Subscribe to RSS
DOI: 10.1055/s-2006-924751
Georg Thieme Verlag KG Stuttgart, New York · Masson Editeur Paris
Extracorporeal Membrane Oxygenation in Infants with Congenital Diaphragmatic Hernia: A Systematic Review of the Evidence
Publication History
Received: May 1, 2006
Accepted after Revision: May 13, 2006
Publication Date:
08 January 2007 (online)
Abstract
Aim: The aim of this study was to evaluate the evidence supporting the use of extracorporeal membrane oxygenation (ECMO) in infants with congenital diaphragmatic hernia (CDH) and severe respiratory failure. Methods: Medline, Embase, ISI Current Contents and Biosis databases were searched using a defined strategy. Case reports and opinion articles were excluded. We performed: 1) a systematic review of non randomised studies comparing mortality when ECMO was not available with a period when ECMO was available. Mortality was classified as “early” (before hospital discharge) and “late” (after discharge). Patients were classified as “ECMO” and “non-ECMO” candidates according to criteria reported by the authors; 2) a meta-analysis of randomised controlled trials (RCTs) comparing ECMO and conventional mechanical ventilation (CMV). Differences in mortality are reported as relative risk (RR) and 95 % confidence intervals. Results: a) Systematic review: 658 studies and 21 (2043 patients) fulfilled the entry criteria. Both early (RR 0.60 [0.51 - 0.70]; p < 0.001) and late mortality (RR 0.63 [0.53 - 0.73]; p < 0.001) were significantly lower when ECMO was available than when ECMO was unavailable. This difference in mortality was observed in “ECMO candidates” (RR 0.46 [0.32 - 0.68]; p < 0.001) but not in “non-ECMO candidates” (RR 0.80 [0.58 - 1.10]; p = 0.17). b) Meta-analysis: 3 trials comparing ECMO and conventional ventilation were identified which included 39 infants with CDH. The early mortality was significantly lower with ECMO compared to CMV (RR 0.73 [95 % CI 0.55 - 0.99]; p < 0.04), however, late mortality was similar in the two groups (RR 0.83 [0.66 - 1.05]; p = 0.12). Conclusions: Non randomised studies suggest a reduction in mortality with ECMO. However, differences in the indications for ECMO and improvements in other treatment modalities may contribute to this reduction. The meta-analysis of RCTs indicates a reduction in early mortality with ECMO but no long-term benefit. A large RCT in infants with CDH and severe respiratory failure is warranted.
Key words
Congenital diaphragmatic hernia - ECMO - extracorporeal life support - extracorporeal membrane oxygenation
References
- 1 Atkinson J B, Poon M W. ECMO and the management of congenital diaphragmatic hernia with large diaphragmatic defects requiring a prosthetic patch. J Pediatr Surg. 1992; 27 754-756
- 2 Bartlett R H, Gazzaniga A B, Jefferies M R. et al . Extracorporeal membrane oxygenation (ECMO) cardiopulmonary support in infancy. Trans Am Soc Artif Intern Organs. 1976; 22 80-93
- 3 Bartlett R H, Roloff D W, Cornell R G. et al . Extracorporeal circulation in neonatal respiratory failure: a prospective randomized study. Pediatrics. 1985; 76 479-487
- 4 Bartlett R H, Gazzaniga A B, Toomasian J. et al . Extracorporeal membrane oxygenation (ECMO) in neonatal respiratory failure. 100 cases. Ann Surg. 1986; 204 236-245
- 5 Bennett C C, Johnson A, Field D J. et al . UK collaborative randomised trial of neonatal extracorporeal membrane oxygenation: follow-up to age 4 years. Lancet. 2001; 357 1094-1096
- 6 Breaux Jr C W, Rouse T M, Cain W S. et al . Improvement in survival of patients with congenital diaphragmatic hernia utilizing a strategy of delayed repair after medical and/or extracorporeal membrane oxygenation stabilization. J Pediatr Surg. 1991; 26 333-336
- 7 Chan D K, Ho L Y, Joseph V T. Mortality among infants with high-risk congenital diaphragmatic hernia in Singapore. J Pediatr Surg. 1997; 32 95-98
- 8 D'Agostino J A, Bernbaum J C, Gerdes M. et al . Outcome for infants with congenital diaphragmatic hernia requiring extracorporeal membrane oxygenation: the first year. J Pediatr Surg. 1995; 30 10-15
- 9 Davis P J, Firmin R K, Manktelow B. et al . Long-term outcome following extracorporeal membrane oxygenation for congenital diaphragmatic hernia: the UK experience. J Pediatr. 2004; 144 309-315
- 10 de la Hunt M N, Madden N, Scott J E. et al . Is delayed surgery really better for congenital diaphragmatic hernia? A prospective randomized clinical trial. J Pediatr Surg. 1996; 31 1554-1556
