Arimatias Raitio1,2, Adeline Salim1, Paul D Losty3,4. 1. Department of Paediatric Surgery, University of Liverpool, Alder Hey Children's Hospital NHS Foundation Trust, Eaton Road, Liverpool, L12 2AP, UK. 2. Department of Paediatric Surgery, University of Turku and Turku University Hospital, Turku, Finland. 3. Department of Paediatric Surgery, University of Liverpool, Alder Hey Children's Hospital NHS Foundation Trust, Eaton Road, Liverpool, L12 2AP, UK. Paul.Losty@liv.ac.uk. 4. Institute of Child Health, University of Liverpool, Liverpool, UK. Paul.Losty@liv.ac.uk.
Abstract
Early reports have suggested survival benefits associated with a hernia sac in congenital diaphragmatic hernia (CDH). However, these studies have included only small subsets of patients. This systematic review aimed to evaluate differences in outcomes of CDH newborns with and without a hernia sac. PubMed and Embase databases were searched using relevant key terms. Papers were independently reviewed by two authors with final selection approved by the senior author. Original search retrieved 537 papers; the final review included 8 studies (n = 837 patients). There were 168 CDH patients (20%) with a hernia sac with an overall survival of 93% vs 73% for CDH newborns without a sac (p < 0.001). Twenty-three percent of patients with a CDH sac required diaphragm patch repair vs 44% patients without a sac (p < 0.001). Pulmonary hypertension was manifested in 44% of CDH babies with a hernia sac vs 64% without a sac (p < 0.001). Three studies compared ECMO requirement: 15% with a hernia sac and 34% without sac, p < 0.001. Conclusion: This study shows significant survival benefits in newborns associated with presence of a CDH sac. This may be likely related to these infants having more favourable physiology with less severe pulmonary hypertension and/or smaller anatomical defects requiring primary closure only. What is Known: • Early reports have suggested survival benefits associated with a hernia sac in CDH. • Previous studies have included only a small number of patients. What is New: • A systematic review of published studies clearly shows that CDH newborns with a hernia sac have better overall survival outcomes and less severe pulmonary hypertension. • ECMO utilization and patch repair were also less often required in newborns with a hernia sac.
Early reports have suggested survival benefits associated with a hernia sac in congenital diaphragmatic hernia (CDH). However, these studies have included only small subsets of patients. This systematic review aimed to evaluate differences in outcomes of CDH newborns with and without a hernia sac. PubMed and Embase databases were searched using relevant key terms. Papers were independently reviewed by two authors with final selection approved by the senior author. Original search retrieved 537 papers; the final review included 8 studies (n = 837 patients). There were 168 CDH patients (20%) with a hernia sac with an overall survival of 93% vs 73% for CDH newborns without a sac (p < 0.001). Twenty-three percent of patients with a CDH sac required diaphragm patch repair vs 44% patients without a sac (p < 0.001). Pulmonary hypertension was manifested in 44% of CDH babies with a hernia sac vs 64% without a sac (p < 0.001). Three studies compared ECMO requirement: 15% with a hernia sac and 34% without sac, p < 0.001. Conclusion: This study shows significant survival benefits in newborns associated with presence of a CDH sac. This may be likely related to these infants having more favourable physiology with less severe pulmonary hypertension and/or smaller anatomical defects requiring primary closure only. What is Known: • Early reports have suggested survival benefits associated with a hernia sac in CDH. • Previous studies have included only a small number of patients. What is New: • A systematic review of published studies clearly shows that CDH newborns with a hernia sac have better overall survival outcomes and less severe pulmonary hypertension. • ECMO utilization and patch repair were also less often required in newborns with a hernia sac.
Authors: E Spaggiari; J Stirnemann; J-P Bernard; L De Saint Blanquat; S Beaudoin; Y Ville Journal: Ultrasound Obstet Gynecol Date: 2013-03 Impact factor: 7.299
Authors: Edward R Oliver; Suzanne E DeBari; Samantha E Adams; Ryne A Didier; Steven C Horii; Teresa Victoria; Holly L Hedrick; N Scott Adzick; Lori J Howell; Julie S Moldenhauer; Beverly G Coleman Journal: Pediatr Radiol Date: 2019-01-11
Authors: Luke R Putnam; Matthew T Harting; Kuojen Tsao; Francesco Morini; Bradley A Yoder; Matías Luco; Pamela A Lally; Kevin P Lally Journal: Pediatrics Date: 2016-11 Impact factor: 7.124
Authors: Suzan Cochius-den Otter; Thomas Schaible; Anne Greenough; Arno van Heijst; Neil Patel; Karel Allegaert; Joost van Rosmalen; Dick Tibboel Journal: BMJ Open Date: 2019-11-05 Impact factor: 2.692
Authors: Kim Heiwegen; Arno Fj van Heijst; Horst Daniels-Scharbatke; Michelle Cp van Peperstraten; Ivo de Blaauw; Sanne Mbi Botden Journal: Eur J Pediatr Date: 2020-01-22 Impact factor: 3.183