Congenital diaphragmatic hernia associated with esophageal atresia, tracheoesophageal fistula and total anomalous pulmonary venous connection in a premature twin newborn

Introduction. Congenital diaphragmatic hernia (CDH) with concomitant esophageal atresia (EA) and tracheo-esophageal fistula (TEF) is a very rare condition, with a high mortality rate. Prematurity and congenital heart anomalies additionally increase the mortality rate. This situation is a great chall...

Full description

Saved in:
Bibliographic Details
Main Authors: Savić Đorđe, Grujić Blagoje, Stanković Nikola, Miličković Maja, Stanković Zoran, Kojović Vladimir
Format: Article
Language:English
Published: Ministry of Defence of the Republic of Serbia, University of Defence, Belgrade 2019-01-01
Series:Vojnosanitetski Pregled
Subjects:
Online Access:http://www.doiserbia.nb.rs/img/doi/0042-8450/2019/0042-84501700135S.pdf
Tags: Add Tag
No Tags, Be the first to tag this record!
_version_ 1850110412931989504
author Savić Đorđe
Grujić Blagoje
Stanković Nikola
Miličković Maja
Stanković Zoran
Kojović Vladimir
author_facet Savić Đorđe
Grujić Blagoje
Stanković Nikola
Miličković Maja
Stanković Zoran
Kojović Vladimir
author_sort Savić Đorđe
collection DOAJ
description Introduction. Congenital diaphragmatic hernia (CDH) with concomitant esophageal atresia (EA) and tracheo-esophageal fistula (TEF) is a very rare condition, with a high mortality rate. Prematurity and congenital heart anomalies additionally increase the mortality rate. This situation is a great challenge for clinicians, requiring multidisciplinary work and adequate treatment strategy. Case report. We presented a premature twin newborn at the gestational age of 33/34 weeks with body mass of 1690 g. The existence of the left CDH was established on prenatal ultrasound exam in the 24th gestational week, and the diagnosis of EA with TEF was made on admittance to our hospital. The cardiac ultrasound exam revealed the total anomalous pulmonary venous connection (TAPVC). The first operation was performed on the day of admittance and consisted of left subcostal laparotomy, diaphragmatic repair, elastic occlusion of the gastroesophageal junction and gastrostomy. The ligation of TEF and esophagoplasty were done 13 days later in the second operation. The lethal outcome during the esophagoplasty was caused by the crisis of pulmonary hypertension and associated congenital heart anomaly (TAPVC). The presence of CDH and EA/TEF in association with TAPVC in a twin newborn has not been reported before in the literature. Conclusion. The treatment of newborns with CDH and EA/TEF requires multidisciplinary well-coordinated team work of pediatric surgeons, anaesthesiologists, neonatologists and pulmologists. The standard protocol for the management does not exist, but well-planned staged operations could enable greater survival rate.
format Article
id doaj-art-46c63b87629c4cacb7ef8362fea0bfab
institution OA Journals
issn 0042-8450
2406-0720
language English
publishDate 2019-01-01
publisher Ministry of Defence of the Republic of Serbia, University of Defence, Belgrade
record_format Article
series Vojnosanitetski Pregled
spelling doaj-art-46c63b87629c4cacb7ef8362fea0bfab2025-08-20T02:37:50ZengMinistry of Defence of the Republic of Serbia, University of Defence, BelgradeVojnosanitetski Pregled0042-84502406-07202019-01-0176774574810.2298/VSP170322135S0042-84501700135SCongenital diaphragmatic hernia associated with esophageal atresia, tracheoesophageal fistula and total anomalous pulmonary venous connection in a premature twin newbornSavić Đorđe0Grujić Blagoje1Stanković Nikola2Miličković Maja3Stanković Zoran4Kojović Vladimir5Mother And Child Health Care Institute of Serbia „Dr Vukan Čupić“, Belgrade, Serbia + University of Belgrade, Faculty of Medicine, Belgrade, SerbiaMother And Child Health Care Institute of Serbia „Dr Vukan Čupić“, Belgrade, Serbia + University of Belgrade, Faculty of Medicine, Belgrade, SerbiaMother And Child Health Care Institute of Serbia „Dr Vukan Čupić“, Belgrade, SerbiaMother And Child Health Care Institute of Serbia „Dr Vukan Čupić“, Belgrade, Serbia + University of Belgrade, Faculty of Medicine, Belgrade, SerbiaMother And Child Health Care Institute of Serbia „Dr Vukan Čupić“, Belgrade, SerbiaMother And Child Health Care Institute of Serbia „Dr Vukan Čupić“, Belgrade, Serbia + University of Belgrade, Faculty of Medicine, Belgrade, SerbiaIntroduction. Congenital diaphragmatic hernia (CDH) with concomitant esophageal atresia (EA) and tracheo-esophageal fistula (TEF) is a very