- 11 Desfrere L, Jarreau P H, Dommergues M. et al . Impact of delayed repair and elective high-frequency oscillatory ventilation on survival of antenatally diagnosed congenital diaphragmatic hernia: first application of these strategies in the more “severe” subgroup of antenatally diagnosed newborns. J Intens Care Med. 2000; 26 924-941
- 12 Dubois A, Storme L, Jaillard S. et al . Congenital hernia of the diaphragm. A retrospective study of 123 cases recorded in the Neonatal Medicine Department, URHC in Lille between 1985 and 1996. Arch Pediatr. 2000; 7 132-142
- 13 Ehren H, Frenckner B, Palmer K. Diaphragmatic hernia in infancy and childhood - 20 years experience. Eur J Pediatr Surg. 1992; 2 327-331
- 14 Evans M, Dudell G. Congenital diaphragmatic hernia: can ECMO save them all?. Pediatr Res. 1991; 29 213A
- 15 Extracorporeal Life Support Organization .ECMO Registry of the Extracorporeal Life Support Organization (ELSO), Ann Arbor, Michigan,. January/2005
- 16 Frenckner B, Ehren H, Granholm T. et al . Improved results in patients who have congenital diaphragmatic hernia using preoperative stabilization, extracorporeal membrane oxygenation, and delayed surgery. J Pediatr Surg. 1997; 32 1185-1189
- 17 German J C, Gazzaniga A B, Amlie R. et al . Management of pulmonary insufficiency in diaphragmatic hernia using extracorporeal circulation with a membrane oxygenator (ECMO). J Pediatr Surg. 1977; 12 905-912
- 18 Hamutcu R, Nield T A, Garg M. et al . Long-term pulmonary sequelae in children who were treated with extracorporeal membrane oxygenation for neonatal respiratory failure. Pediatrics. 2004; 114 1292-1296
- 19 Harrington K P, Goldman A P. The role of extracorporeal membrane oxygenation in congenital diaphragmatic hernia. Semin Pediatr Surg. 2005; 14 72-76
- 20 Harrison M R, Adzick N S, Estes J M. et al . A prospective study of the outcome for fetuses with diaphragmatic hernia. JAMA. 1994; 271 382-384
- 21 Heiss K, Manning P, Oldham K T. et al . Reversal of mortality for congenital diaphragmatic hernia with ECMO. Ann Surg. 1989; 209 225-230
- 22 Jaillard S M, Pierrat V, Dubois A. et al . Outcome at 2 years of infants with congenital diaphragmatic hernia: a population-based study. Ann Thorac Surg. 2003; 75 250-256
- 23 Kamata S, Usui N, Ishikawa S. et al . Prolonged preoperative stabilization using high-frequency oscillatory ventilation does not improve the outcome in neonates with congenital diaphragmatic hernia. Pediatr Surg Int. 1998; 13 542-546
- 24 Kato T, Yoshino H, Hebiguchi T. et al . Therapeutic strategy and clinical outcome in congenital diaphragmatic hernia. Nippon Geka Gakkai Zasshi. 1997; 98 1001-1007
- 25 Kays D W, Langham Jr M R, Ledbetter D J. et al . Detrimental effects of standard medical therapy in congenital diaphragmatic hernia. Ann Surg. 1999; 230 340-348
- 26 Keshen T H, Gursoy M, Shew S B. et al . Does extracorporeal membrane oxygenation benefit neonates with congenital diaphragmatic hernia? Application of a predictive equation. J Pediatr Surg. 1997; 32 818-822
- 27 Khan A M, Lally K P. The role of extracorporeal membrane oxygenation in the management of infants with congenital diaphragmatic hernia. Semin Perinatol. 2005; 29 118-122
- 28 Kinsella J P, Neish S R, Shaffer E. et al . Low-dose inhalational nitric oxide in persistent pulmonary hypertension of the newborn. Lancet. 1992; 340 819-820
- 29 Kinsella J P, Truog W E, Walsh W F. et al . Randomized, multicenter trial of inhaled nitric oxide and high-frequency oscillatory ventilation in severe, persistent pulmonary hypertension of the newborn. J Pediatr. 1997; 131 55-62
- 30 Lessin M S, Thompson I M, Deprez M F. et al . Congenital diaphragmatic hernia with or without extracorporeal membrane oxygenation: are we making progress?. J Am Coll Surg. 1995; 181 65-71
- 31 Lyrenne R K, Phillips J B. Control of pulmonary vascular resistance in the fetus and newborn. Clin Perinatol. 1984; 11 561-584
- 32 Mallik K, Rodgers B M, McGahren E D. Congenital diaphragmatic hernia: experience in a single institution from 1978 through 1994. Ann Thorac Surg. 1995; 60 1331-1335
- 33 McGahren E D, Mallik K, Rodgers B M. Neurological outcome is diminished in survivors of congenital diaphragmatic hernia requiring extracorporeal membrane oxygenation. J Pediatr Surg. 1997; 32 1216-1220
- 34 Moyer V, Moya F, Tibboel R. et al . Late versus early surgical correction for congenital diaphragmatic hernia in newborn infants. Cochrane Database Syst Rev. 2003; 3 CD001695
