Scientific Letter
Invasive Mucinous Adenocarcinoma in Congenital Pulmonary Airway Malformation: A Case Report
Adenocarcinoma mucinoso invasivo sobre malformación congénita de la vía aérea pulmonar (MCVAP): a propósito de un caso
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En el lóbulo inferior izquierdo se observa área de desestructuración del parénquima pulmonar con menor vascularización (círculo rojo). La imagen quística de mayor tamaño presenta paredes engrosadas y nivel hidroaéreo (flecha). B: angio-TC torácica con reconstrucciones MIP y MPR. Existe una rama arterial proveniente del tronco celíaco (flechas) que irriga a la imagen quística así como a la zona de desestructuración. C: radiografía de tórax en proyección lateral. Se observa una consolidación en el lóbulo inferior izquierdo (círculo rojo), que en el contexto clínico de la paciente es sugestivo de neumonía. D: TC torácica con contraste intravenoso y adquisición de imágenes en fase arterial. Persiste estable la lesión quística de mayor tamaño (flecha). Se aprecia una consolidación hipodensa en el seno de la MCVAP (círculo rojo), con vasos en su interior. E: visión macroscópica de la pieza quirúrgica. Se observa una condensación blanquecina (círculo rojo), indurada, que se extiende a través del parénquima pulmonar en forma de nodulillos de tamaño variable. F: visión microscópica de la lesión. Extensa malformación arquitectural con áreas quísticas (flechas negras) en relación con MCVAP. Proliferación neoplásica de células epiteliales mucinosas atípicas que cubren los septos alveolares, con áreas de patrón seudoneumónico (círculos rojos).</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Alexandre Soler-Perromat, Ivan Vollmer, José Ramírez, Marcelo Sánchez" "autores" => array:4 [ 0 => array:2 [ "nombre" => "Alexandre" "apellidos" => "Soler-Perromat" ] 1 => array:2 [ "nombre" => "Ivan" "apellidos" => "Vollmer" ] 2 => array:2 [ "nombre" => "José" "apellidos" => "Ramírez" ] 3 => array:2 [ "nombre" => "Marcelo" "apellidos" => "Sánchez" ] ] ] ] ] "idiomaDefecto" => "es" "Traduccion" => array:1 [ "en" => array:9 [ "pii" => "S1579212919301272" "doi" => "10.1016/j.arbr.2019.05.006" "estado" => "S300" "subdocumento" => "" "abierto" => array:3 [ "ES" => false "ES2" => false "LATM" => false ] "gratuito" => false "lecturas" => array:1 [ "total" => 0 ] "idiomaDefecto" => "en" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1579212919301272?idApp=UINPBA00003Z" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0300289618304617?idApp=UINPBA00003Z" "url" => "/03002896/0000005500000007/v1_201907020949/S0300289618304617/v1_201907020949/es/main.assets" ] ] "itemSiguiente" => array:19 [ "pii" => "S1579212919301259" "issn" => "15792129" "doi" => "10.1016/j.arbr.2019.05.005" "estado" => "S300" "fechaPublicacion" => "2019-07-01" "aid" => "2045" "copyright" => "SEPAR" "documento" => "simple-article" "crossmark" => 1 "subdocumento" => "crp" "cita" => "Arch Bronconeumol. 2019;55:386-7" "abierto" => array:3 [ "ES" => false "ES2" => false "LATM" => false ] "gratuito" => false "lecturas" => array:2 [ "total" => 103 "formatos" => array:3 [ "EPUB" => 48 "HTML" => 34 "PDF" => 21 ] ] "en" => array:11 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Scientific Letter</span>" "titulo" => "Hemoptysis and Bronchiolitis Obliterans in Children With Recurrent Respiratory Papillomatosis: Adverse Reactions to Nebulized Cidofovir" "tienePdf" => "en" "tieneTextoCompleto" => "en" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "386" "paginaFinal" => "387" ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Hemoptisis y bronquiolitis obliterante en niños con papilomatosis laríngea recurrente: reacciones adversas al cidofovir nebulizado" ] ] "contieneTextoCompleto" => array:1 [ "en" => true ] "contienePdf" => array:1 [ "en" => true ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1355 "Ancho" => 1665 "Tamanyo" => 202090 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A–C) Fiberoptic bronchoscopy image: pearly skin lesions in the posterior third of the vocal cords and vestibular folds of the glottis, extending to the trachea, causing 90% stenosis of the tracheal lumen. 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Axial computed tomography with the lung (A) and mediastinal (B) window settings showing an area of consolidation in the left lower lobe containing a markedly dilated bronchus. In (C), histopathological section demonstrating a dilated bronchiole with peribronchiolar infiltration of neoplastic cells (hematoxylin and eosin stain, magnification 40×). In (D), histological section showing proliferation of lymphoid cells (hematoxylin and eosin stain, magnification 400×).