Neonatal repair of total anomalous pulmonary venous connection accompanied by unilateral lung agenesis and Goldenhar syndrome: a case report.
Goldenhar syndrome
Total anomalous pulmonary venous connection
Unilateral lung agenesis
Journal
Journal of cardiothoracic surgery
ISSN: 1749-8090
Titre abrégé: J Cardiothorac Surg
Pays: England
ID NLM: 101265113
Informations de publication
Date de publication:
21 Nov 2021
21 Nov 2021
Historique:
received:
24
05
2021
accepted:
08
11
2021
entrez:
22
11
2021
pubmed:
23
11
2021
medline:
24
11
2021
Statut:
epublish
Résumé
Total anomalous pulmonary venous connection accompanied by unilateral lung agenesis and Goldenhar syndrome is extremely rare. We present a case of total anomalous pulmonary venous connection accompanied by unilateral lung agenesis and Goldenhar syndrome in a patient who was diagnosed based on transthoracic echocardiography and computed tomography. We observed complete absence of the lung, the bronchial tree, and vascular structures on the right side, with abnormal drainage of the left pulmonary veins into the innominate vein. The patient showed clear clinical evidence of pulmonary venous obstruction and underwent surgery 3 days after birth. The pulmonary venous chamber containing the vertical vein was anastomosed to the left atrium using 7-0 PDS running sutures via a median sternotomy. Echocardiography and computed tomography performed 1 year postoperatively revealed no pulmonary venous obstruction. We report a rare case of total anomalous pulmonary venous connection accompanied by unilateral lung agenesis and Goldenhar syndrome, which was successfully repaired 3 days after birth. A median sternotomy is a safe and effective approach for surgical repair of congenital heart disease with unilateral lung agenesis. Repair of the supra cardiac total anomalous pulmonary connection using the vertical vein is feasible in patients with a small pulmonary venous chamber.
Sections du résumé
BACKGROUND
BACKGROUND
Total anomalous pulmonary venous connection accompanied by unilateral lung agenesis and Goldenhar syndrome is extremely rare.
CASE PRESENTATION
METHODS
We present a case of total anomalous pulmonary venous connection accompanied by unilateral lung agenesis and Goldenhar syndrome in a patient who was diagnosed based on transthoracic echocardiography and computed tomography. We observed complete absence of the lung, the bronchial tree, and vascular structures on the right side, with abnormal drainage of the left pulmonary veins into the innominate vein. The patient showed clear clinical evidence of pulmonary venous obstruction and underwent surgery 3 days after birth. The pulmonary venous chamber containing the vertical vein was anastomosed to the left atrium using 7-0 PDS running sutures via a median sternotomy. Echocardiography and computed tomography performed 1 year postoperatively revealed no pulmonary venous obstruction.
CONCLUSION
CONCLUSIONS
We report a rare case of total anomalous pulmonary venous connection accompanied by unilateral lung agenesis and Goldenhar syndrome, which was successfully repaired 3 days after birth. A median sternotomy is a safe and effective approach for surgical repair of congenital heart disease with unilateral lung agenesis. Repair of the supra cardiac total anomalous pulmonary connection using the vertical vein is feasible in patients with a small pulmonary venous chamber.
Identifiants
pubmed: 34802434
doi: 10.1186/s13019-021-01722-5
pii: 10.1186/s13019-021-01722-5
pmc: PMC8607644
doi:
Types de publication
Case Reports
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Pagination
337Informations de copyright
© 2021. The Author(s).
Références
J Cardiothorac Surg. 2019 Feb 8;14(1):34
pubmed: 30736816
Asian Cardiovasc Thorac Ann. 2015 Jul;23(6):716-8
pubmed: 24585319
Ann Card Anaesth. 2017 Jan;20(Supplement):S61-S66
pubmed: 28074825
Interact Cardiovasc Thorac Surg. 2007 Feb;6(1):126-7
pubmed: 17669789
Ann Thorac Surg. 2014 May;97(5):1652-8
pubmed: 24507772
Cardiol Young. 2012 Jun;22(3):344-5
pubmed: 22309948