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Clinical Image
Late diagnosis of complex congenital pulmonary malformation: From symptoms and radiology to histopathology
Malformación pulmonar congénita compleja de diagnóstico tardío: de la clínica y radiología a la histopatología
Susan Pumacayo-Cárdenasa,
Corresponding author
pumacayocardenas@gmail.com

Corresponding author.
, Ernesto León-Bejaranob, Arturo Recabarren-Lozadab
a Departamento de Imagen Cardiaca, Instituto Nacional de Cardiología Ignacio Chavez, Universidad Nacional Autónoma de México, Ciudad de México, Mexico
b Departamento de Pediatría General, Clínica San Juan de Dios, Arequipa, Peru
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            "entidad" => "Departamento de Imagen Cardiaca&#44; Instituto Nacional de Cardiolog&#237;a Ignacio Chavez&#44; Universidad Nacional Aut&#243;noma de M&#233;xico&#44; Ciudad de M&#233;xico&#44; Mexico"
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        "titulo" => "Malformaci&#243;n pulmonar cong&#233;nita compleja de diagn&#243;stico tard&#237;o&#58; de la cl&#237;nica y radiolog&#237;a a la histopatolog&#237;a"
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          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A&#41; Anteroposterior chest X-ray shows right shift of the trachea and mediastinum&#44; signs of air trapping&#44; presence of a radiopaque image involving the left lower lobe&#44; where multiple rounded radiolucent images &#40;arrow&#41;&#44; and left pleural effusion are seen&#46; B&#41; Chest CT shows solid-cystic lesion&#44; without air bronchogram&#44; with multiple cysts of variable size &#40;arrow&#41;&#44; from 5 to 15&#8239;mm&#44; some converging to form larger cavities &#40;asterisk&#41;&#46; C and D&#41; Gross examination found a specimen measuring 12&#8239;&#215;&#8239;6&#8239;&#215;&#8239;8&#8239;cm of friable tissue &#40;arrow&#41; with multiple cavities of varying size and purulent content &#40;asterisk&#41;&#46; E&#41; Microscopy &#40;H&#38;E staining &#215;40&#41; shows marked architectural alteration with multiple cystic formations &#40;asterisks&#41;&#44; the largest cystic formations &#40;right&#44; magnification &#215;100&#41; being covered by a pseudostratified columnar epithelium &#40;arrow&#41; surrounded by thin fibromuscular walls and dense inflammatory infiltrate in the cystic and interstitial space&#44; consistent with infected congenital cystic adenomatoid malformation type 2&#46; F&#41; Follow-up CT showing right and left upper lobe compensatory emphysema&#44; and left posterior pleural and lateral thickening&#46;</p>"
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Our patient was a young girl&#44; 6 years of age&#44; with no prenatal check-ups or previous episodes of pneumonia&#46; She was hospitalized for a 5-day history of signs and symptoms consistent with left basal pneumonia and mild respiratory distress&#46; Chest X-ray showed pleural and parenchymal involvement of the left lower lobe &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A&#41;&#46; She was treated with broad-spectrum antibiotics&#46; Serologies for echinococcus granulosus and tuberculous mycobacteria were negative&#46; Clinical and radiological progress were unfavorable&#44; so a chest computed tomography &#40;CT&#41; was performed &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>B&#41;&#44; which suggested complicated pulmonary sequestration or congenital malformation&#46; Surgical treatment &#40;left lower lobectomy&#41; was performed&#59; the anatomical piece &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>C and D&#41; had its own visceral pleura&#44; with independent irrigation from a fine artery originating in the thoracic aorta&#44; characteristic of extralobar pulmonary sequestration &#40;EPS&#41;&#59; the histopathological study &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>E&#41; confirmed infected congenital cystic adenomatoid malformation &#40;CCAM&#41; type 2&#46; Her subsequent progress was favorable&#44; and a follow-up CT was performed at 6 months &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>F&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">CCAM is a rare congenital abnormality with a prevalence of approximately 8&#8211;9&#47;100&#44;000&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a> The association with EPS<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> &#40;hybrid form&#41;&#44; as in the present case&#44; is very rare&#44; even more so in late presentation &#40;older than 1&#8239;year of age&#41;&#46; It is important for the clinician to be aware of the clinical and radiological course&#44; and the differential diagnoses that include congenital &#40;diaphragmatic hernia&#44; pulmonary cysts&#44; etc&#46;&#41; and infectious &#40;necrotizing pneumonia&#44; tuberculosis&#44; abscess&#44; and pulmonary hydatidosis&#41; causes&#44; in order to make an early diagnosis and provide timely treatment&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p></span>"
