array:24 [
  "pii" => "S2341287923000509"
  "issn" => "23412879"
  "doi" => "10.1016/j.anpede.2023.02.010"
  "estado" => "S300"
  "fechaPublicacion" => "2023-04-01"
  "aid" => "3307"
  "copyright" => "Asociación Española de Pediatría"
  "copyrightAnyo" => "2022"
  "documento" => "article"
  "crossmark" => 1
  "subdocumento" => "sco"
  "cita" => "An Pediatr (Barc). 2023;98:323-4"
  "abierto" => array:3 [
    "ES" => true
    "ES2" => true
    "LATM" => true
  ]
  "gratuito" => true
  "lecturas" => array:1 [
    "total" => 0
  ]
  "Traduccion" => array:1 [
    "es" => array:19 [
      "pii" => "S1695403322001631"
      "issn" => "16954033"
      "doi" => "10.1016/j.anpedi.2022.06.008"
      "estado" => "S300"
      "fechaPublicacion" => "2023-04-01"
      "aid" => "3307"
      "copyright" => "Asociación Española de Pediatría"
      "documento" => "article"
      "crossmark" => 1
      "subdocumento" => "sco"
      "cita" => "An Pediatr (Barc). 2023;98:323-4"
      "abierto" => array:3 [
        "ES" => true
        "ES2" => true
        "LATM" => true
      ]
      "gratuito" => true
      "lecturas" => array:1 [
        "total" => 0
      ]
      "es" => array:11 [
        "idiomaDefecto" => true
        "cabecera" => "<span class="elsevierStyleTextfn">IM&#193;GENES EN PEDIATR&#205;A</span>"
        "titulo" => "S&#237;ndrome de distr&#233;s respiratorio por hipoplasia pulmonar y linfedema cong&#233;nito neonatal"
        "tienePdf" => "es"
        "tieneTextoCompleto" => "es"
        "paginas" => array:1 [
          0 => array:2 [
            "paginaInicial" => "323"
            "paginaFinal" => "324"
          ]
        ]
        "titulosAlternativos" => array:1 [
          "en" => array:1 [
            "titulo" => "Respiratory distress syndrome due to pulmonary hypoplasia and neonatal congenital lymphedema"
          ]
        ]
        "contieneTextoCompleto" => array:1 [
          "es" => true
        ]
        "contienePdf" => array:1 [
          "es" => true
        ]
        "resumenGrafico" => array:2 [
          "original" => 0
          "multimedia" => array:7 [
            "identificador" => "fig0005"
            "etiqueta" => "Figura 1"
            "tipo" => "MULTIMEDIAFIGURA"
            "mostrarFloat" => true
            "mostrarDisplay" => false
            "figura" => array:1 [
              0 => array:4 [
                "imagen" => "gr1.jpeg"
                "Alto" => 605
                "Ancho" => 805
                "Tamanyo" => 66913
              ]
            ]
            "descripcion" => array:1 [
              "es" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Edema duro&#44; fr&#237;o y sim&#233;trico en piernas y dorso de pies con engrosamiento d&#233;rmico y displasia ungueal &#40;flecha&#41;&#46;</p>"
            ]
          ]
        ]
        "autores" => array:1 [
          0 => array:2 [
            "autoresLista" => "Julio C&#233;sar Moreno Alfonso, Alberto P&#233;rez Mart&#237;nez, Ada Y&#46; Molina Caballero, Carlos Bardaj&#237; Pascual"
            "autores" => array:4 [
              0 => array:2 [
                "nombre" => "Julio C&#233;sar"
                "apellidos" => "Moreno Alfonso"
              ]
              1 => array:2 [
                "nombre" => "Alberto"
                "apellidos" => "P&#233;rez Mart&#237;nez"
              ]
              2 => array:2 [
                "nombre" => "Ada Y&#46;"
                "apellidos" => "Molina Caballero"
              ]
              3 => array:2 [
                "nombre" => "Carlos"
                "apellidos" => "Bardaj&#237; Pascual"
              ]
            ]
          ]
        ]
      ]
      "idiomaDefecto" => "es"
      "Traduccion" => array:1 [
        "en" => array:9 [
          "pii" => "S2341287923000509"
          "doi" => "10.1016/j.anpede.2023.02.010"
          "estado" => "S300"
          "subdocumento" => ""
          "abierto" => array:3 [
            "ES" => true
            "ES2" => true
            "LATM" => true
          ]
          "gratuito" => true
          "lecturas" => array:1 [
            "total" => 0
          ]
          "idiomaDefecto" => "en"
          "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2341287923000509?idApp=UINPBA00005H"
        ]
      ]
      "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1695403322001631?idApp=UINPBA00005H"
      "url" => "/16954033/0000009800000004/v2_202304070135/S1695403322001631/v2_202304070135/es/main.assets"
    ]
  ]
  "itemSiguiente" => array:19 [
    "pii" => "S2341287923000686"
    "issn" => "23412879"
    "doi" => "10.1016/j.anpede.2023.02.018"
