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Journal of Medical Case Reports

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Open Access 01-12-2023 | Hemoptysis | Case report

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

Authors: Abate Yeshidinber Weldetsadik, Abdi Kebede, Binyam Gebremedhin Godu, Maru Gama

Published in: Journal of Medical Case Reports | Issue 1/2023

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.

Porres DV, Morenza OP, Pallisa E, Roque A, Andreu J, Martínez M. Learning from the pulmonary veins. Radiographics. 2013;33(4):999–1022. https://doi.org/10.1148/rg.334125043.CrossRefPubMed

Pourmoghadam KK, Moore JW, Khan M, et al. Congenital unilateral pulmonary venous atresia: definitive diagnosis and treatment. Pediatr Cardiol. 2003;24(1):73–9. https://doi.org/10.1007/s00246-002-0220-6.CrossRefPubMed

Tang C, Duan H, Zhou K, et al. Isolated unilateral pulmonary vein atresia with hemoptysis in a child: a case report and literature review. Medicine (Baltimore). 2018;97(34):e11882. https://doi.org/10.1097/MD.0000000000011882.CrossRefPubMed

Sacco O, Fregonese B, Fregonese L, Gambini C, Pongiglione G, Rossi GA. Recurrent unilateral bacterial pneumonias and interstitial fibrosis associated with pulmonary vein atresia: successful treatment with endovascular stent implantation. Pediatr Pulmonol. 2002;34(4):324–8. https://doi.org/10.1002/ppul.10158.CrossRefPubMed

Kozak MF, Kozak AC, Souza AS, Souza AS Jr. Left pulmonary vein atresia: the contribution of multislice computed tomography. Pediatr Cardiol. 2011;32:108–10.CrossRefPubMed

Cao M, Cai H, Ding J, Zhuang Y, Wang Z. Bronchial varices in congenital unilateral pulmonary vein atresia. Am J Respir Crit Care Med. 2013;187(11):1267–8. https://doi.org/10.1164/rccm.201211-1974IM.CrossRefPubMed

Tissot C, Corbelli R, Aggoun Y, Beghetti M. ED Cruz Bronchoscopic diagnosis of asymptomatic unilateral pulmonary vein atresia in an infant. Pediatr Cardiol. 2008;29:976–9.CrossRefPubMed

Kim Y, Yoo IR, Ahn MI, Han DH. Asymptomatic adults with isolated, unilateral right pulmonary vein atresia: multidetector CT findings. Br J Radiol. 2011;84(1002):e109–13. https://doi.org/10.1259/bjr/51344661.CrossRefPubMedPubMedCentral

Dillman JR, Yarram SG, Hernandez RJ. Imaging of pulmonary venous developmental anomalies. AJR Am J Roentgenol. 2009;192(5):1272–85. https://doi.org/10.2214/AJR.08.1526.CrossRefPubMed

10.

Latson LA, Prieto LR. Congenital and acquired pulmonary vein stenosis. Circulation. 2007;115:103–8.CrossRefPubMed

11.

Okamoto R, Miyazaki O, Aoki H, et al. Mediastinal and hilar soft tissue mass-like lesions in congenital unilateral pulmonary vein atresia: a retrospective review of seven pediatric patients. Pediatr Int. 2020;62(11):1234–40. https://doi.org/10.1111/ped.14307.CrossRefPubMed

12.

Chen Y, Xiao Y, Zhang Y, Zhu G, Dou R, Gu H. A 12-year-old girl with dyspnea and unilateral interlobular septal thickening. Chest. 2019;156(3):e57–61. https://doi.org/10.1016/j.chest.2019.03.022.CrossRefPubMed

13.

Har-Even Cohn R, Hicks M, Lacson A, Hicks A. Left hypoplastic lung and hemoptysis-rare familial unilateral pulmonary vein atresia. Clin Case Rep. 2020;8(9):1698–703. https://doi.org/10.1002/ccr3.2982.CrossRefPubMedPubMedCentral

14.

Levin TL, Betz BW, Gennarini LM, Wircberg C. Unilateral absence of the pulmonary veins: an unusual diagnosis with characteristic imaging findings. Clin Imaging. 2019;55:107–11. https://doi.org/10.1016/j.clinimag.2019.01.022.CrossRefPubMed

15.

Biradar B, Sharma A, Malhi AS, Kumar S. Unilateral pulmonary vein atresia: diagnostic dilemma unfolded on imaging. BMJ Case Rep. 2018. https://doi.org/10.1136/bcr-2017-224154.CrossRefPubMedPubMedCentral

Title: Unilateral pulmonary vein atresia presenting with recurrent hemoptysis and bronchial varices in an Ethiopian adolescent: a case report
Authors: Abate Yeshidinber Weldetsadik
Abdi Kebede
Binyam Gebremedhin Godu
Maru Gama
Publication date: 01-12-2023
Publisher: BioMed Central
Keywords: Hemoptysis
Hemoptysis
Pneumonectomy
Published in: Journal of Medical Case Reports / Issue 1/2023
Electronic ISSN: 1752-1947
DOI: https://doi.org/10.1186/s13256-023-03956-4

At a glance: The ONWARDS insulin icodec trials

Springer Medicine

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

Abstract

Background

Case presentation

Conclusion

At a glance: The ONWARDS insulin icodec trials

Springer Medicine

Abstract

Background

Case presentation

Conclusion

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