Advertisement

Changing populations being treated with ECMO in the neonatal period – who are the others?

Published:November 16, 2022DOI:https://doi.org/10.1016/j.siny.2022.101402

      Highlights

      • The neonatal population treated with ECMO has changed and broadened over time
      • Less information is available about these newer populations, but many seem to have satisfactory outcomes
      • ECMO therapy is potentially lifesaving, but also highly invasive and the risk-benefit ratio much be evaluated for each individual patient with the available evidence
      • The role of ECMO in multi-factorial respiratory failure with a better understanding of who is being treated with ECMO
      • The impact of genetic testing on ECMO consideration and utilization
      • Long term outcomes for neonates with atypical causes of respiratory failure and ECMO use.

      Abstract

      Extracorporeal life support via extracorporeal membrane oxygenation (ECMO) has served the sickest of neonates for almost 50 years. Naturally, the characteristics of neonates receiving ECMO have changed. Advances in care have averted the need for ECMO for some, while complex cases with uncertain outcomes, previously not eligible for ECMO, are now considered. Characterizing the disease states and outcomes for neonates on ECMO is challenging as many infants do not fall into classic categories, i.e. meconium aspiration syndrome (MAS), respiratory distress syndrome (RDS), or congenital diaphragmatic hernia (CDH). Since 2017, over one third of neonatal respiratory ECMO runs reported to the Extracorporeal Life Support Organization Registry are grouped as Other, a catch-all that encompasses those with a diagnosis not included in the classic categories. This review summarizes the historical neonatal ECMO population, reviews advances in therapy and technology impacting neonatal care, and addresses the unknowns in the ever-growing category of Other.

      Keywords

      To read this article in full you will need to make a payment

      Purchase one-time access:

      Academic & Personal: 24 hour online accessCorporate R&D Professionals: 24 hour online access
      One-time access price info
      • For academic or personal research use, select 'Academic and Personal'
      • For corporate R&D use, select 'Corporate R&D Professionals'

      Subscribe:

      Subscribe to Seminars in Fetal and Neonatal Medicine
      Already a print subscriber? Claim online access
      Already an online subscriber? Sign in
      Institutional Access: Sign in to ScienceDirect