    "estado" => "S300"
    "fechaPublicacion" => "2023-04-01"
    "aid" => "3413"
    "copyright" => "Asociaci&#243;n Espa&#241;ola de Pediatr&#237;a"
    "documento" => "simple-article"
    "crossmark" => 1
    "subdocumento" => "cor"
    "cita" => "An Pediatr &#40;Barc&#41;. 2023;98:325-6"
    "abierto" => array:3 [
      "ES" => true
      "ES2" => true
      "LATM" => true
    ]
    "gratuito" => true
    "lecturas" => array:1 [
      "total" => 0
    ]
    "en" => array:10 [
      "idiomaDefecto" => true
      "cabecera" => "<span class="elsevierStyleTextfn">Letter to the Editor</span>"
      "titulo" => "Letter to the Editor of <span class="elsevierStyleItalic">Anales de Pediatr&#237;a &#40;Barc&#41;</span> about the work of Salinas-Salvador et al&#46;&#58; &#8220;Retrospective study on the effectiveness and safety of the shortened 5&#8211;7-day antibiotic regimen for acute streptococcal pharyngotonsillitis compared to the classic 10-day regimen&#8221;&#46; Authors reply"
      "tienePdf" => "en"
      "tieneTextoCompleto" => "en"
      "paginas" => array:1 [
        0 => array:2 [
          "paginaInicial" => "325"
          "paginaFinal" => "326"
        ]
      ]
      "titulosAlternativos" => array:1 [
        "es" => array:1 [
          "titulo" => "Carta a la editora de <span class="elsevierStyleSmallCaps">Anales de Pediatr&#237;a &#40;Barc&#41;</span> sobre el trabajo de Salinas-Salvador y colaboradores&#58; &#171;Estudio retrospectivo sobre la efectividad y seguridad de la pauta antibi&#243;tica reducida a 5-7 d&#237;as en la faringoamigdalitis aguda estreptoc&#243;cica comparada con la pauta cl&#225;sica de 10 d&#237;as&#187;&#46; Respuesta de los autores"
        ]
      ]
      "contieneTextoCompleto" => array:1 [
        "en" => true
      ]
      "contienePdf" => array:1 [
        "en" => true
      ]
      "autores" => array:1 [
        0 => array:2 [
          "autoresLista" => "Bel&#233;n Salinas Salvador, C&#233;sar Garc&#237;a Vera"
          "autores" => array:2 [
            0 => array:2 [
              "nombre" => "Bel&#233;n"
              "apellidos" => "Salinas Salvador"
            ]
            1 => array:2 [
              "nombre" => "C&#233;sar"
              "apellidos" => "Garc&#237;a Vera"
            ]
          ]
        ]
      ]
    ]
    "idiomaDefecto" => "en"
    "Traduccion" => array:1 [
      "es" => array:9 [
        "pii" => "S1695403323000401"
        "doi" => "10.1016/j.anpedi.2023.02.004"
        "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/S1695403323000401?idApp=UINPBA00005H"
      ]
    ]
    "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2341287923000686?idApp=UINPBA00005H"
    "url" => "/23412879/0000009800000004/v2_202304070152/S2341287923000686/v2_202304070152/en/main.assets"
  ]
  "itemAnterior" => array:19 [
    "pii" => "S2341287923000558"
    "issn" => "23412879"
    "doi" => "10.1016/j.anpede.2023.02.013"
    "estado" => "S300"
    "fechaPublicacion" => "2023-04-01"
    "aid" => "3267"
    "copyright" => "Asociaci&#243;n Espa&#241;ola de Pediatr&#237;a"
    "documento" => "article"
    "crossmark" => 1
    "subdocumento" => "sco"
    "cita" => "An Pediatr &#40;Barc&#41;. 2023;98:321-2"
    "abierto" => array:3 [
      "ES" => true
      "ES2" => true
      "LATM" => true
    ]
    "gratuito" => true
    "lecturas" => array:1 [
      "total" => 0
    ]
    "en" => array:11 [
      "idiomaDefecto" => true
      "cabecera" => "<span class="elsevierStyleTextfn">Images in Paediatrics</span>"
      "titulo" => "Capillary malformation of the lower lip&#58; CLAPO syndrome"
      "tienePdf" => "en"
      "tieneTextoCompleto" => "en"
      "paginas" => array:1 [
        0 => array:2 [
          "paginaInicial" => "321"
          "paginaFinal" => "322"
        ]
      ]
      "titulosAlternativos" => array:1 [
        "es" => array:1 [
          "titulo" => "Malformaci&#243;n capilar del labio inferior&#58; s&#237;ndrome CLAPO"
        ]
      ]
      "contieneTextoCompleto" => array:1 [
        "en" => true
      ]
      "contienePdf" => array:1 [
        "en" => true
      ]
      "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" => 2173
              "Ancho" => 2487
              "Tamanyo" => 323821
            ]
          ]
          "detalles" => array:1 [
            0 => array:3 [
              "identificador" => "at0005"
              "detalle" => "Figure "
              "rol" => "short"
            ]
          ]
          "descripcion" => array:1 [