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    "fechaRecibido" => "2019-06-11"
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        "etiqueta" => "&#9734;"
        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Pumacayo-C&#225;rdenas S&#44; Le&#243;n-Bejarano E&#44; Recabarren-Lozad A&#46; Malformaci&#243;n pulmonar cong&#233;nita compleja de diagn&#243;stico tard&#237;o&#58; de la cl&#237;nica y radiolog&#237;a a la histopatolog&#237;a&#46; Arch Bronconeumol&#46; 2020&#59;56&#58;523&#46;</p>"
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          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A&#41; Anteroposterior chest X-ray shows right shift of the trachea and mediastinum&#44; signs of air trapping&#44; presence of a radiopaque image involving the left lower lobe&#44; where multiple rounded radiolucent images &#40;arrow&#41;&#44; and left pleural effusion are seen&#46; B&#41; Chest CT shows solid-cystic lesion&#44; without air bronchogram&#44; with multiple cysts of variable size &#40;arrow&#41;&#44; from 5 to 15&#8239;mm&#44; some converging to form larger cavities &#40;asterisk&#41;&#46; C and D&#41; Gross examination found a specimen measuring 12&#8239;&#215;&#8239;6&#8239;&#215;&#8239;8&#8239;cm of friable tissue &#40;arrow&#41; with multiple cavities of varying size and purulent content &#40;asterisk&#41;&#46; E&#41; Microscopy &#40;H&#38;E staining &#215;40&#41; shows marked architectural alteration with multiple cystic formations &#40;asterisks&#41;&#44; the largest cystic formations &#40;right&#44; magnification &#215;100&#41; being covered by a pseudostratified columnar epithelium &#40;arrow&#41; surrounded by thin fibromuscular walls and dense inflammatory infiltrate in the cystic and interstitial space&#44; consistent with infected congenital cystic adenomatoid malformation type 2&#46; F&#41; Follow-up CT showing right and left upper lobe compensatory emphysema&#44; and left posterior pleural and lateral thickening&#46;</p>"
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                    0 => array:2 [
                      "titulo" => "Children with congenital cystic adenomatoid malformation of the lung CT diagnosis"
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                        0 => array:2 [
                          "etal" => false
                          "autores" => array:2 [
                            0 => "Z&#46;J&#46; Zhang"
                            1 => "M&#46;X&#46; Huang"
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                        "tituloSerie" => "Int J Clin Exp Med"
                        "fecha" => "2015"
                        "volumen" => "8"
                        "paginaInicial" => "4415"
                        "paginaFinal" => "4419"
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                            "url" => "https://www.ncbi.nlm.nih.gov/pubmed/26064363"
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                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Congenital cystic lesions of the lungs&#58; the perils of misdiagnosis &#8211; a single-center experience"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => false
                          "autores" => array:5 [
                            0 => "V&#46; Shankar Raman"
                            1 => "S&#46; Agarwala"
                            2 => "V&#46; Bhatnagar"
                            3 => "S&#46;S&#46; Panda"
                            4 => "K&#46;G&#46; Arun"
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                    0 => array:2 [
                      "doi" => "10.4103/0970-2113.152616"
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                        "tituloSerie" => "Lung India"
                        "fecha" => "2015"
                        "volumen" => "32"
                        "paginaInicial" => "116"
                        "paginaFinal" => "118"
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                          0 => array:2 [
                            "url" => "https://www.ncbi.nlm.nih.gov/pubmed/25814794"
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Article information
ISSN: 15792129
Original language: English
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