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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Congenital lung malformations &#40;CLMs&#41; constitute a heterogeneous group of lung anomalies involving the airways&#44; parenchyma and blood vessels&#46; Historically&#44; they have been considered infrequent&#44; as their annual incidence has been documented to be of 1 case per 10&#44;000&#8211;35&#44;000 pregnancies<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a> or about 30&#8211;42 cases per 100&#44;000 inhabitants in the general population&#46; There are also authors that assert that their diagnosis has improved considerably with the introduction of routine prenatal ultrasound examinations&#44;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> but few studies have analysed how their incidence has actually been changing in recent times&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">In order to establish the incidence of CLMs in the autonomous community of Cantabria &#40;Spain&#41;&#44; we conducted a retrospective descriptive study of all cases of CLM managed in the Department of Paediatrics of the Hospital Universitario Marqu&#233;s de Valdecilla in Santander&#44; &#40;Cantabria&#44; Spain&#41;&#44; specifically in the Units of Paediatric Pulmonology and Paediatric Surgery&#44; in the period between January 2007 and December 2017&#46; Our hospital is the tertiary care hospital that manages all complicated pregnancies in Cantabria&#44; and we identified the patients using information obtained from the Department of Admissions and Clinical Records&#46; In addition&#44; we obtained data on the number of births during this period from the Instituto C&#225;ntabro de Estad&#237;stica &#40;Statistical Institute of Cantabria&#41; and the Instituto Nacional de Estad&#237;stica &#40;National Institute of Statistics&#41;&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">A total of 16 cases of CLM were diagnosed during the period under study&#44; corresponding to 9 female patients and 7 male patients&#46; The diagnosis was prenatal in 15 cases &#40;93&#46;7&#37;&#41;&#44; with the defect identified in the ultrasound examination performed at 22 weeks&#8217; gestation&#46; All of these patients were evaluated with a chest radiograph and a chest ultrasound examination within 24<span class="elsevierStyleHsp" style=""></span>h from birth&#44; and these imaging tests confirmed the presence of a CLM in the patients whose second trimester ultrasound examination had been positive&#46; In the remaining patient&#44; treated in 2007&#44; the CLM was diagnosed at age 4 months by means of a chest radiograph and a computed tomography angiogram&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall"><a class="elsevierStyleCrossRef" href="#tbl0005">Table 1</a> presents the data on the annual incidence of CLM&#46; The mean annual incidence was of 2&#46;05 cases per 10&#44;000 births &#40;standard deviation&#44; 3&#46;26&#41; and the median was 1&#46;98 cases &#40;interquartile range&#44; 6&#46;79&#41;&#44; calculated with data through December 2016&#46; We could not calculate the incidence for 2017 because at the time of this writing there is no published data on the number of births in Cantabria during this year&#46; We ought to highlight the considerable variability between years&#44; as 2 cases were diagnosed in a total of 31&#44;137 children born between 2007 and 2012&#44; compared to 12 cases diagnosed in a total of 17&#44;710 children born between 2013 and 2016&#46;</p><elsevierMultimedia ident="tbl0005"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">These data are consistent with the findings of a study by Stocker et al&#46;&#44;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a> who also found a significant interannual variability and reported an incidence of 1&#46;27 cases per 10&#44;000 births between 1994 and 1998 and an incidence that was nearly triple of 4&#46;15 cases per 10&#44;000 births between 2008 and 2012&#46; Given the increase in the number of CLMs in recent years&#44; we need to consider whether this results from an actual increase in incidence or improvements in diagnostic techniques&#46; Stocker et al&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a> favour the hypothesis of an increased incidence&#44; which is also supported by data in the EUROCAT registry&#44; which shows an increase of 6&#46;5&#37; in some CLMs &#40;95&#37; CI&#44; 3&#46;5&#37;&#8211;9&#46;4&#37;&#41;&#44; a phenomenon for which there is currently no plausible explanation&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">4</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">A salient finding in our study was the predominance of the female sex&#44; contrary to previous studies&#44;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">5</span></a> which have historically reported a predominance of the male sex&#46; This fact&#44; along with the significant interannual variability&#44; suggests that further studies with a longer follow-up are required to establish the current actual incidence of CLMs&#44; which will also require the participation of a larger number of hospitals to increase the number of cases included in the analyses&#46; Establishing the incidence of CLMs more accurately is relevant for the purpose of improving the management of these patients&#44; as the increase in the number of cases diagnosed prenatally has raised controversy regarding the correct approach to the management of asymptomatic patients&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">6</span></a></p></span>"
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          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Annual incidence of congenital lung malformations in Cantabria&#44; 2007&#8211;2016&#46;</p>"
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Scientific Letter
Does the incidence of congenital pulmonary malformations vary? 11 years of experience
¿Varía la incidencia de las malformaciones pulmonares congénitas?: 11 años de experiencia
Rocío Sancho Gutiérreza, Elena Pérez Belmontea, Ernesto de Diego Garcíab, Antonia Jesús López Lópezb, María Jesús Cabero Péreza,
Corresponding author
mariajesuscabero@gmail.com

Corresponding author.