</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Edson Marchiori, Bruno Hochhegger, Gláucia Zanetti" "autores" => array:3 [ 0 => array:2 [ "nombre" => "Edson" "apellidos" => "Marchiori" ] 1 => array:2 [ "nombre" => "Bruno" "apellidos" => "Hochhegger" ] 2 => array:2 [ "nombre" => "Gláucia" "apellidos" => "Zanetti" ] ] ] ] ] "idiomaDefecto" => "en" "Traduccion" => array:1 [ "en" => array:9 [ "pii" => "S0300289618304629" "doi" => "10.1016/j.arbres.2018.11.017" "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/S0300289618304629?idApp=UINPBA00003Z" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1579212919301867?idApp=UINPBA00003Z" "url" => "/15792129/0000005500000007/v1_201907020952/S1579212919301867/v1_201907020952/en/main.assets" ] "en" => array:15 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Scientific Letter</span>" "titulo" => "Invasive Mucinous Adenocarcinoma in Congenital Pulmonary Airway Malformation: A Case Report" "tieneTextoCompleto" => true "saludo" => "To the Editor," "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "384" "paginaFinal" => "385" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Alexandre Soler-Perromat, Ivan Vollmer, José Ramírez, Marcelo Sánchez" "autores" => array:4 [ 0 => array:3 [ "nombre" => "Alexandre" "apellidos" => "Soler-Perromat" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 1 => array:4 [ "nombre" => "Ivan" "apellidos" => "Vollmer" "email" => array:1 [ 0 => "vollmer@clinic.ub.es" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 2 => array:3 [ "nombre" => "José" "apellidos" => "Ramírez" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "Marcelo" "apellidos" => "Sánchez" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicio de Radiodiagnóstico (CDIC), Hospital Clínic, Barcelona, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Anatomía Patológica (CDB), Hospital Clínic, Barcelona, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Adenocarcinoma mucinoso invasivo sobre malformación congénita de la vía aérea pulmonar (MCVAP): a propósito de un caso" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 989 "Ancho" => 1400 "Tamanyo" => 197752 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A: High-resolution CT, axial plane. Left lower lobe shows an area of destruction of the pulmonary parenchyma with less vascularity (red circle). The image of the larger cyst shows thickened walls and air-fluid level (arrow). B: Chest angio-CT scan with MIP and MPR reconstructions. An arterial branch is seen emerging from the celiac trunk (arrows) that supplies blood to the cystic mass and to the area of destruction. C: Chest X-ray, lateral projection. Consolidation seen in the left lower lobe (red circle), which in the clinical context of the patient was suggestive of pneumonia. D: Chest CT with intravenous contrast and imaging in arterial phase. The larger cystic lesion remains stable (arrow). Hypodense consolidation containing vessels seen within the CPAM (red circle). E: Macroscopic view of the surgical specimen. A whitish, indurated, condensation (red circle) is seen, extending through the pulmonary parenchyma in the form of nodules of variable size. F: Microscopic view of the lesion. Extensive architectural malformation with cystic areas (black arrows) associated with CPAM. Neoplastic proliferation of atypical mucinous epithelial cells covering the alveolar septa, with areas of pseudopneumonic pattern (red circles).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">We report the case of a 29-year-old woman, former smoker, with congenital pulmonary airway malformation (CPAM) diagnosed previously using chest computed tomography (CT) (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A). The malformation consisted of anomalous arterial vascularization (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>B), giving rise to a hybrid lesion: CPAM+pulmonary sequestration.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">Three and a half years later, the patient presented in the emergency department due to fever, cough, and pain in the left hemithorax. Chest radiograph was performed (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>C), which revealed consolidation in the left lower lobe, which in the clinical context of the patient was suggestive of pneumonia. Given her persistent pain and dry cough after antibiotic treatment, we decided to expand the study with a chest CT (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>D), which showed consolidation within the CPAM. The differential diagnosis of this consolidation was determined to be either a bacterial superinfection of the CPAM or a neoproliferative process. Bacterial superinfection was the most probable diagnosis given the visualization of consolidation, accompanied by a concomitant predisposing lesion. A neoproliferative process, on the other hand, was less likely, taking into account the uncommon pattern of presentation and the young age of the patient. Due to the suspicion of superinfection and the high probability of recurrence, we decided to perform lower left lobectomy. The surgical specimen was sent for pathology study (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>E and F), which gave a definitive diagnosis of invasive mucinous adenocarcinoma in CPAM, with a pseudopneumonic pattern.</p><p id="par0015" class="elsevierStylePara elsevierViewall">CPAMs form a heterogeneous group of cystic lesions and non-cystic lesions caused by early changes in the development of the pulmonary airway. The estimated incidence is 1/25<span class="elsevierStyleHsp" style=""></span>000–35<span class="elsevierStyleHsp" style=""></span>000 newborns.