            "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A&#41; Capillary malformation between the brows and in the lower lip &#40;arrow&#41; with right facial asymmetry &#40;arrowhead&#41;&#46; Overgrowth of the first toe of the feet &#40;arrowhead&#41; with bilateral sandal gap &#40;double-headed arrow&#41;&#58; B&#41; right foot&#44; C&#41; left foot&#46; Phenotype compatible with CLAPO syndrome&#46;</p>"
          ]
        ]
      ]
      "autores" => array:1 [
        0 => array:2 [
          "autoresLista" => "Julio C&#233;sar Moreno Alfonso, Juan Carlos L&#243;pez Guti&#233;rrez, Paloma Elena Triana Junco, Mar&#237;a San Basilio Berenguer"
          "autores" => array:4 [
            0 => array:2 [
              "nombre" => "Julio C&#233;sar"
              "apellidos" => "Moreno Alfonso"
            ]
            1 => array:2 [
              "nombre" => "Juan Carlos"
              "apellidos" => "L&#243;pez Guti&#233;rrez"
            ]
            2 => array:2 [
              "nombre" => "Paloma Elena"
              "apellidos" => "Triana Junco"
            ]
            3 => array:2 [
              "nombre" => "Mar&#237;a"
              "apellidos" => "San Basilio Berenguer"
            ]
          ]
        ]
      ]
    ]
    "idiomaDefecto" => "en"
    "Traduccion" => array:1 [
      "es" => array:9 [
        "pii" => "S1695403322000236"
        "doi" => "10.1016/j.anpedi.2022.03.005"
        "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/S1695403322000236?idApp=UINPBA00005H"
      ]
    ]
    "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2341287923000558?idApp=UINPBA00005H"
    "url" => "/23412879/0000009800000004/v2_202304070152/S2341287923000558/v2_202304070152/en/main.assets"
  ]
  "en" => array:16 [
    "idiomaDefecto" => true
    "cabecera" => "<span class="elsevierStyleTextfn">Images in Paediatrics</span>"
    "titulo" => "Respiratory distress syndrome due to pulmonary hypoplasia and neonatal congenital lymphedema"
    "tieneTextoCompleto" => true
    "paginas" => array:1 [
      0 => array:2 [
        "paginaInicial" => "323"
        "paginaFinal" => "324"
      ]
    ]
    "autores" => array:1 [
      0 => array:4 [
        "autoresLista" => "Julio C&#233;sar Moreno Alfonso, Alberto P&#233;rez Mart&#237;nez, Ada Y&#46; Molina Caballero, Carlos Bardaj&#237; Pascual"
        "autores" => array:4 [
          0 => array:4 [
            "nombre" => "Julio C&#233;sar"
            "apellidos" => "Moreno Alfonso"
            "email" => array:1 [
              0 => "juliomoreno.md@gmail.com"
            ]
            "referencia" => array:1 [
              0 => array:2 [
                "etiqueta" => "<span class="elsevierStyleSup">&#42;</span>"
                "identificador" => "cor0005"
              ]
            ]
          ]
          1 => array:3 [
            "nombre" => "Alberto"
            "apellidos" => "P&#233;rez Mart&#237;nez"
            "email" => array:1 [
              0 => "alperezma@gmail.com"
            ]
          ]
          2 => array:3 [
            "nombre" => "Ada Y&#46;"
            "apellidos" => "Molina Caballero"
            "email" => array:1 [
              0 => "adyemoca@yahoo.com"
            ]
          ]
          3 => array:3 [
            "nombre" => "Carlos"
            "apellidos" => "Bardaj&#237; Pascual"
            "email" => array:1 [
              0 => "carlos.bardaji.pascual@navarra.es"
            ]
          ]
        ]
        "afiliaciones" => array:1 [
          0 => array:2 [
            "entidad" => "Servicio de Cirug&#237;a Pedi&#225;trica&#44; Hospital Universitario de Navarra&#44; Pamplona&#44; Spain"
            "identificador" => "aff0005"
          ]
        ]
        "correspondencia" => array:1 [
          0 => array:3 [
            "identificador" => "cor0005"
            "etiqueta" => "&#8270;"
            "correspondencia" => "Corresponding author&#46;"
          ]
        ]
      ]
    ]
    "titulosAlternativos" => array:1 [
      "es" => array:1 [
        "titulo" => "S&#237;ndrome de distr&#233;s respiratorio por hipoplasia pulmonar y linfedema cong&#233;nito neonatal"
      ]
    ]
    "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" => 1009
            "Ancho" => 1342
            "Tamanyo" => 143732
          ]
        ]
        "detalles" => array:1 [
          0 => array:3 [
            "identificador" => "at0005"
            "detalle" => "Figure "
            "rol" => "short"
          ]
        ]
        "descripcion" => array:1 [
          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Hard&#44; cold and symmetrical oedema in legs and dorsum of feet with thickening of the dermis and ungual dysplasia &#40;arrow&#41;&#46;</p>"