rare condition, with a high mortality rate. Prematurity and congenital heart anomalies additionally increase the mortality rate. This situation is a great challenge for clinicians, requiring multidisciplinary work and adequate treatment strategy. Case report. We presented a premature twin newborn at the gestational age of 33/34 weeks with body mass of 1690 g. The existence of the left CDH was established on prenatal ultrasound exam in the 24th gestational week, and the diagnosis of EA with TEF was made on admittance to our hospital. The cardiac ultrasound exam revealed the total anomalous pulmonary venous connection (TAPVC). The first operation was performed on the day of admittance and consisted of left subcostal laparotomy, diaphragmatic repair, elastic occlusion of the gastroesophageal junction and gastrostomy. The ligation of TEF and esophagoplasty were done 13 days later in the second operation. The lethal outcome during the esophagoplasty was caused by the crisis of pulmonary hypertension and associated congenital heart anomaly (TAPVC). The presence of CDH and EA/TEF in association with TAPVC in a twin newborn has not been reported before in the literature. Conclusion. The treatment of newborns with CDH and EA/TEF requires multidisciplinary well-coordinated team work of pediatric surgeons, anaesthesiologists, neonatologists and pulmologists. The standard protocol for the management does not exist, but well-planned staged operations could enable greater survival rate.http://www.doiserbia.nb.rs/img/doi/0042-8450/2019/0042-84501700135S.pdfinfant, prematurecongenital abnormalitieshernia, diaphragmatic esophageal atresiatracheoesophageal fistulaheart defects, congenitaldigestive system surgical procedures
spellingShingle Savić Đorđe
Grujić Blagoje
Stanković Nikola
Miličković Maja
Stanković Zoran
Kojović Vladimir
Congenital diaphragmatic hernia associated with esophageal atresia, tracheoesophageal fistula and total anomalous pulmonary venous connection in a premature twin newborn
Vojnosanitetski Pregled
infant, premature
congenital abnormalities
hernia, diaphragmatic esophageal atresia
tracheoesophageal fistula
heart defects, congenital
digestive system surgical procedures
title Congenital diaphragmatic hernia associated with esophageal atresia, tracheoesophageal fistula and total anomalous pulmonary venous connection in a premature twin newborn
title_full Congenital diaphragmatic hernia associated with esophageal atresia, tracheoesophageal fistula and total anomalous pulmonary venous connection in a premature twin newborn
title_fullStr Congenital diaphragmatic hernia associated with esophageal atresia, tracheoesophageal fistula and total anomalous pulmonary venous connection in a premature twin newborn
title_full_unstemmed Congenital diaphragmatic hernia associated with esophageal atresia, tracheoesophageal fistula and total anomalous pulmonary venous connection in a premature twin newborn
title_short Congenital diaphragmatic hernia associated with esophageal atresia, tracheoesophageal fistula and total anomalous pulmonary venous connection in a premature twin newborn
title_sort congenital diaphragmatic hernia associated with esophageal atresia tracheoesophageal fistula and total anomalous pulmonary venous connection in a premature twin newborn
topic infant, premature
congenital abnormalities
hernia, diaphragmatic esophageal atresia
tracheoesophageal fistula
heart defects, congenital
digestive system surgical procedures
url http://www.doiserbia.nb.rs/img/doi/0042-8450/2019/0042-84501700135S.pdf
work_keys_str_mv AT savicđorđe congenitaldiaphragmaticherniaassociatedwithesophagealatresiatracheoesophagealfistulaandtotalanomalouspulmonaryvenousconnectioninaprematuretwinnewborn
AT grujicblagoje congenitaldiaphragmaticherniaassociatedwithesophagealatresiatracheoesophagealfistulaandtotalanomalouspulmonaryvenousconnectioninaprematuretwinnewborn
AT stankovicnikola congenitaldiaphragmaticherniaassociatedwithesophagealatresiatracheoesophagealfistulaandtotalanomalouspulmonaryvenousconnectioninaprematuretwinnewborn
AT milickovicmaja congenitaldiaphragmaticherniaassociatedwithesophagealatresiatracheoesophagealfistulaandtotalanomalouspulmonaryvenousconnectioninaprematuretwinnewborn
AT stankoviczoran congenitaldiaphragmaticherniaassociatedwithesophagealatresiatracheoesophagealfistulaandtotalanomalouspulmonaryvenousconnectioninaprematuretwinnewborn
AT kojovicvladimir congenitaldiaphragmaticherniaassociatedwithesophagealatresiatracheoesophagealfistulaandtotalanomalouspulmonaryvenousconnectioninaprematuretwinnewborn