Late onset congenital cystic adenomatous malformation associated with intra-lobar pulmonary sequestration
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Dabó, R. Gomes, V. Hespanhol, H. Queiroga" "autores" => array:4 [ 0 => array:2 [ "Iniciales" => "H." "apellidos" => "Dabó" ] 1 => array:2 [ "Iniciales" => "R." "apellidos" => "Gomes" ] 2 => array:2 [ "Iniciales" => "V." "apellidos" => "Hespanhol" ] 3 => array:2 [ "Iniciales" => "H." "apellidos" => "Queiroga" ] ] ] ] ] "idiomaDefecto" => "en" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/X087321591536499X?idApp=UINPBA00004E" "url" => "/08732159/0000002100000005/v0_201604141143/X087321591536499X/v0_201604141143/en/main.assets" ] "en" => array:11 [ "idiomaDefecto" => true "titulo" => "Late onset congenital cystic adenomatous malformation associated with intra-lobar pulmonary sequestration" "tieneTextoCompleto" => true "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "282" "paginaFinal" => "284" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "E. Silva, J. Vale, A. Reis" "autores" => array:3 [ 0 => array:4 [ "Iniciales" => "E." "apellidos" => "Silva" "email" => array:1 [ 0 => "eloisacs@gmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "affa" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor1" ] ] ] 1 => array:3 [ "Iniciales" => "J." "apellidos" => "Vale" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "affa" ] ] ] 2 => array:3 [ "Iniciales" => "A." "apellidos" => "Reis" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "affa" ] ] ] ] "afiliaciones" => array:1 [ 0 => array:3 [ "entidad" => "Pulmonology Department, Centro-Hospitalar Tondela-Viseu, Portugal" "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "affa" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor1" "etiqueta" => "<span class="elsevierStyleSup">*</span>" "correspondencia" => "Corresponding author. eloisacs@gmail.com" ] ] ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig1" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "copyright" => "Elsevier España" "figura" => array:1 [ 0 => array:4 [ "imagen" => "320v21n05-90436500fig1.jpg" "Alto" => 2363 "Ancho" => 1795 "Tamanyo" => 2143579 ] ] "descripcion" => array:1 [ "en" => "(A) The first chest CT performed, showing a heterogeneous posteromedial consolidation in the left lower lobe comprising multiple cysts. (B) The second chest CT showing posteromedial hepatization of the left lower field, compatible with superimposed infection. (C) CT angiography showing the anomalous artery arising from the thoracic aorta. (D) CT angiography images showing the venous drainage (arrow) to the pulmonary veins. (E¿F) 3D CT reconstructions showing the anomalous artery." ] ] ] "textoCompleto" => "<p class="elsevierStylePara">Dear Editor,</p><p class="elsevierStylePara">Lung congenital malformations, which include congenital cystic adenomatous malformation (CCAM) and pulmonary sequestration (PS), are rare. PS accounts for 0.15–6.45% of lung malformations and is characterized by a nonfunctioning mass of pulmonary tissue that receives arterial supply from the systemic circulation and lacks communication with the tracheobronchial tree. It is classified as intralobar PS (IL-PS) if it is contained within the normal lung and drains to the pulmonary veins and as extralobar PS (EL-PS) if it is separated from the surrounding parenchyma by his own pleural covering and drains to the systemic circulation.<a href="#bib8" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">1</span></a><span class="elsevierStyleSup">, </span><a href="#bib9" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">2</span></a><span class="elsevierStyleSup">, </span><a href="#bib10" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">3</span></a> CCAM is responsible for 25–30% of congenital lung malformations. It is a hamartomatous lesion with anomalous development, characterized by a mass of lung tissue from different pulmonary origins and various degrees of cyst formation that communicates with the tracheobronchial tree and is supplied and drained by the pulmonary circulation.<a href="#bib8" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">1</span></a><span class="elsevierStyleSup">, </span><a href="#bib9" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">2</span></a><span class="elsevierStyleSup">, </span><a href="#bib10" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">3</span></a> CCAM is classified into 5 types, based on histological features and level of malformation in the airway and lung.