 41 | 86 |
| 2021 Abril | 113 | 77 | 190 |
| 2021 Marzo | 92 | 18 | 110 |
| 2021 Febrero | 49 | 17 | 66 |
| 2021 Enero | 39 | 10 | 49 |
| 2020 Diciembre | 30 | 10 | 40 |
| 2020 Noviembre | 27 | 14 | 41 |
| 2020 Octubre | 27 | 4 | 31 |
| 2020 Septiembre | 35 | 15 | 50 |
| 2020 Agosto | 26 | 11 | 37 |
| 2020 Julio | 17 | 14 | 31 |
| 2020 Junio | 19 | 13 | 32 |
| 2020 Mayo | 29 | 16 | 45 |
| 2020 Abril | 52 | 20 | 72 |
| 2020 Marzo | 7 | 6 | 13 |
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