a Servicio de Pediatría, Hospital Universitario Marqués de Valdecilla, Santander, Cantabria, Spain
b Cirugía Pediátrica, Hospital Universitario Marqués de Valdecilla, Santander, Cantabria, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Congenital lung malformations &#40;CLMs&#41; constitute a heterogeneous group of lung anomalies involving the airways&#44; parenchyma and blood vessels&#46; Historically&#44; they have been considered infrequent&#44; as their annual incidence has been documented to be of 1 case per 10&#44;000&#8211;35&#44;000 pregnancies<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a> or about 30&#8211;42 cases per 100&#44;000 inhabitants in the general population&#46; There are also authors that assert that their diagnosis has improved considerably with the introduction of routine prenatal ultrasound examinations&#44;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> but few studies have analysed how their incidence has actually been changing in recent times&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">In order to establish the incidence of CLMs in the autonomous community of Cantabria &#40;Spain&#41;&#44; we conducted a retrospective descriptive study of all cases of CLM managed in the Department of Paediatrics of the Hospital Universitario Marqu&#233;s de Valdecilla in Santander&#44; &#40;Cantabria&#44; Spain&#41;&#44; specifically in the Units of Paediatric Pulmonology and Paediatric Surgery&#44; in the period between January 2007 and December 2017&#46; Our hospital is the tertiary care hospital that manages all complicated pregnancies in Cantabria&#44; and we identified the patients using information obtained from the Department of Admissions and Clinical Records&#46; In addition&#44; we obtained data on the number of births during this period from the Instituto C&#225;ntabro de Estad&#237;stica &#40;Statistical Institute of Cantabria&#41; and the Instituto Nacional de Estad&#237;stica &#40;National Institute of Statistics&#41;&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">A total of 16 cases of CLM were diagnosed during the period under study&#44; corresponding to 9 female patients and 7 male patients&#46; The diagnosis was prenatal in 15 cases &#40;93&#46;7&#37;&#41;&#44; with the defect identified in the ultrasound examination performed at 22 weeks&#8217; gestation&#46; All of these patients were evaluated with a chest radiograph and a chest ultrasound examination within 24<span class="elsevierStyleHsp" style=""></span>h from birth&#44; and these imaging tests confirmed the presence of a CLM in the patients whose second trimester ultrasound examination had been positive&#46; In the remaining patient&#44; treated in 2007&#44; the CLM was diagnosed at age 4 months by means of a chest radiograph and a computed tomography angiogram&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall"><a class="elsevierStyleCrossRef" href="#tbl0005">Table 1</a> presents the data on the annual incidence of CLM&#46; The mean annual incidence was of 2&#46;05 cases per 10&#44;000 births &#40;standard deviation&#44; 3&#46;26&#41; and the median was 1&#46;98 cases &#40;interquartile range&#44; 6&#46;79&#41;&#44; calculated with data through December 2016&#46; We could not calculate the incidence for 2017 because at the time of this writing there is no published data on the number of births in Cantabria during this year&#46; We ought to highlight the considerable variability between years&#44; as 2 cases were diagnosed in a total of 31&#44;137 children born between 2007 and 2012&#44; compared to 12 cases diagnosed in a total of 17&#44;710 children born between 2013 and 2016&#46;</p><elsevierMultimedia ident="tbl0005"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">These data are consistent with the findings of a study by Stocker et al&#46;&#44;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a> who also found a significant interannual variability and reported an incidence of 1&#46;27 cases per 10&#44;000 births between 1994 and 1998 and an incidence that was nearly triple of 4&#46;15 cases per 10&#44;000 births between 2008 and 2012&#46; Given the increase in the number of CLMs in recent years&#44; we need to consider whether this results from an actual increase in incidence or improvements in diagnostic techniques&#46; Stocker et al&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a> favour the hypothesis of an increased incidence&#44; which is also supported by data in the EUROCAT registry&#44; which shows an increase of 6&#46;5&#37; in some CLMs &#40;95&#37; CI&#44; 3&#46;5&#37;&#8211;9&#46;4&#37;&#41;&#44; a phenomenon for which there is currently no plausible explanation&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">4</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">A salient finding in our study was the predominance of the female sex&#44; contrary to previous studies&#44;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">5</span></a> which have historically reported a predominance of the male sex&#46; This fact&#44; along with the significant interannual variability&#44; suggests that further studies with a longer follow-up are required to establish the current actual incidence of CLMs&#44; which will also require the participation of a larger number of hospitals to increase the number of cases included in the analyses&#46; Establishing the incidence of CLMs more accurately is relevant for the purpose of improving the management of these patients&#44; as the increase in the number of cases diagnosed prenatally has raised controversy regarding the correct approach to the management of asymptomatic patients&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">6</span></a></p></span>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0010">Please cite this article as&#58; Sancho Guti&#233;rrez R&#44; P&#233;rez Belmonte E&#44; de Diego Garc&#237;a E&#44; L&#243;pez L&#243;pez AJ&#44; Cabero P&#233;rez MJ&#46; &#191;Var&#237;a la incidencia de las malformaciones pulmonares cong&#233;nitas&#63;&#58; 11 a&#241;os de experiencia&#46; An Pediatr &#40;Barc&#41;&#46; 2019&#59;91&#58;53&#8211;54&#46;</p>"