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> There are 5 subtypes, Type I being the most frequent and the one associated with the largest cysts.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">2</span></a> Most are supplied by blood from the pulmonary circulation, with the exception of hybrid lesions that may receive blood directly from the systemic arterial circulation.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">Malignant transformation has been reported in some cases of type I CPAM, a situation that is associated with a mutation in the K-<span class="elsevierStyleItalic">ras</span> gene. In 2013, Ishida et al. published a case report with a review of the literature,<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a> in which the authors described the fourth documented case to date of this type of CPAM-associated neoplasm with K-<span class="elsevierStyleItalic">ras</span> mutation. The case we describe also had the K<span class="elsevierStyleItalic">-ras</span> mutation. Radiological management is complex, since the similarity of this entity and bacterial pneumonia on imaging studies may delay diagnosis.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a> Pseudopneumonic pattern of the tumor is associated with a worse prognosis, while treatment of choice is surgical resection.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Soler-Perromat A, Vollmer I, Ramírez J, Sánchez M. Adenocarcinoma mucinoso invasivo sobre malformación congénita de la vía aérea pulmonar (MCVAP): a propósito de un caso. Arch Bronconeumol. 2019;55:384–385.</p>" ] ] "multimedia" => array:1 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 989 "Ancho" => 1400 "Tamanyo" => 197752 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A: High-resolution CT, axial plane. Left lower lobe shows an area of destruction of the pulmonary parenchyma with less vascularity (red circle). The image of the larger cyst shows thickened walls and air-fluid level (arrow). B: Chest angio-CT scan with MIP and MPR reconstructions. An arterial branch is seen emerging from the celiac trunk (arrows) that supplies blood to the cystic mass and to the area of destruction. C: Chest X-ray, lateral projection. Consolidation seen in the left lower lobe (red circle), which in the clinical context of the patient was suggestive of pneumonia. D: Chest CT with intravenous contrast and imaging in arterial phase. The larger cystic lesion remains stable (arrow). Hypodense consolidation containing vessels seen within the CPAM (red circle). E: Macroscopic view of the surgical specimen. A whitish, indurated, condensation (red circle) is seen, extending through the pulmonary parenchyma in the form of nodules of variable size. F: Microscopic view of the lesion. Extensive architectural malformation with cystic areas (black arrows) associated with CPAM. Neoplastic proliferation of atypical mucinous epithelial cells covering the alveolar septa, with areas of pseudopneumonic pattern (red circles).</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" => "Congenital pulmonary airway malformation in an adult male: a case report with literature review" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "D. Baral" 1 => "B. Adhikari" 2 => "D. Zaccarini" 3 => "R.M. Dongol" 4 => "B. 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Multidetector" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1148/radiol.2473062124" "Revista" => array:6 [ "tituloSerie" => "Radiology" "fecha" => "2008" "volumen" => "247" "paginaInicial" => "632" "paginaFinal" => "648" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/18487532" "web" => "Medline" ] ] ] ] ] ] ] ] 2 => array:3 [ "identificador" => "bib0040" "etiqueta" => "3" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Congenital lung abnormalities: embryologic features, prenatal diagnosis, and postnatal radiologic-pathologic correlation" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "D.R. Biyyam" 1 => "T. Chapman" 2 => "M.R. Ferguson" 3 => "G. Deutsch" 4 => "M.K. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 3 | 1 | 4 |
| 2024 October | 43 | 25 | 68 |
| 2024 September | 45 | 18 | 63 |
| 2024 August | 57 | 35 | 92 |
| 2024 July | 46 | 25 | 71 |
| 2024 June | 42 | 32 | 74 |
| 2024 May | 64 | 52 | 116 |
| 2024 April | 34 | 34 | 68 |
| 2024 March | 37 | 24 | 61 |
| 2024 February | 33 | 18 | 51 |
| 2023 March | 9 | 5 | 14 |
| 2023 February | 31 | 16 | 47 |
| 2023 January | 36 | 30 | 66 |
| 2022 December | 53 | 37 | 90 |
| 2022 November | 47 | 31 | 78 |
| 2022 October | 68 | 42 | 110 |
| 2022 September | 45 | 17 | 62 |
| 2022 August | 46 | 46 | 92 |
| 2022 July | 39 | 42 | 81 |
| 2022 June | 42 | 32 | 74 |
| 2022 May | 35 | 51 | 86 |
| 2022 April | 36 | 30 | 66 |
| 2022 March | 37 | 40 | 77 |
| 2022 February | 44 | 30 | 74 |
| 2022 January | 32 | 48 | 80 |
| 2021 December | 28 | 42 | 70 |
| 2021 November | 41 | 48 | 89 |
| 2021 October | 40 | 54 | 94 |
| 2021 September | 32 | 41 | 73 |
| 2021 August | 25 | 36 | 61 |
| 2021 July | 22 | 22 | 44 |
| 2020 October | 1 | 0 | 1 |
| 2020 August | 2 | 4 | 6 |
| 2020 March | 26 | 3 | 29 |
| 2020 February | 18 | 13 | 31 |
| 2020 January | 21 | 9 | 30 |
| 2019 December | 2 | 0 | 2 |
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