<a href="#bib11" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">4</span></a> These lesions are usually diagnosed in the neonatal period, their presentation in adulthood is less frequent.<a href="#bib8" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">1</span></a><span class="elsevierStyleSup">, </span><a href="#bib9" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">2</span></a> Despite the fact that they are two of the most common lung malformations their association is uncommon, particularly for IL-PS and CCAM.<a href="#bib9" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">2</span></a><span class="elsevierStyleSup">, </span><a href="#bib12" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">5</span></a></p><p class="elsevierStylePara">We report a case of a 36 year-old woman, never smoker, observed in the outpatient clinic for recurrent pneumonias. Over the past 5 years, she reported several pneumonias, all slowly resolving and in need of multiple courses of antibiotics. Between infections she complained of dry cough and left pleuritic chest pain. She worked as a care assistant in a continuing care unit and apart from the recurrent infections had no other significant medical history. Physical examination was unremarkable and laboratory findings and bronchoscopy were normal. Chest X-ray showed a reticular opacity in the left lower field and a computed tomography (CT), performed two months earlier, showed a heterogeneous consolidation on the left lower field with millimetric air spaces compatible with cysts (<a href="#f0005" class="elsevierStyleCrossRefs">Fig. 1</a>A). Chest CT was repeated, revealing a 10 cm heterogeneous consolidation on the left inferior lobe, with arterial supply arising from the thoracic descending aorta and venous drainage to the pulmonary veins (<a href="#f0005" class="elsevierStyleCrossRefs">Fig. 1</a>B–F). The second CT was performed during another infectious episode and radiologic findings suggested IL-PS with superimposed infection. The infection was resolved and the patient was submitted to left inferior lobectomy with an uneventful post-operative recovery. Surgical procedure confirmed the anomalous vascularization and histological findings were compatible with CCAM type 2. A diagnosis of CCAM type 2 associated with IL-PS was established based on imaging and histological findings. The patient remained asymptomatic 1 year after surgery.</p><a name="f0005" class="elsevierStyleCrossRefs"></a><p class="elsevierStylePara"><img src="320v21n05-90436500fig1.jpg" alt="(A) The first chest CT performed, showing a heterogeneous posteromedial consolidation in the left lower lobe comprising multiple cysts. (B) The second chest CT showing posteromedial hepatization of the left lower field, compatible with superimposed infection. (C) CT angiography showing the anomalous artery arising from the thoracic aorta. (D) CT angiography images showing the venous drainage (arrow) to the pulmonary veins. (E–F) 3D CT reconstructions showing the anomalous artery."></img></p><p class="elsevierStylePara">Fig. 1. (A) The first chest CT performed, showing a heterogeneous posteromedial consolidation in the left lower lobe comprising multiple cysts. (B) The second chest CT showing posteromedial hepatization of the left lower field, compatible with superimposed infection. (C) CT angiography showing the anomalous artery arising from the thoracic aorta. (D) CT angiography images showing the venous drainage (arrow) to the pulmonary veins. (E–F) 3D CT reconstructions showing the anomalous artery.</p><p class="elsevierStylePara">This case combines an unusual association of congenital malformations – IL-PS and CCAM – with an infrequent diagnosis in adulthood. Few cases are described in literature of CCAM and IL-PS association.<a href="#bib9" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">2</span></a><span class="elsevierStyleSup">, </span><a href="#bib12" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">5</span></a><span class="elsevierStyleSup">, </span><a href="#bib13" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">6</span></a><span class="elsevierStyleSup">, </span><a href="#bib14" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">7</span></a> We found reports of presenting in adulthood were even scarcer.