Scientific Letter
Isolated right pulmonary artery agenesis
Agenesia aislada de la arteria pulmonar derecha
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Nótese la marcada disminución del tamaño pulmonar derecho y la desviación mediastínica hacia ese mismo lado, así como la hiperinsuflación pulmonar izquierda con herniación del lóbulo superior.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Víctor Adán Lanceta, Ainhoa Jiménez Olmos, Carlos Martín de Vicente, Juan Pablo García Íñiguez" "autores" => array:4 [ 0 => array:2 [ "nombre" => "Víctor" "apellidos" => "Adán Lanceta" ] 1 => array:2 [ "nombre" => "Ainhoa" "apellidos" => "Jiménez Olmos" ] 2 => array:2 [ "nombre" => "Carlos" "apellidos" => "Martín de Vicente" ] 3 => array:2 [ "nombre" => "Juan Pablo" "apellidos" => "García Íñiguez" ] ] ] ] ] "idiomaDefecto" => "es" "Traduccion" => array:1 [ "en" => array:9 [ "pii" => "S2341287916301466" "doi" => "10.1016/j.anpede.2016.04.004" "estado" => "S300" "subdocumento" => "" "abierto" => array:3 [ "ES" => false "ES2" => false "LATM" => false ] "gratuito" => false "lecturas" => array:1 [ "total" => 0 ] "idiomaDefecto" => 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(B) Chest helical CT with 3D image reconstruction. (B.1) Detail of right pulmonary artery agenesis. (B.2) Collateral vessels originating in the descending aorta that supply the right lung parenchyma. (B.3) Right pulmonary hypoplasia.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Unilateral pulmonary artery agenesis (UPAA) is a rare malformation that may present in isolation or being associated with cardiovascular malformations such as tetralogy of Fallot, transposition of the great vessels, septal defects and aortic arch malformations.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a> Its prevalence in the absence of congenital cardiovascular malformations is of one in 200<span class="elsevierStyleHsp" style=""></span>000–300<span class="elsevierStyleHsp" style=""></span>000 individuals, and it is usually underdiagnosed in the paediatric age group.</p><p id="par0010" class="elsevierStylePara elsevierViewall">We present the case of a male infant aged 4 months with no relevant medical history and normal prenatal ultrasound findings that was admitted for acute bronchiolitis caused by human parainfluenza virus type 3, with evidence of lung contraction in the right hemithorax and increased density in the anterior segment of the upper right lobe on chest X-ray. The patient was admitted two more times, at 5 and 8 months, due to obstructive bronchiolitis manifesting with cough, hypoxaemia, wheezing and laboured breathing. In the second hospitalization, respiratory syncytial virus was isolated from a nasopharyngeal aspirate sample. The chest X-rays performed during these episodes showed progressive compression of the right hemithorax with ipsilateral mediastinal shift and hyperinflation of the left lung with herniation of the left upper lobe towards the right lung (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). These findings led to the performance of a computed tomography (TC) scan with contrast, which revealed the absence of the proximal section of the right pulmonary artery with mild hypoplasia of the ipsilateral lung and the presence of two collateral vessels originating in the descending aorta (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>) and supplying the right lung, with a tortuous trajectory and extensive branching. The findings were compatible with right UPAA. The cardiologic workup was completed with an echocardiographic examination that ruled out the presence of other congenital malformations. The clinical manifestations of the patient included recurrent episodes of obstructive bronchitis of mild to moderate severity with absence of symptoms between episodes and normal findings in physical examination. The patient's growth and development have been normal and he is not receiving specific treatment for this condition due to the absence of complications and severe symptoms until present.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Unilateral pulmonary artery agenesis results from the intrauterine involution of the proximal sixth aortic arch, which leads to the absence of the proximal pulmonary artery. The foetal development of the lungs and the associated bronchovascular anatomy are usually normal, since the hilar and distal pulmonary arteries and their branches are supplied by an ipsilateral patent ductus arteriosus, although they are not connected to the pulmonary trunk. After birth, closure of the ductus arteriosus leads to a reduction in blood flow that in turn results in pulmonary hypoplasia and the development of aortopulmonary collateral vessels.