Neonatal repair of total anomalous pulmonary venous connection accompanied by unilateral lung agenesis and Goldenhar syndrome: a case report

Abstract Background Total anomalous pulmonary venous connection accompanied by unilateral lung agenesis and Goldenhar syndrome is extremely rare. Case presentation We present a case of total anomalous pulmonary venous connection accompanied by unilateral lung agenesis and Goldenhar syndrome in a pat...

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Autores principales:	Takahiro Ito, Ikuo Hagino, Mitsuru Aoki, Kentaro Umezu, Tomohiro Saito, Akiyo Suzuki
Formato:	article
Lenguaje:	EN
Publicado:	BMC 2021
Materias:	Total anomalous pulmonary venous connection Goldenhar syndrome Unilateral lung agenesis Surgery RD1-811 Anesthesiology RD78.3-87.3
Acceso en línea:	https://doaj.org/article/dc6ddc5cbef84eb8baaf13b3ee11c84d
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spelling	oai:doaj.org-article:dc6ddc5cbef84eb8baaf13b3ee11c84d2021-11-28T12:30:54ZNeonatal repair of total anomalous pulmonary venous connection accompanied by unilateral lung agenesis and Goldenhar syndrome: a case report10.1186/s13019-021-01722-51749-8090https://doaj.org/article/dc6ddc5cbef84eb8baaf13b3ee11c84d2021-11-01T00:00:00Zhttps://doi.org/10.1186/s13019-021-01722-5https://doaj.org/toc/1749-8090Abstract Background Total anomalous pulmonary venous connection accompanied by unilateral lung agenesis and Goldenhar syndrome is extremely rare. Case presentation 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 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.Takahiro ItoIkuo HaginoMitsuru AokiKentaro UmezuTomohiro SaitoAkiyo SuzukiBMCarticleTotal anomalous pulmonary venous connectionGoldenhar syndromeUnilateral lung agenesisSurgeryRD1-811AnesthesiologyRD78.3-87.3ENJournal of Cardiothoracic Surgery, Vol 16, Iss 1, Pp 1-5 (2021)
institution	DOAJ
collection	DOAJ
language	EN
topic	Total anomalous pulmonary venous connection Goldenhar syndrome Unilateral lung agenesis Surgery RD1-811 Anesthesiology RD78.3-87.3
spellingShingle	Total anomalous pulmonary venous connection Goldenhar syndrome Unilateral lung agenesis Surgery RD1-811 Anesthesiology RD78.3-87.3 Takahiro Ito Ikuo Hagino Mitsuru Aoki Kentaro Umezu Tomohiro Saito Akiyo Suzuki Neonatal repair of total anomalous pulmonary venous connection accompanied by unilateral lung agenesis and Goldenhar syndrome: a case report
description	Abstract Background Total anomalous pulmonary venous connection accompanied by unilateral lung agenesis and Goldenhar syndrome is extremely rare. Case presentation 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 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.
format	article
author	Takahiro Ito Ikuo Hagino Mitsuru Aoki Kentaro Umezu Tomohiro Saito Akiyo Suzuki
author_facet	Takahiro Ito Ikuo Hagino Mitsuru Aoki Kentaro Umezu Tomohiro Saito Akiyo Suzuki
author_sort	Takahiro Ito
title	Neonatal repair of total anomalous pulmonary venous connection accompanied by unilateral lung agenesis and Goldenhar syndrome: a case report
title_short	Neonatal repair of total anomalous pulmonary venous connection accompanied by unilateral lung agenesis and Goldenhar syndrome: a case report
title_full	Neonatal repair of total anomalous pulmonary venous connection accompanied by unilateral lung agenesis and Goldenhar syndrome: a case report
title_fullStr	Neonatal repair of total anomalous pulmonary venous connection accompanied by unilateral lung agenesis and Goldenhar syndrome: a case report
title_full_unstemmed	Neonatal repair of total anomalous pulmonary venous connection accompanied by unilateral lung agenesis and Goldenhar syndrome: a case report
title_sort	neonatal repair of total anomalous pulmonary venous connection accompanied by unilateral lung agenesis and goldenhar syndrome: a case report
publisher	BMC
publishDate	2021
url	https://doaj.org/article/dc6ddc5cbef84eb8baaf13b3ee11c84d
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Neonatal repair of total anomalous pulmonary venous connection accompanied by unilateral lung agenesis and Goldenhar syndrome: a case report

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