        ]
      ]
    ]
    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">We present the case of a male newborn aged 20&#8239;h in whom prenatal examinations had been normal and with a history of congenital oedema that had not been evaluated in first-degree relatives&#46; At birth&#44; he had respiratory distress that required non-invasive ventilation and swelling of the lower extremities&#46; The physical examination revealed hard&#44; cold oedema in both legs with thickening of the skin and ungual dysplasia &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#44; in addition to hypoventilation in the base of the right lung&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">A chest radiograph and computed tomography scan evinced right pulmonary hypoplasia &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>A&#41;&#44; and a lymphoscintigraphy was requested due to suspicion of congenital lymphoedema&#44; which confirmed the absence of lymphatic vessels in the lower extremities &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>B&#41;&#46; The patient received a clinical diagnosis of type I congenital lymphoedema type I&#44; or Milroy disease&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">At present&#44; at age 7 months&#44; the patient is free of respiratory symptoms and in treatment with lower extremity compression and physical therapy&#44; with a favourable response &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46; Genetic testing is underway&#46;</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Milroy disease is a rare condition with an estimated incidence of 1 case per 6000 live births with a 1&#58;2&#46;3 male-to-female ratio&#46; It is caused by a variant in the <span class="elsevierStyleItalic">VEGFR3</span> gene encoding the vascular endothelial growth factor receptor 3&#44; resulting in aplasia or dysplasia of lymphatic vessels&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> It manifests as congenital progressive oedema in the lower extremities&#44; in isolation or as part of a syndrome&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Although it is infrequent and it is still unclear what it is associated with&#44; the presence of congenital lymphoedema must prompt an investigation of the family history and a lymphatic and genetic evaluation&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Appropriate multidisciplinary treatment may prevent progression to functional limitations&#44; complications and sequelae&#46; In selected cases refractory to conservative treatment&#44; surgical treatment with vascularized lymph node transfer is a possible alternative that has achieved good results in recent case series&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Funding</span><p id="par0025" class="elsevierStylePara elsevierViewall">This research did not receive any external funding&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Conflicts of interest</span><p id="par0030" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare&#46;</p></span></span>"
    "textoCompletoSecciones" => array:1 [
      "secciones" => array:3 [
        0 => array:2 [
          "identificador" => "sec0005"
          "titulo" => "Funding"
        ]
        1 => array:2 [
          "identificador" => "sec0010"
          "titulo" => "Conflicts of interest"
        ]
        2 => array:1 [
          "titulo" => "References"
        ]
      ]
    ]
    "pdfFichero" => "main.pdf"
    "tienePdf" => true
    "fechaRecibido" => "2022-03-28"
    "fechaAceptado" => "2022-06-09"
    "multimedia" => array:3 [
      0 => array:8 [
        "identificador" => "fig0005"
        "etiqueta" => "Figure 1"
        "tipo" => "MULTIMEDIAFIGURA"
        "mostrarFloat" => true
        "mostrarDisplay" => false
        "figura" => array:1 [
          0 => array:4 [
            "imagen" => "gr1.jpeg"
            "Alto" => 1009
            "Ancho" => 1342
            "Tamanyo" => 143732
          ]
        ]
        "detalles" => array:1 [
          0 => array:3 [
            "identificador" => "at0005"
            "detalle" => "Figure "
            "rol" => "short"
          ]
        ]
        "descripcion" => array:1 [
          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Hard&#44; cold and symmetrical oedema in legs and dorsum of feet with thickening of the dermis and ungual dysplasia &#40;arrow&#41;&#46;</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" => 1570
            "Ancho" => 2609
            "Tamanyo" => 229966
          ]
        ]
        "detalles" => array:1 [