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> We believe that these are the reasons why no anomalies were detected in our patient in prenatal ultrasounds and pulmonary and vascular manifestations developed after birth.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Patients with isolated UPAA may be asymptomatic, with the disease being detected by chance when chest radiography is performed. Other patients may present with respiratory distress and heart failure, recurrent respiratory disease, exercise limitation, haemoptysis and/or pulmonary hypertension (PHTN).<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a> In infants aged less than one year, there is a predominance of respiratory distress symptoms (56%) associated with PHTN with subsequent heart failure. After one year of age, children may present with recurrent respiratory infections (23–37%) and/or haemoptysis (14–20%) due to increased aortopulmonary collateral circulation. In adulthood, UPAA is also associated with exercise limitation (18–45%). Physical examination findings may be normal, and abnormal findings on auscultation may include detection of a heart murmur and/or hypoventilation with or without abnormal lung sounds in the hypoplastic hemithorax.</p><p id="par0025" class="elsevierStylePara elsevierViewall">The final diagnosis is based on imaging tests.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">4</span></a> Chest X-rays show an ipsilateral contracted lung with ipsilateral mediastinal shift and hyperinflation of the contralateral lung that may be accompanied by herniation towards the affected side.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">5</span></a> The diagnosis is confirmed by chest CT and/or MRI with contrast, and the diagnostic workup is completed with echocardiography.</p><p id="par0030" class="elsevierStylePara elsevierViewall">At present, the most widely accepted approach to the management of isolated UPAA is conservative treatment of asymptomatic forms and surgical intervention in cases of severe disease or with severe complications.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">4</span></a> The first-line approach in the surgical treatment of UPAA is two-stage repair with stenting of the ductus arteriosus followed by surgical anastomosis. Some studies report the use of surgery even in asymptomatic cases, but surgical intervention in these patients is very controversial because there is a high probability of reintervention due to postoperative complications such as thrombosis of primary surgical anastomosis.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">6</span></a> The associated PHTN can be treated with antihypertensive drugs, and haemoptysis can be treated by collateral artery embolization. Lobectomy or pneumonectomy can be considered in cases with haemoptysis that does not respond to embolization or with recurrent respiratory infections that result in complications such as bronchiectasis.</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: Adán Lanceta V, Jiménez Olmos A, Martín de Vicente C, García Íñiguez JP. Agenesia aislada de la arteria pulmonar derecha. An Pediatr (Barc). 2017;86:45–46.</p>" ] ] "multimedia" => array:2 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 567 "Ancho" => 1605 "Tamanyo" => 84553 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Chest X-rays at ages 4 months (A), 5 months (B) and 8 months (C). The major findings are the markedly reduced size of the right lung and the ipsilateral mediastinal shift, and left lung hyperinflation with herniation of the left upper lobe.</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 732 "Ancho" => 1754 "Tamanyo" => 153735 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">(A) Chest helical CT with contrast revealing right pulmonary artery agenesis and right pulmonary hypoplasia. (B) Chest helical CT with 3D image reconstruction. (B.1) Detail of right pulmonary artery agenesis. (B.2) Collateral vessels originating in the descending aorta that supply the right lung parenchyma. (B.3) Right pulmonary hypoplasia.