- 35 Nagaya M, Tsuda M, Murahashi O. et al . Management of congenital diaphragmatic hernia by extracorporeal membrane oxygenation (ECMO). Eur J Pediatr Surg. 1991; 1 10-14
- 36 Nagaya M, Tsuda M, Hiraiwa K. et al . Extracorporeal membrane oxygenation (ECMO): applications and results in patients with congenital diaphragmatic hernia. Pediatr Surg Int. 1993; 8 294-297
- 37 Nakayama D K, Motoyama E K, Tagge E M. Effect of preoperative stabilization on respiratory system compliance and outcome in newborn infants with congenital diaphragmatic hernia. J Pediatr. 1991; 118 793-799
- 38 Nio M, Haase G, Kennaugh J. et al . A prospective randomized trial of delayed versus immediate repair of congenital diaphragmatic hernia. J Pediatr Surg. 1994; 29 618-621
- 39 Okazaki T, Kohno S, Hasegawa S. et al . Congenital diaphragmatic hernia: efficacy of ultrasound examination in its management. Pediatr Surg Int. 2003; 19 176-179
- 40 Reyes C, Chang L K, Waffarn F. et al . Delayed repair of congenital diaphragmatic hernia with early high-frequency oscillatory ventilation during preoperative stabilization. J Pediatr Surg. 1998; 33 1010-1014
- 41 Roberts J D, Polaner D M, Lang P. et al . Inhaled nitric oxide in persistent pulmonary hypertension of the newborn. Lancet. 1992; 340 818-819
- 42 Rothenbach P, Lange P, Powell D. The use of extracorporeal membrane oxygenation in infants with congenital diaphragmatic hernia. Semin Perinatol. 2005; 29 40-44
- 43 Sakai H, Tamura M, Hosokawa Y. et al . Effect of surgical repair on respiratory mechanics in congenital diaphragmatic hernia. J Pediatr. 1987; 111 432-438
- 44 Schumacher R E, Roloff D W, Chapman R. et al . Extracorporeal membrane oxygenation in term newborns. A prospective cost-benefit analysis. ASAIO J. 1993; 39 873-879
- 45 Shanley C J, Hirschl R B, Schumacher R E. et al . Extracorporeal life support for neonatal respiratory failure. A 20-year experience. Ann Surg. 1994; 220 269-280
- 46 Somaschini M, D'Alessio A, Bellan C. et al . Congenital diaphragmatic hernia: increase of survival using advanced ventilation techniques and preoperative stabilization. Ital J Pediatr. 1998; 24 128-132
- 47 Somaschini M, Locatelli G, Salvoni L. et al . Impact of new treatments for respiratory failure on outcome of infants with congenital diaphragmatic hernia. Eur J Pediatr. 1999; 158 780-784
- 48 Stege G, Fenton A, Jaffray B. Nihilism in the 1990s: the true mortality of congenital diaphragmatic hernia. Pediatrics. 2003; 112 532-535
- 49 The Neonatal Inhaled Nitric Oxide Study Group . Inhaled nitric oxide and hypoxic respiratory failure in infants with congenital diaphragmatic hernia. Pediatrics. 1997; 99 838-845
- 50 UK Collaborative ECMO Trial Group . UK collaborative randomised trial of neonatal extracorporeal membrane oxygenation. UK Collaborative ECMO Trial Group. Lancet. 1996; 348 75-82
- 51 UK Collaborative ECMO Trial Group . The collaborative UK ECMO (Extracorporeal Membrane Oxygenation) trial: follow-up to 1 year of age. Pediatrics. 1998; 101 E1
- 52 vd Staak F H, de Haan A F, Geven W B. et al . Improving survival for patients with high-risk congenital diaphragmatic hernia by using extracorporeal membrane oxygenation. J Pediatr Surg. 1995; 30 1463-1467
- 53 Weber T R, Kountzman B, Dillon P A. et al . Improved survival in congenital diaphragmatic hernia with evolving therapeutic strategies. Arch Surg. 1998; 133 498-502
- 54 West K W, Bengston K, Rescorla F J. et al . Delayed surgical repair and ECMO improves survival in congenital diaphragmatic hernia. Ann Surg. 1992; 216 454-460
- 55 Wiener E S. Congenital posterolateral diaphragmatic hernia: new dimensions in management. Surgery. 1982; 92 670-681
- 56 Wilson J M, Lund D P, Lillehei C W. et al . Congenital diaphragmatic hernia - a tale of two cities: the Boston experience. J Pediatr Surg. 1997; 32 401-405
- 57 Wung J T, James L S, Kilchevsky E. et al . Management of infants with severe respiratory failure and persistence of the fetal circulation without hyperventilation. Pediatrics. 1985; 76 488-494
- 58 Wung J T, Sahni R, Moffitt S T. et al . Congenital diaphragmatic hernia: survival treated with very delayed surgery, spontaneous respiration, and no chest tube. J Pediatr Surg. 1995; 30 406-409
- 59 Zelen M. Play the winner rule and the controlled clinical trial. J Am Stat Assoc. 1969; 64 131-146
Prof. Agostino Pierro
UCL Institute of Child Health and Great Ormond Street Hospital for Children NHS Trust
30 Guilford Street
London WC1N 1EH
United Kingdom
Email: pierro.sec@ich.ucl.ac.uk