          0 => array:3 [
            "identificador" => "at0010"
            "detalle" => "Figure "
            "rol" => "short"
          ]
        ]
        "descripcion" => array:1 [
          "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">&#40;A&#41; Computed tomography evincing bilateral partial pulmonary hypoplasia&#44; more marked on the right side &#40;arrow&#41;&#46; &#40;B&#41; Lymphoscintigraphy with visualization of axillary lymph nodes but not of popliteal and inguinal nodes&#44; compatible with congenital lymphatic aplasia&#46;</p>"
        ]
      ]
      2 => array:8 [
        "identificador" => "fig0015"
        "etiqueta" => "Figure 3"
        "tipo" => "MULTIMEDIAFIGURA"
        "mostrarFloat" => true
        "mostrarDisplay" => false
        "figura" => array:1 [
          0 => array:4 [
            "imagen" => "gr3.jpeg"
            "Alto" => 2363
            "Ancho" => 1287
            "Tamanyo" => 155610
          ]
        ]
        "detalles" => array:1 [
          0 => array:3 [
            "identificador" => "at0015"
            "detalle" => "Figure "
            "rol" => "short"
          ]
        ]
        "descripcion" => array:1 [
          "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Progressive reduction of lymphoedema after 6 months of compression and physical therapy&#46;</p>"
        ]
      ]
    ]
    "bibliografia" => array:2 [
      "titulo" => "References"
      "seccion" => array:1 [
        0 => array:2 [
          "identificador" => "bibs0005"
          "bibliografiaReferencia" => array:4 [
            0 => array:3 [
              "identificador" => "bib0005"
              "etiqueta" => "1"
              "referencia" => array:1 [
                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Linfedema cong&#233;nito secundario a enfermedad de Milroy"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => false
                          "autores" => array:4 [
                            0 => "E&#46; Urbaneja"
                            1 => "R&#46; Garrote"
                            2 => "M&#46;A&#46; Pino"
                            3 => "H&#46; Gonz&#225;lez"
                          ]
                        ]
                      ]
                    ]
                  ]
                  "host" => array:1 [
                    0 => array:2 [
                      "doi" => "10.1016/j.anpedi.2015.12.007"
                      "Revista" => array:8 [
                        "tituloSerie" => "An Pediatr &#40;Barc&#41;&#46;"
                        "fecha" => "2017"
                        "volumen" => "86"
                        "numero" => "3"
                        "paginaInicial" => "169"
                        "paginaFinal" => "170"
                        "link" => array:1 [
                          0 => array:2 [
                            "url" => "https://www.ncbi.nlm.nih.gov/pubmed/26818397"
                            "web" => "Medline"
                          ]
                        ]
                        "itemHostRev" => array:3 [
                          "pii" => "S0953620521001503"
                          "estado" => "S300"
                          "issn" => "09536205"
                        ]
                      ]
                    ]
                  ]
                ]
              ]
            ]
            1 => array:3 [
              "identificador" => "bib0010"
              "etiqueta" => "2"
              "referencia" => array:1 [
                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Clinical aspects of congenital primary lymphedema"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => false
                          "autores" => array:4 [
                            0 => "A&#46;C&#46; De Godoy"
                            1 => "L&#46;M&#46; De Godoy"
                            2 => "J&#46;M&#46; De Godoy"
                            3 => "M&#46; De Fatima"
                          ]
                        ]
                      ]
                    ]
                  ]
                  "host" => array:1 [
                    0 => array:2 [
                      "doi" => "10.3233/PRM-190642"
                      "Revista" => array:7 [
                        "tituloSerie" => "J Pediatr Rehabil Med&#46;"
                        "fecha" => "2021"
                        "volumen" => "14"
                        "numero" => "1"
                        "paginaInicial" => "51"
                        "paginaFinal" => "53"
                        "link" => array:1 [
                          0 => array:2 [