<a href="#bib13" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">6</span></a></p><p class="elsevierStylePara">These lesions are usually diagnosed prenatally or in the first years of life. Both can have a wide spectrum of presentations ranging from asymptomatic to neonatal death depending on the size of the mass and consequent physiological impairment.<a href="#bib8" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">1</span></a><span class="elsevierStyleSup">, </span><a href="#bib9" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">2</span></a> After the first year of life recurrent infections are the most common presentation for both pathologies.<a href="#bib8" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">1</span></a><span class="elsevierStyleSup">, </span><a href="#bib9" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">2</span></a><span class="elsevierStyleSup">, </span><a href="#bib10" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">3</span></a><span class="elsevierStyleSup">, </span><a href="#bib11" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">4</span></a> It is important to note that potential for malignant transformation has been described in both entities, with stronger evidence in CCAM.<a href="#bib10" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">3</span></a> Imaging has an important part in diagnosis of these lesions. Before birth ultrasound has a key role and after birth CT scan is the gold standard. Morphological features, location and blood supply can help distinguish the different malformations but this differentiation can be particularly difficult. Final diagnosis is established by histological analysis.<a href="#bib8" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">1</span></a><span class="elsevierStyleSup">, </span><a href="#bib9" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">2</span></a><span class="elsevierStyleSup">, </span><a href="#bib10" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">3</span></a> CCAM is found in most cases to be an isolated finding, although type 2 has been described with other anomalies.<a href="#bib8" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">1</span></a><span class="elsevierStyleSup">, </span><a href="#bib9" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">2</span></a> Whereas the existence of further congenital anomalies, such as lung, cardiac, diaphragm or chest wall anomalies, is more frequent in PS, particularly in cases of EL-PS, it only happens in 15% of IL-PS cases.<a href="#bib8" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">1</span></a><span class="elsevierStyleSup">, </span><a href="#bib12" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">5</span></a> The mainstay of treatment for CCAM and PS is surgical resection, lobectomy is the usual procedure of choice.<a href="#bib8" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">1</span></a><span class="elsevierStyleSup">, </span><a href="#bib9" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">2</span></a><span class="elsevierStyleSup">, </span><a href="#bib10" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">3</span></a> The authors present this case because it is so rare. Although uncommon and infrequently diagnosed in adults, congenital malformations should be included in the differential diagnosis of repeated infections, particularly if they are on the same lobe.</p><a name="sec0005" class="elsevierStyleCrossRefs"></a><span class="elsevierStyleSectionTitle">Conflicts of interest</span><p class="elsevierStylePara">The authors have no conflicts of interest to declare.