Letter to the Editor
Isolated Unilateral Pulmonary Vein Atresia in Adults
Atresia aislada unilateral de venas pulmonares en el adulto
M. Teresa Gómez Hernández
, María Rodríguez Pérez, Marcelo F. Jiménez
Corresponding author
Departamento de Cirugía Torácica, Hospital Universitario de Salamanca, Salamanca, Spain
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Arteriografía de aorta torácica que muestra arteria bronquial irregular y tortuosa con origen en cara anterior y superior de aorta torácica descendente (C).</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "M. Teresa Gómez Hernández, María Rodríguez Pérez, Marcelo F. Jiménez" "autores" => array:3 [ 0 => array:2 [ "nombre" => "M. Teresa" "apellidos" => "Gómez Hernández" ] 1 => array:2 [ "nombre" => "María" "apellidos" => "Rodríguez Pérez" ] 2 => array:2 [ "nombre" => "Marcelo" "apellidos" => "F. 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A Case Report and Review of the Literature" "tienePdf" => "en" "tieneTextoCompleto" => "en" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "423" "paginaFinal" => "424" ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Atresia congénita unilateral de venas pulmonares en adultos. Descripción de un caso y revisión de la literatura" ] ] "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" => 1021 "Ancho" => 995 "Tamanyo" => 129958 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">The CT scan shows absence of right pulmonary veins draining into the left atrium, as well as the inferior phrenic artery with origin in the right renal artery.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Juan Santos-Morano, Silvia Rodríguez-Hernández, Jesica Ivana Hilares-Vera" "autores" => array:3 [ 0 => array:2 [ "nombre" => "Juan" "apellidos" => "Santos-Morano" ] 1 => array:2 [ "nombre" => "Silvia" "apellidos" => "Rodríguez-Hernández" ] 2 => array:2 [ "nombre" => "Jesica Ivana" "apellidos" => "Hilares-Vera" ] ] ] ] ] "idiomaDefecto" => "en" "Traduccion" => array:1 [ "es" => array:9 [ "pii" => "S030028961400430X" "doi" => "10.1016/j.arbres.2014.10.002" "estado" => "S300" "subdocumento" => "" "abierto" => array:3 [ "ES" => true "ES2" => true "LATM" => true ] "gratuito" => true "lecturas" => array:1 [ "total" => 0 ] "idiomaDefecto" => "es" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S030028961400430X?idApp=UINPBA00003Z" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1579212915001755?idApp=UINPBA00003Z" "url" => "/15792129/0000005100000008/v2_201511200100/S1579212915001755/v2_201511200100/en/main.assets" ] "en" => array:16 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Letter to the Editor</span>" "titulo" => "Isolated Unilateral Pulmonary Vein Atresia in Adults" "tieneTextoCompleto" => true "saludo" => "To the Editor," "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "424" "paginaFinal" => "425" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "M. Teresa Gómez Hernández, María Rodríguez Pérez, Marcelo F. Jiménez" "autores" => array:3 [ 0 => array:4 [ "nombre" => "M. Teresa" "apellidos" => "Gómez Hernández" "email" => array:1 [ 0 => "mteresa.gomez.hernandez@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "María" "apellidos" => "Rodríguez Pérez" ] 2 => array:2 [ "nombre" => "Marcelo F." "apellidos" => "Jiménez" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Departamento de Cirugía Torácica, Hospital Universitario de Salamanca, Salamanca, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Atresia aislada unilateral de venas pulmonares en el adulto" ] ] "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" => 685 "Ancho" => 1950 "Tamanyo" => 249745 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Pulmonary arteriography showing hypoplasia of the right pulmonary artery, providing little perfusion, almost entirely limited to the upper lobe (A), in which right venous drainage cannot be seen (B). Arteriography of the thoracic aorta showing an irregular, winding bronchial artery with origin in the anterior superior side of the descending thoracic aorta (C).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">We present the case of a 19-year-old woman, smoker (8 pack-years), and regular cannabis user, with a history of severe bronchitis in childhood and frequent respiratory infections. She presented for episodes of coughing with hemoptoic expectoration since she was 15-years-old. The episodes were initially sporadic, but had become more frequent in recent months, sometimes occurring every 48<span class="elsevierStyleHsp" style=""></span>h. On examination, she had decreased breath sounds in the right hemithorax, and acropachy. The chest radiograph showed loss of volume in the right lung and attenuation of the right hilum. The right pulmonary veins could not be identified on computed tomography (CT), and a diffuse increase in density was observed in the affected lung. Cardiac magnetic resonance imaging (MRI) ruled out associated heart disease, and the echocardiography ruled out pulmonary hypertension. Pulmonary arteriography was performed (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A and B), in which hypoplasia of the right pulmonary artery and absence of right pulmonary venous return were observed. Arteriography of the thoracic artery (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>C) showed right bronchial artery hypertrophy originating in the descending aorta. Lung function test results were: FEV1: 2.040 (75.5%), FVC: 2.800 (90.1%) and DLCOc SB: 69.3%. Varices were observed in the distal trachea and start of the right main bronchus on fiber optic bronchoscopy. Considering the previous findings, we opted for surgical treatment, specifically, right pneumonectomy. The immediate post-operative period was uneventful, and the patient is currently asymptomatic.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">Unilateral pulmonary vein atresia is a rare condition that is associated with congenital cardiac malformations in 32% of cases.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> It is usually diagnosed in childhood and, if untreated, can be fatal in as many as 50% of cases.