                            "url" => "https://www.ncbi.nlm.nih.gov/pubmed/32986626"
                            "web" => "Medline"
                          ]
                        ]
                      ]
                    ]
                  ]
                ]
              ]
            ]
            2 => array:3 [
              "identificador" => "bib0015"
              "etiqueta" => "3"
              "referencia" => array:1 [
                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Enfermedad de Milroy o linfedema primario cong&#233;nito asociado a carcinoma espinocelular invasor"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => false
                          "autores" => array:4 [
                            0 => "O&#46; Cheirif"
                            1 => "L&#46; Ram&#237;rez"
                            2 => "S&#46; Toussaint"
                            3 => "L&#46; Lammoglia"
                          ]
                        ]
                      ]
                    ]
                  ]
                  "host" => array:1 [
                    0 => array:2 [
                      "doi" => "10.1016/j.ad.2016.05.011"
                      "Revista" => array:7 [
                        "tituloSerie" => "Actas Dermosifiliogr&#46;"
                        "fecha" => "2016"
                        "volumen" => "107"
                        "numero" => "10"
                        "paginaInicial" => "864"
                        "paginaFinal" => "866"
                        "link" => array:1 [
                          0 => array:2 [
                            "url" => "https://www.ncbi.nlm.nih.gov/pubmed/27374380"
                            "web" => "Medline"
                          ]
                        ]
                      ]
                    ]
                  ]
                ]
              ]
            ]
            3 => array:3 [
              "identificador" => "bib0020"
              "etiqueta" => "4"
              "referencia" => array:1 [
                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Surgical treatment of Milroy disease"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => true
                          "autores" => array:6 [
                            0 => "A&#46; Bolletta"
                            1 => "G&#46; Di Taranto"
                            2 => "S&#46;H&#46; Chen"
                            3 => "R&#46; Elia"
                            4 => "V&#46; Amorosi"
                            5 => "J&#46;C&#46; Chan"
                          ]
                        ]
                      ]
                    ]
                  ]
                  "host" => array:1 [
                    0 => array:2 [
                      "doi" => "10.1002/jso.25583"
                      "Revista" => array:7 [
                        "tituloSerie" => "J Surg Oncol&#46;"
                        "fecha" => "2020"
                        "volumen" => "121"
                        "numero" => "1"
                        "paginaInicial" => "175"
                        "paginaFinal" => "181"
                        "link" => array:1 [
                          0 => array:2 [
                            "url" => "https://www.ncbi.nlm.nih.gov/pubmed/31165487"
                            "web" => "Medline"
                          ]
                        ]
                      ]
                    ]
                  ]
                ]
              ]
            ]
          ]
        ]
      ]
    ]
  ]
  "idiomaDefecto" => "en"
  "url" => "/23412879/0000009800000004/v2_202304070152/S2341287923000509/v2_202304070152/en/main.assets"
  "Apartado" => array:4 [
    "identificador" => "77701"
    "tipo" => "SECCION"
    "en" => array:2 [
      "titulo" => "Images in Paediatrics"
      "idiomaDefecto" => true
    ]
    "idiomaDefecto" => "en"
  ]
  "PDF" => "https://static.elsevier.es/multimedia/23412879/0000009800000004/v2_202304070152/S2341287923000509/v2_202304070152/en/main.pdf?idApp=UINPBA00005H&text.app=https://www.analesdepediatria.org/"
  "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2341287923000509?idApp=UINPBA00005H"
]
Share
Journal Information
Vol. 98. Issue 4.
Pages 323-324 (1 April 2023)
Vol. 98. Issue 4.
Pages 323-324 (1 April 2023)
Images in Paediatrics
Full text access
Respiratory distress syndrome due to pulmonary hypoplasia and neonatal congenital lymphedema
Síndrome de distrés respiratorio por hipoplasia pulmonar y linfedema congénito neonatal
Visits
1635
Julio César Moreno Alfonso
Corresponding author
juliomoreno.md@gmail.com