</p><p class="elsevierStylePara">Corresponding author. eloisacs@gmail.com</p>" "pdfFichero" => "320v21n05a90436500pdf001.pdf" "tienePdf" => true "multimedia" => array:2 [ 0 => array:8 [ "identificador" => "fig1" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "copyright" => "Elsevier España" "figura" => array:1 [ 0 => array:4 [ "imagen" => "320v21n05-90436500fig1.jpg" "Alto" => 2363 "Ancho" => 1795 "Tamanyo" => 2143579 ] ] "descripcion" => array:1 [ "en" => "(A) The first chest CT performed, showing a heterogeneous posteromedial consolidation in the left lower lobe comprising multiple cysts. (B) The second chest CT showing posteromedial hepatization of the left lower field, compatible with superimposed infection. (C) CT angiography showing the anomalous artery arising from the thoracic aorta. (D) CT angiography images showing the venous drainage (arrow) to the pulmonary veins. (E¿F) 3D CT reconstructions showing the anomalous artery." ] ] 1 => array:6 [ "identificador" => "fig2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "copyright" => "Elsevier España" "descripcion" => array:1 [ "en" => "(A) The first chest CT performed, showing a heterogeneous posteromedial consolidation in the left lower lobe comprising multiple cysts. (B) The second chest CT showing posteromedial hepatization of the left lower field, compatible with superimposed infection. (C) CT angiography showing the anomalous artery arising from the thoracic aorta. (D) CT angiography images showing the venous drainage (arrow) to the pulmonary veins. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 13 | 7 | 20 |
| 2024 October | 50 | 31 | 81 |
| 2024 September | 78 | 28 | 106 |
| 2024 August | 70 | 39 | 109 |
| 2024 July | 69 | 38 | 107 |
| 2024 June | 55 | 37 | 92 |
| 2024 May | 62 | 44 | 106 |
| 2024 April | 61 | 36 | 97 |
| 2024 March | 57 | 33 | 90 |
| 2024 February | 49 | 24 | 73 |
| 2024 January | 43 | 24 | 67 |
| 2023 December | 43 | 30 | 73 |
| 2023 November | 49 | 31 | 80 |
| 2023 October | 45 | 42 | 87 |
| 2023 September | 50 | 31 | 81 |
| 2023 August | 39 | 23 | 62 |
| 2023 July | 44 | 29 | 73 |
| 2023 June | 51 | 14 | 65 |
| 2023 May | 65 | 24 | 89 |
| 2023 April | 64 | 22 | 86 |
| 2023 March | 89 | 20 | 109 |
| 2023 February | 54 | 26 | 80 |
| 2023 January | 47 | 14 | 61 |
| 2022 December | 80 | 20 | 100 |
| 2022 November | 75 | 46 | 121 |
| 2022 October | 83 | 39 | 122 |
| 2022 September | 53 | 24 | 77 |
| 2022 August | 75 | 49 | 124 |
| 2022 July | 54 | 49 | 103 |
| 2022 June | 42 | 35 | 77 |
| 2022 May | 58 | 40 | 98 |
| 2022 April | 57 | 33 | 90 |
| 2022 March | 47 | 61 | 108 |
| 2022 February | 44 | 37 | 81 |
| 2022 January | 44 | 38 | 82 |
| 2021 December | 39 | 37 | 76 |
| 2021 November | 53 | 43 | 96 |
| 2021 October | 41 | 46 | 87 |
| 2021 September | 42 | 30 | 72 |
| 2021 August | 41 | 27 | 68 |
| 2021 July | 43 | 23 | 66 |
| 2021 June | 56 | 30 | 86 |
| 2021 May | 39 | 36 | 75 |
| 2021 April | 71 | 61 | 132 |
| 2021 March | 48 | 20 | 68 |
| 2021 February | 38 | 20 | 58 |
| 2021 January | 37 | 18 | 55 |
| 2020 December | 33 | 16 | 49 |
| 2020 November | 41 | 26 | 67 |
| 2020 October | 44 | 13 | 57 |
| 2020 September | 56 | 23 | 79 |
| 2020 August | 57 | 16 | 73 |
| 2020 July | 75 | 19 | 94 |
| 2020 June | 66 | 24 | 90 |
| 2020 May | 48 | 16 | 64 |
| 2020 April | 52 | 12 | 64 |
| 2020 March | 45 | 18 | 63 |
| 2020 February | 76 | 30 | 106 |
| 2020 January | 63 | 14 | 77 |
| 2019 December | 53 | 19 | 72 |
| 2019 November | 65 | 14 | 79 |
| 2019 October | 55 | 22 | 77 |
| 2019 September | 36 | 17 | 53 |
| 2019 August | 109 | 12 | 121 |
| 2019 July | 106 | 18 | 124 |
| 2019 June | 105 | 11 | 116 |
| 2019 May | 125 | 17 | 142 |
| 2019 April | 109 | 26 | 135 |
| 2019 March | 146 | 15 | 161 |
| 2019 February | 118 | 7 | 125 |
| 2019 January | 135 | 32 | 167 |
| 2018 December | 76 | 7 | 83 |
| 2018 November | 18 | 2 | 20 |
| 2018 October | 17 | 11 | 28 |
| 2018 September | 21 | 9 | 30 |
| 2018 August | 36 | 28 | 64 |
| 2018 July | 32 | 17 | 49 |
| 2018 June | 37 | 15 | 52 |
| 2018 May | 47 | 11 | 58 |
| 2018 April | 65 | 15 | 80 |
| 2018 March | 101 | 25 | 126 |
| 2018 February | 23 | 8 | 31 |
| 2018 January | 47 | 18 | 65 |
| 2017 December | 54 | 20 | 74 |
| 2017 November | 11 | 20 | 31 |
| 2017 October | 21 | 11 | 32 |
| 2017 September | 13 | 10 | 23 |
| 2017 August | 26 | 9 | 35 |
| 2017 July | 14 | 11 | 25 |
| 2017 June | 20 | 15 | 35 |
| 2017 May | 17 | 17 | 34 |
| 2017 April | 9 | 7 | 16 |
| 2017 March | 10 | 5 | 15 |
| 2017 February | 5 | 7 | 12 |
| 2017 January | 5 | 5 | 10 |
| 2016 December | 7 | 6 | 13 |
| 2016 November | 9 | 7 | 16 |
| 2016 October | 2 | 4 | 6 |
| 2016 September | 7 | 5 | 12 |
| 2016 August | 9 | 6 | 15 |
| 2016 July | 9 | 11 | 20 |
| 2016 April | 13 | 12 | 25 |
| 2016 March | 22 | 12 | 34 |
| 2016 February | 31 | 15 | 46 |
| 2016 January | 22 | 17 | 39 |
| 2015 December | 62 | 32 | 94 |
| 2015 November | 61 | 46 | 107 |
| 2015 October | 78 | 63 | 141 |
| 2015 September | 115 | 91 | 206 |
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