Unilateral pulmonary vein atresia presenting with recurrent hemoptysis and bronchial varices in an Ethiopian adolescent: a case report

Abstract Background Congenital unilateral pulmonary vein atresia is a rare anomaly resulting from failure of the pulmonary vein to incorporate in the left atrium. It is a very rare cause of recurrent respiratory infections and hemoptysis requiring a high index of suspicion for proper diagnosis and m...

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Main Authors: Abate Yeshidinber Weldetsadik, Abdi Kebede, Binyam Gebremedhin Godu, Maru Gama
Format: Article
Language:English
Published: BMC 2023-06-01
Series:Journal of Medical Case Reports
Subjects:
Online Access:https://doi.org/10.1186/s13256-023-03956-4
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author Abate Yeshidinber Weldetsadik
Abdi Kebede
Binyam Gebremedhin Godu
Maru Gama
author_facet Abate Yeshidinber Weldetsadik
Abdi Kebede
Binyam Gebremedhin Godu
Maru Gama
author_sort Abate Yeshidinber Weldetsadik
collection DOAJ
description Abstract Background Congenital unilateral pulmonary vein atresia is a rare anomaly resulting from failure of the pulmonary vein to incorporate in the left atrium. It is a very rare cause of recurrent respiratory infections and hemoptysis requiring a high index of suspicion for proper diagnosis and management in early childhood. Case presentation We report a 13-year old Anuac (Ethiopia, Region of Gambela) male adolescent with a delayed diagnosis of isolated atresia of the left pulmonary veins despite early childhood presentation with recurrent chest infections, hemoptysis and exercise intolerance. Contrast enhanced CT of thorax with reconstructed planes confirmed the diagnosis. He underwent pneumonectomy for severe and recurrent symptoms and did well on subsequent follow ups after 6 months of pneumonectomy. Conclusion Although a rare anomaly, congenital unilateral pulmonary vein atresia should be considered in the differential diagnosis of a child presenting with recurrent chest infections, exercise intolerance and hemoptysis to facilitate early appropriate diagnosis and treatment.
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spelling doaj.art-dc726331d9e44d66b9c0a358a1df6cf82023-06-04T11:29:45ZengBMCJournal of Medical Case Reports1752-19472023-06-011711510.1186/s13256-023-03956-4Unilateral pulmonary vein atresia presenting with recurrent hemoptysis and bronchial varices in an Ethiopian adolescent: a case reportAbate Yeshidinber Weldetsadik0Abdi Kebede1Binyam Gebremedhin Godu2Maru Gama3Department of Pediatrics and Child Health, Saint Paul’s Hospital Millennium Medical CollegeDepartment of Pediatrics and Child Health, Saint Paul’s Hospital Millennium Medical CollegePediatric Surgery Unit, Saint Paul’s Hospital Millennium Medical CollegeDepartment of Surgery, Pediatric Surgery Unit, Saint Paul’s Hospital Millennium Medical CollegeAbstract Background Congenital unilateral pulmonary vein atresia is a rare anomaly resulting from failure of the pulmonary vein to incorporate in the left atrium. It is a very rare cause of recurrent respiratory infections and hemoptysis requiring a high index of suspicion for proper diagnosis and management in early childhood. Case presentation We report a 13-year old Anuac (Ethiopia, Region of Gambela) male adolescent with a delayed diagnosis of isolated atresia of the left pulmonary veins despite early childhood presentation with recurrent chest infections, hemoptysis and exercise intolerance. Contrast enhanced CT of thorax with reconstructed planes confirmed the diagnosis. He underwent pneumonectomy for severe and recurrent symptoms and did well on subsequent follow ups after 6 months of pneumonectomy. Conclusion Although a rare anomaly, congenital unilateral pulmonary vein atresia should be considered in the differential diagnosis of a child presenting with recurrent chest infections, exercise intolerance and hemoptysis to facilitate early appropriate diagnosis and treatment.https://doi.org/10.1186/s13256-023-03956-4Pulmonary vein atresiaRecurrent hemoptysisChest infectionCase reportEthiopia
spellingShingle Abate Yeshidinber Weldetsadik
Abdi Kebede
Binyam Gebremedhin Godu
Maru Gama
Unilateral pulmonary vein atresia presenting with recurrent hemoptysis and bronchial varices in an Ethiopian adolescent: a case report
Journal of Medical Case Reports
Pulmonary vein atresia
Recurrent hemoptysis
Chest infection
Case report
Ethiopia
title Unilateral pulmonary vein atresia presenting with recurrent hemoptysis and bronchial varices in an Ethiopian adolescent: a case report
title_full Unilateral pulmonary vein atresia presenting with recurrent hemoptysis and bronchial varices in an Ethiopian adolescent: a case report
title_fullStr Unilateral pulmonary vein atresia presenting with recurrent hemoptysis and bronchial varices in an Ethiopian adolescent: a case report
title_full_unstemmed Unilateral pulmonary vein atresia presenting with recurrent hemoptysis and bronchial varices in an Ethiopian adolescent: a case report
title_short Unilateral pulmonary vein atresia presenting with recurrent hemoptysis and bronchial varices in an Ethiopian adolescent: a case report
title_sort unilateral pulmonary vein atresia presenting with recurrent hemoptysis and bronchial varices in an ethiopian adolescent a case report
topic Pulmonary vein atresia
Recurrent hemoptysis
Chest infection
Case report
Ethiopia
url https://doi.org/10.1186/s13256-023-03956-4
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