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                  \t\t\t\t" scope="col" style="border-bottom: 2px solid black">Year&nbsp;\t\t\t\t\t\t\n
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              "nota" => "<p class="elsevierStyleNotepara" id="npar0005">We have not included data for 2017 because at the time of this writing the Instituto Nacional de Estad&#237;stica has not yet published the total number of births in Cantabria&#46;</p>"
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          ]
        ]
        "descripcion" => array:1 [
          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Annual incidence of congenital lung malformations in Cantabria&#44; 2007&#8211;2016&#46;</p>"
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                          "autores" => array:2 [
                            0 => "J&#46; Wall"
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                    0 => array:2 [
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                0 => array:2 [
                  "contribucion" => array:1 [
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                      "titulo" => "The increasing incidence of foetal echogenic congenital lung malformations&#58; an observational study"
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                        0 => array:2 [
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                          "autores" => array:5 [
                            0 => "L&#46;J&#46; Stocker"
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                            2 => "M&#46;P&#46; Stanton"
                            3 => "R&#46; Parasuraman"
                            4 => "D&#46;T&#46; Howe"
                          ]
                        ]
                      ]
                    ]
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                  "host" => array:1 [
                    0 => array:2 [
                      "doi" => "10.1002/pd.4507"
                      "Revista" => array:6 [
                        "tituloSerie" => "Prenat Diagn"
                        "fecha" => "2015"
                        "volumen" => "35"
                        "paginaInicial" => "148"
                        "paginaFinal" => "153"
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                          0 => array:2 [
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              "etiqueta" => "4"
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                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Trends in congenital anomalies in Europe from 1980 to 2012"
                      "autores" => array:1 [
                        0 => array:2 [
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                          "autores" => array:6 [
                            0 => "J&#46;K&#46; Morris"
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                            2 => "R&#46; Greenlees"
                            3 => "M&#46; Loane"
                            4 => "M&#46;C&#46; Addor"
                            5 => "L&#46; Arriola"
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                    ]
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                      "doi" => "10.1371/journal.pone.0194986"
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                        "tituloSerie" => "PLOS ONE"
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                0 => array:2 [
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                            0 => "I&#46; Ruchonnet-Metrailler"
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                            3 => "P&#46; Cros"
                            4 => "H&#46; Ducoin"
                            5 => "A&#46; Hadchouel"
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                      ]
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                        "tituloSerie" => "Pediatrics"
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                        "volumen" => "133"
                        "paginaInicial" => "e1285"
                        "paginaFinal" => "e1291"
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                          "etal" => false
                          "autores" => array:6 [
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                            1 => "N&#46; Musili"
                            2 => "N&#46; Matusko"
                            3 => "S&#46; Baker"
                            4 => "J&#46;D&#46; Geiger"
                            5 => "S&#46;M&#46; Kunisaki"
                          ]
                        ]
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Article information
ISSN: 23412879
Original language: English
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Idiomas
Anales de Pediatría (English Edition)
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