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">2</span></a> Recurrent lung infection and dyspnea on exertion are the most common manifestations. Rupture of bronchial varices can sometimes cause hemoptysis, and therefore bronchoscopic findings may be of interest when this condition is suspected. CT and MRI are useful diagnostic tools, although definitive diagnosis is provided by pulmonary arteriography. Arteriography of the thoracic aorta and selective arteriography of the bronchial arteries enable the origin and anatomical distribution of collateral branches to be identified. Pneumonectomy is considered the treatment of choice in cases that present with pulmonary hypertension, recurrent infections or significant hemoptysis. This eliminates the focus of the infections, the left-right shunt and the dead space that contributes to exercise intolerance.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a> Reparatory surgery can be considered in cases detected at a young age. Embolization of the bronchial artery has been described in an adult patient whose only symptom was hemoptysis.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a> Conservative treatment and follow-up may be the management of choice in asymptomatic patients without pulmonary hypertension.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a> In our case, repeated hemoptysis, the history of frequent respiratory infections and prevention of pulmonary hypertension prompted us to choose resection of the affected lung in this patient.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Funding</span><p id="par0015" class="elsevierStylePara elsevierViewall">None.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Conflict of Interests</span><p id="par0020" class="elsevierStylePara elsevierViewall">The authors state that they do not have any conflicts of interest directly or indirectly related with the contents of the manuscript.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:3 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Funding" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Conflict of Interests" ] 2 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Gómez Hernández MT, Rodríguez Pérez M, Jiménez MF. Atresia aislada unilateral de venas pulmonares en el adulto. Arch Bronconeumol. 2015;51:424–425.</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" => 685 "Ancho" => 1950 "Tamanyo" => 249745 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Pulmonary arteriography showing hypoplasia of the right pulmonary artery, providing little perfusion, almost entirely limited to the upper lobe (A), in which right venous drainage cannot be seen (B). Arteriography of the thoracic aorta showing an irregular, winding bronchial artery with origin in the anterior superior side of the descending thoracic aorta (C).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0030" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Congenital unilateral pulmonary venous atresia: definitive diagnosis and treatment" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "K.K. Pourmoghadam" 1 => "J.W. Moore" 2 => "M. Khan" 3 => "E.M. Geary" 4 => "N. Madan" 5 => "B.J. Wolfson" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1007/s00246-002-0220-6" "Revista" => array:6 [ "tituloSerie" => "Pediatr Cardiol" "fecha" => "2003" "volumen" => "24" "paginaInicial" => "73" "paginaFinal" => "79" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/12360396" "web" => "Medline" ] ] ] ] ] ] ] ] 1 => array:3 [ "identificador" => "bib0035" "etiqueta" => "2" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Isolated pulmonary vein atresia" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "S. Cullen" 1 => "P.F. Deasy" 2 => "E. Tempany" 3 => "D.F. Duff" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:6 [ "tituloSerie" => "Br Heart J" "fecha" => "1990" "volumen" => "63" "paginaInicial" => "350" "paginaFinal" => "354" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/2375897" "web" => "Medline" ] ] ] ] ] ] ] ] 2 => array:3 [ "identificador" => "bib0040" "etiqueta" => "3" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Unilateral pulmonary vein atresia: clinical and radiographic spectrum" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "L.B. Beerman" 1 => "K.S. Oh" 2 => "S.C. Park" 3 => "M.D. Freed" 4 => "H.M. Sondheimer" 5 => "F.J. Fricker" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1007/BF02076334" "Revista" => array:6 [ "tituloSerie" => "Pediatr Cardiol" "fecha" => "1983" "volumen" => "4" "paginaInicial" => "105" "paginaFinal" => "112" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/6878066" "web" => "Medline" ] ] ] ] ] ] ] ] 3 => array:3 [ "identificador" => "bib0045" "etiqueta" => "4" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Congenital unilateral pulmonary vein atresia: radiologic findings in three adult patients" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "L.E. Heyneman" 1 => "R.L. Nolan" 2 => "J.K. Harrison" 3 => "H.P. McAdams" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:5 [ "tituloSerie" => "Am J Roentgenol" "fecha" => "2001" "volumen" => "177" "paginaInicial" => "681" "paginaFinal" => "685" ] ] ] ] ] ] 4 => array:3 [ "identificador" => "bib0050" "etiqueta" => "5" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Right pulmonary venous atresia: a case report and review of literature" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "Ş. Savaş Bozbaş" 1 => "B. Varan" 2 => "Ş. Akçay" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:6 [ "tituloSerie" => "Tuberk Toraks" "fecha" => "2012" "volumen" => "60" "paginaInicial" => "254" "paginaFinal" => "257" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/23030751" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/15792129/0000005100000008/v2_201511200100/S1579212915001792/v2_201511200100/en/main.assets" "Apartado" => array:4 [ "identificador" => "45358" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Letters to the Editor" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/15792129/0000005100000008/v2_201511200100/S1579212915001792/v2_201511200100/en/main.pdf?idApp=UINPBA00003Z&text.app=https://archbronconeumol.org/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1579212915001792?idApp=UINPBA00003Z" ]
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