Case for Diagnosis
Linear Hyperkeratotic Papules in a Full Term Newborn
Pápulas hiperqueratóticas con disposición lineal en un recién nacido a término
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Díez de los Ríos Quintanero, S. del Pozo Arribas, C. Martínez-Mera, L. Nájera" "autores" => array:4 [ 0 => array:4 [ "nombre" => "B." "apellidos" => "Díez de los Ríos Quintanero" "email" => array:1 [ 0 => "blancadiezdelosrios@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" => "S." "apellidos" => "del Pozo Arribas" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "C." "apellidos" => "Martínez-Mera" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "L." "apellidos" => "Nájera" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] ] ] "afiliaciones" => array:3 [ 0 => array:3 [ "entidad" => "Neonatal Unit, Pediatrics Department, University Hospital Puerta de Hierro-Majadahonda, Madrid, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Dermatology Department, University Hospital Puerta de Hierro-Majadahonda, Madrid, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Pathology Department, University Hospital Puerta de Hierro-Majadahonda, Madrid, Spain" "etiqueta" => "c" "identificador" => "aff0015" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Pápulas hiperqueratóticas con disposición lineal en un recién nacido a término" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 568 "Ancho" => 755 "Tamanyo" => 211247 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Acanthosis, focal hyperparakeratosis and acrosyringia (H and E, ×20).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">We present a male newborn, was the second of no-consanguineous healthy parents, born from a full-term spontaneous delivery. At birth, he presented cutaneous lesions in a Blaschko-linear distribution. The affection was extended in the right half of the body, spreading into the trunk, extremities and genitalia characterized by withish keratotic tiny papules, and in soles and palms were forming hyperkeratotic plaques. The mucose were normal (shown in <a class="elsevierStyleCrossRefs" href="#fig0005">Figs. 1 and 2</a>).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">A complete physical examination revealed no other abnormalities. Skin biopsy showed acanthosis, focal hyperparakeratosis filling follicular infundibula and acrosyringia, and thin columns of parakeratosis emerging from the intraepidermal segments of the anexa (shown in <a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). The lesions spontaneously improved in the first month, remaining just the sole, palm and arm. A transfontanellar ultrasound was performed, without abnormalities. The patient continued with normal development and is followed by the dermatology and paediatrician department.</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">What is your diagnosis?</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Diagnosis</span><p id="par0020" class="elsevierStylePara elsevierViewall">Porokeratotic adnexal ostial nevus (PAON).</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Comments</span><p id="par0025" class="elsevierStylePara elsevierViewall">The diagnosis of this newborn was a Porokeratotic adnexal ostial nevus (PAON). Initially the physical examination with the characteristic distribution of the lesions following Blaschko lines gave us the diagnostic suspicion of this disease. However until the results of the biopsy we did not have the definitive diagnosis.</p><p id="par0030" class="elsevierStylePara elsevierViewall">PAON is a rare benign hamartomatous malformation, usually with a Blaschkoid pattern.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> It is typically congenital, but it is not always presented in the neonatal period and there are some cases of late-onset PAON. Clinically, PAON present two different types of lesions: palmoplantar papules that present keratotic plugs with central pits, and keratotic papules with a linear distribution in the rest of the body. It is mostly unilateral but there are some cases described of bilateral affection<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">2</span></a>. The exact pathogenesis is unclear but may be produced by a mutation in GJB2 gene, which encoded the connexin 26, a gap junctional protein that participates in keratinocyte growth and differentiation. This mutation is associated with Keratitis-ichthyosis-deafness syndrome, considering PAON a mosaic form of this syndrome. Its inheritance is autosomal dominant although it could express as a de novo mutation. In some patients coexist problems like seizures, hemiparesis, hyperthyroidism, polyneuropathy, scoliosis, deafness or development delay.<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">2–4</span></a> However, the majority of PAON cases present as solitary lesions.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a> The differential diagnosis includes linear porokeratosis, inflammatory linear epidermal nevus, incontinentia pigmenti, Darier disease, hypomelanosis of Ito and linear lichen striatus among others.</p><p id="par0035" class="elsevierStylePara elsevierViewall">We have to consider this entity in the Blaschkoid pattern of neonatal lesions and make the histopathology study to achieve the diagnosis.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">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:4 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Diagnosis" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Comments" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Conflicts of interest" ] 3 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "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" => 568 "Ancho" => 755 "Tamanyo" => 69455 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Linear micropapules following the Blaschko lines.</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" => 568 "Ancho" => 755 "Tamanyo" => 70992 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Hyperkeratotic plaques in right sole.</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" => 568 "Ancho" => 755 "Tamanyo" => 211247 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Acanthosis, focal hyperparakeratosis and acrosyringia (H and E, ×20).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0030" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Porokeratotic eccrine ostial and dermal duct naevus with dermatomal trunk involvement: literature review and report on the efficacy of laser treatment" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "C.S. Leung" 1 => "W.Y.M. Tang" 2 => "W.Y. Lam" 3 => "W.K.K. Fung" 4 => "K.K. Lo" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1046/j.1365-2133.1998.02186.x" "Revista" => array:6 [ "tituloSerie" => "Br J Dermatol" "fecha" => "1998" "volumen" => "138" "paginaInicial" => "684" "paginaFinal" => "688" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/9640380" "web" => "Medline" ] ] ] ] ] ] ] ] 1 => array:3 [ "identificador" => "bib0035" "etiqueta" => "2" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Porokeratotic adnexal ostial naevus: review on the entity and therapeutic approach" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "M. Llamas-Velasco" 1 => "N. Hilty" 2 => "W. 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Sugarman" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:5 [ "tituloSerie" => "Pediatric Dermatol" "fecha" => "2018" "volumen" => "35" "paginaInicial" => "21" "paginaFinal" => "29" ] ] ] ] ] ] 4 => array:3 [ "identificador" => "bib0050" "etiqueta" => "5" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Multiple proliferating pilar tumors with porokeratotic adnexal ostial nevus: a rare association" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "J. Maghfour" 1 => "J. Kane" 2 => "C. DiMarco" 3 => "L. Robinson-Bostom" 4 => "S. Chai" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.jdcr.2019.12.019" "Revista" => array:6 [ "tituloSerie" => "JAAD Case Rep" "fecha" => "2020" "volumen" => "6" "paginaInicial" => "344" "paginaFinal" => "347" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/32258317" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/00017310/0000011500000005/v1_202405030647/S000173102300073X/v1_202405030647/en/main.assets" "Apartado" => array:4 [ "identificador" => "6161" "tipo" => "SECCION" "es" => array:2 [ "titulo" => "Casos para el diagnóstico" "idiomaDefecto" => true ] "idiomaDefecto" => "es" ] "PDF" => "https://static.elsevier.es/multimedia/00017310/0000011500000005/v1_202405030647/S000173102300073X/v1_202405030647/en/main.pdf?idApp=UINPBA000044&text.app=https://actasdermo.org/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S000173102300073X?idApp=UINPBA000044" ]
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