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:6 [ 0 => array:3 [ "identificador" => "bib0035" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Unilateral absence of pulmonary artery in children: bronchovascular anatomy, natural course and effect of treatment on lung growth" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "M. Alison" 1 => "L. Garel" 2 => "J.L. Bigras" 3 => "J. Déry" 4 => "C. 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Dahdah" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1007/s00246-010-9689-6" "Revista" => array:6 [ "tituloSerie" => "Pediatr Cardiol" "fecha" => "2010" "volumen" => "31" "paginaInicial" => "607" "paginaFinal" => "610" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/20300746" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/23412879/0000008600000001/v1_201612270102/S2341287916301466/v1_201612270102/en/main.assets" "Apartado" => array:4 [ "identificador" => "38181" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Scientific letters" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/23412879/0000008600000001/v1_201612270102/S2341287916301466/v1_201612270102/en/main.pdf?idApp=UINPBA00005H&text.app=https://analesdepediatria.org/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2341287916301466?idApp=UINPBA00005H" ]
| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 20 | 9 | 29 |
| 2024 October | 108 | 52 | 160 |
| 2024 September | 79 | 28 | 107 |
| 2024 August | 108 | 58 | 166 |
| 2024 July | 84 | 38 | 122 |
| 2024 June | 81 | 23 | 104 |
| 2024 May | 78 | 34 | 112 |
| 2024 April | 62 | 38 | 100 |
| 2024 March | 66 | 30 | 96 |
| 2024 February | 59 | 34 | 93 |
| 2024 January | 94 | 27 | 121 |
| 2023 December | 107 | 19 | 126 |
| 2023 November | 68 | 28 | 96 |
| 2023 October | 51 | 26 | 77 |
| 2023 September | 55 | 21 | 76 |
| 2023 August | 63 | 14 | 77 |
| 2023 July | 71 | 29 | 100 |
| 2023 June | 93 | 27 | 120 |
| 2023 May | 59 | 18 | 77 |
| 2023 April | 40 | 20 | 60 |
| 2023 March | 82 | 21 | 103 |
| 2023 February | 64 | 11 | 75 |
| 2023 January | 67 | 22 | 89 |
| 2022 December | 86 | 36 | 122 |
| 2022 November | 85 | 27 | 112 |
| 2022 October | 85 | 40 | 125 |
| 2022 September | 79 | 40 | 119 |
| 2022 August | 79 | 58 | 137 |
| 2022 July | 45 | 45 | 90 |
| 2022 June | 48 | 28 | 76 |
| 2022 May | 52 | 43 | 95 |
| 2022 April | 55 | 30 | 85 |
| 2022 March | 98 | 55 | 153 |
| 2022 February | 58 | 26 | 84 |
| 2022 January | 88 | 45 | 133 |
| 2021 December | 58 | 47 | 105 |
| 2021 November | 60 | 55 | 115 |
| 2021 October | 94 | 80 | 174 |
| 2021 September | 51 | 44 | 95 |
| 2021 August | 52 | 47 | 99 |
| 2021 July | 60 | 46 | 106 |
| 2021 June | 46 | 45 | 91 |
| 2021 May | 54 | 46 | 100 |
| 2021 April | 126 | 44 | 170 |
| 2021 March | 82 | 43 | 125 |
| 2021 February | 49 | 17 | 66 |
| 2021 January | 48 | 32 | 80 |
| 2020 December | 58 | 33 | 91 |
| 2020 November | 53 | 14 | 67 |
| 2020 October | 56 | 24 | 80 |
| 2020 September | 53 | 30 | 83 |
| 2020 August | 58 | 15 | 73 |
| 2020 July | 47 | 23 | 70 |
| 2020 June | 45 | 14 | 59 |
| 2020 May | 53 | 17 | 70 |
| 2020 April | 27 | 16 | 43 |
| 2020 March | 77 | 20 | 97 |
| 2020 February | 36 | 15 | 51 |
| 2020 January | 24 | 17 | 41 |
| 2019 December | 50 | 22 | 72 |
| 2019 November | 22 | 11 | 33 |
| 2019 October | 28 | 8 | 36 |
| 2019 September | 36 | 11 | 47 |
| 2019 August | 40 | 31 | 71 |
| 2019 July | 40 | 16 | 56 |
| 2019 June | 43 | 22 | 65 |
| 2019 May | 61 | 21 | 82 |
| 2019 April | 59 | 19 | 78 |
| 2019 March | 39 | 15 | 54 |
| 2019 February | 39 | 17 | 56 |
| 2019 January | 21 | 12 | 33 |
| 2018 December | 42 | 29 | 71 |
| 2018 November | 70 | 22 | 92 |
| 2018 October | 91 | 31 | 122 |
| 2018 September | 27 | 23 | 50 |
| 2018 August | 8 | 0 | 8 |
| 2018 July | 2 | 0 | 2 |
| 2018 June | 2 | 0 | 2 |
| 2018 May | 6 | 0 | 6 |
| 2018 April | 29 | 0 | 29 |
| 2018 March | 20 | 0 | 20 |
| 2018 February | 13 | 0 | 13 |
| 2018 January | 33 | 0 | 33 |
| 2017 December | 20 | 0 | 20 |
| 2017 November | 23 | 0 | 23 |
| 2017 October | 15 | 0 | 15 |
| 2017 September | 17 | 0 | 17 |
| 2017 August | 7 | 0 | 7 |
| 2017 July | 22 | 1 | 23 |
| 2017 June | 22 | 14 | 36 |
| 2017 May | 25 | 10 | 35 |
| 2017 April | 95 | 71 | 166 |
| 2017 March | 15 | 13 | 28 |
| 2017 February | 12 | 3 | 15 |
| 2017 January | 26 | 19 | 45 |
| 2016 December | 3 | 8 | 11 |
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