Corresponding author.
, Alberto Pérez Martínez, Ada Y. Molina Caballero, Carlos Bardají Pascual
Servicio de Cirugía Pediátrica, Hospital Universitario de Navarra, Pamplona, Spain
This item has received
Article information
Full Text
Bibliography
Download PDF
Statistics
Figures (3)
Show moreShow less
Full Text

We present the case of a male newborn aged 20 h in whom prenatal examinations had been normal and with a history of congenital oedema that had not been evaluated in first-degree relatives. At birth, he had respiratory distress that required non-invasive ventilation and swelling of the lower extremities. The physical examination revealed hard, cold oedema in both legs with thickening of the skin and ungual dysplasia (Fig. 1), in addition to hypoventilation in the base of the right lung.

Figure 1.

Hard, cold and symmetrical oedema in legs and dorsum of feet with thickening of the dermis and ungual dysplasia (arrow).

(0.14MB).

A chest radiograph and computed tomography scan evinced right pulmonary hypoplasia (Fig. 2A), and a lymphoscintigraphy was requested due to suspicion of congenital lymphoedema, which confirmed the absence of lymphatic vessels in the lower extremities (Fig. 2B). The patient received a clinical diagnosis of type I congenital lymphoedema type I, or Milroy disease.

Figure 2.

(A) Computed tomography evincing bilateral partial pulmonary hypoplasia, more marked on the right side (arrow). (B) Lymphoscintigraphy with visualization of axillary lymph nodes but not of popliteal and inguinal nodes, compatible with congenital lymphatic aplasia.

(0.22MB).

At present, at age 7 months, the patient is free of respiratory symptoms and in treatment with lower extremity compression and physical therapy, with a favourable response (Fig. 3). Genetic testing is underway.

Figure 3.

Progressive reduction of lymphoedema after 6 months of compression and physical therapy.

(0.15MB).

Milroy disease is a rare condition with an estimated incidence of 1 case per 6000 live births with a 1:2.3 male-to-female ratio. It is caused by a variant in the VEGFR3 gene encoding the vascular endothelial growth factor receptor 3, resulting in aplasia or dysplasia of lymphatic vessels.1 It manifests as congenital progressive oedema in the lower extremities, in isolation or as part of a syndrome.2 Although it is infrequent and it is still unclear what it is associated with, the presence of congenital lymphoedema must prompt an investigation of the family history and a lymphatic and genetic evaluation.3 Appropriate multidisciplinary treatment may prevent progression to functional limitations, complications and sequelae. In selected cases refractory to conservative treatment, surgical treatment with vascularized lymph node transfer is a possible alternative that has achieved good results in recent case series.4

Funding

This research did not receive any external funding.

Conflicts of interest

The authors have no conflicts of interest to declare.

References
[1]
E. Urbaneja, R. Garrote, M.A. Pino, H. González.
Linfedema congénito secundario a enfermedad de Milroy.
An Pediatr (Barc)., 86 (2017), pp. 169-170
[2]
A.C. De Godoy, L.M. De Godoy, J.M. De Godoy, M. De Fatima.
Clinical aspects of congenital primary lymphedema.
J Pediatr Rehabil Med., 14 (2021), pp. 51-53
[3]
O. Cheirif, L. Ramírez, S. Toussaint, L. Lammoglia.
Enfermedad de Milroy o linfedema primario congénito asociado a carcinoma espinocelular invasor.
Actas Dermosifiliogr., 107 (2016), pp. 864-866
[4]
A. Bolletta, G. Di Taranto, S.H. Chen, R. Elia, V. Amorosi, J.C. Chan, et al.
Surgical treatment of Milroy disease.
J Surg Oncol., 121 (2020), pp. 175-181
Copyright © 2022. Asociación Española de Pediatría
Download PDF
Idiomas
Anales de Pediatría (English Edition)
Article options
Tools
es en

¿Es usted profesional sanitario apto para prescribir o dispensar medicamentos?

Are you a health professional able to prescribe or dispense drugs?