      References

        • Sharma J.
        • Sherman A.
        • Rimal A.
        • Haney B.
        • Weiner J.
        • Pallotto E.
        Neonatal respiratory extracorporeal membrane oxygenation and primary diagnosis: trends between two decades.
        J Perinatol. 2020; 40: 269-274
        • Bartlett R.H.
        • Roloff D.W.
        • Cornell R.G.
        • Andrews A.F.
        • Dillon P.W.
        • Zwischenberger J.B.
        Extracorporeal circulation in neonatal respiratory failure: a prospective randomized study.
        Pediatrics. 1985; 76: 479-487
        • Cashen K.
        • Thiagarajan R.R.
        • Collins Jr., J.W.
        • Rycus P.T.
        • Backer C.L.
        • Reynolds M.
        • et al.
        Extracorporeal membrane oxygenation in pediatric trisomy 21: 30 Years of experience from the extracorporeal life support organization registry.
        J Pediatr. 2015; 167: 403-408
        • Bartlett R.H.
        Esperanza: the first neonatal ECMO patient.
        Am Soc Artif Intern Organs J. 2017; 63: 832-843
        • O'Rourke P.P.
        • Crone R.K.
        • Vacanti J.P.
        • Ware J.H.
        • Lillehei C.W.
        • Parad R.B.
        • et al.
        Extracorporeal membrane oxygenation and conventional medical therapy in neonates with persistent pulmonary hypertension of the newborn: a prospective randomized study.
        Pediatrics. 1989; 84: 957-963
      1. UK collaborative randomised trial of neonatal extracorporeal membrane oxygenation. UK Collaborative ECMO Trail Group.
        Lancet. 1996; 348: 75-82
        • Toomasian J.M.
        • Snedecor S.M.
        • Cornell R.G.
        • Cilley R.E.
        • Bartlett R.H.
        National experience with extracorporeal membrane oxygenation for newborn respiratory failure. Data from 715 cases.
        ASAIO (Am Soc Artif Intern Organs) Trans. 1988; 34: 140-147
        • ECLS Registry Report
        International summary - April, 2022.
        Extracorporeal Life Support Organization, 2022
        • Lotze A.
        • Knight G.R.
        • Martin G.R.
        • Bulas D.I.
        • Hull W.M.
        • O'Donnell R.M.
        • et al.
        Improved pulmonary outcome after exogenous surfactant therapy for respiratory failure in term infants requiring extracorporeal membrane oxygenation.
        J Pediatr. 1993; 122: 261-268
        • Lotze A.
        • Mitchell B.R.
        • Bulas D.I.
        • Zola E.M.
        • Shalwitz R.A.
        • Gunkel J.H.
        Multicenter study of surfactant (beractant) use in the treatment of term infants with severe respiratory failure. Survanta in Term Infants Study Group.
        J Pediatr. 1998; 132: 40-47
        • El Shahed A.I.
        • Dargaville P.A.
        • Ohlsson A.
        • Soll R.
        Surfactant for meconium aspiration syndrome in term and late preterm infants.
        Cochrane Database Syst Rev. 2014; : CD002054
        • Lotze A.
        • Knight G.R.
        • Anderson K.D.
        • Hull W.M.
        • Whitsett J.A.
        • O'Donnell R.M.
        • et al.
        Surfactant (beractant) therapy for infants with congenital diaphragmatic hernia on ECMO: evidence of persistent surfactant deficiency.
        J Pediatr Surg. 1994; 29: 407-412
        • Colby C.E.
        • Lally K.P.
        • Hintz S.R.
        • Lally P.A.
        • Tibboel D.
        • Moya F.R.
        • et al.
        Surfactant replacement therapy on ECMO does not improve outcome in neonates with congenital diaphragmatic hernia.
        J Pediatr Surg. 2004; 39: 1632-1637
        • Van Meurs K.
        • Congenital Diaphragmatic Hernia Study G.
        Is surfactant therapy beneficial in the treatment of the term newborn infant with congenital diaphragmatic hernia?.
        J Pediatr. 2004; 145: 312-316
        • Pepke-Zaba J.
        • Higenbottam T.W.
        • Dinh-Xuan A.T.
        • Stone D.
        • Wallwork J.
        Inhaled nitric oxide as a cause of selective pulmonary vasodilatation in pulmonary hypertension.
        Lancet. 1991; 338: 1173-1174
        • DeMarco V.
        • Skimming J.
        • Ellis T.M.
        • Cassin S.
        Nitric oxide inhalation. Effects on the ovine neonatal pulmonary and systemic circulations.
        Chest. 1994; 105 (2S): 91S
        • Barrington K.J.
        • Finer N.
        • Pennaforte T.
        • Altit G.
        Nitric oxide for respiratory failure in infants born at or near term.
        Cochrane Database Syst Rev. 2017; 1: CD000399
      2. Inhaled nitric oxide and hypoxic respiratory failure in infants with congenital diaphragmatic hernia. The Neonatal Inhaled Nitric Oxide Study Group (NINOS).
        Pediatrics. 1997; 99: 838-845
        • Carter J.M.
        • Gerstmann D.R.
        • Clark R.H.
        • Snyder G.
        • Cornish J.D.
        • Null Jr., D.M.
        • et al.
        High-frequency oscillatory ventilation and extracorporeal membrane oxygenation for the treatment of acute neonatal respiratory failure.
        Pediatrics. 1990; 85: 159-164
        • Varnholt V.
        • Lasch P.
        • Suske G.
        • Kachel W.
        • Brands W.
        High frequency oscillatory ventilation and extracorporeal membrane oxygenation in severe persistent pulmonary hypertension of the newborn.
        Eur J Pediatr. 1992; 151: 769-774
        • Clark R.H.
        • Yoder B.A.
        • Sell M.S.
        Prospective, randomized comparison of high-frequency oscillation and conventional ventilation in candidates for extracorporeal membrane oxygenation.
        J Pediatr. 1994; 124: 447-454
        • Kinsella J.P.
        • Truog W.E.
        • Walsh W.F.
        • Goldberg R.N.
        • Bancalari E.
        • Mayock D.E.
        • 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
        • Deprest J.A.
        • Nicolaides K.H.
        • Benachi A.
        • Gratacos E.
        • Ryan G.
        • Persico N.
        • et al.
        Randomized trial of fetal surgery for severe left diaphragmatic hernia.
        N Engl J Med. 2021; 385: 107-118
        • Kays D.W.
        • Islam S.
        • Perkins J.M.
        • Larson S.D.
        • Taylor J.A.
        • Talbert J.L.
        Outcomes in the physiologically most severe congenital diaphragmatic hernia (CDH) patients: whom should we treat?.
        J Pediatr Surg. 2015; 50: 893-897
        • Deprest J.
        • Flake A.
        How should fetal surgery for congenital diaphragmatic hernia be implemented in the post-TOTAL trial era: a discussion.
        Prenat Diagn. 2022; 42: 301-309
        • Gien J.
        • Kinsella J.P.
        • Behrendt N.J.
        • Zaretsky M.V.
        • Galan H.L.
        • Liechty K.W.
        Improved survival for infants with severe congenital diaphragmatic hernia.
        J Perinatol. 2022; 42: 1189-1194
        • Barbaro R.P.
        • Paden M.L.
        • Guner Y.S.
        • Raman L.
        • Ryerson L.M.
        • Alexander P.
        • et al.
        Pediatric extracorporeal life support organization registry international report 2016.
        Am Soc Artif Intern Organs J. 2017; 63: 456-463
        • Roberts J.
        • Keene S.
        • Heard M.
        • McCracken C.
        • Gauthier T.W.
        Successful primary use of VVDL+V ECMO with cephalic drain in neonatal respiratory failure.
        J Perinatol. 2016; 36: 126-131
        • Guner Y.S.
        • Harting M.T.
        • Fairbairn K.
        • Delaplain P.T.
        • Zhang L.
        • Chen Y.
        • et al.
        Outcomes of infants with congenital diaphragmatic hernia treated with venovenous versus venoarterial extracorporeal membrane oxygenation: a propensity score approach.
        J Pediatr Surg. 2018; 53: 2092-2099
        • Lillie J.
        • Boot L.
        • Speggiorin S.
        • Nyman A.G.
        Factors behind decline of venovenous extracorporeal membrane oxygenation to support neonatal respiratory failure.
        Pediatr Crit Care Med. 2020; 21: e502-e504
        • Delaplain P.T.
        • Yu P.T.
        • Ehwerhemuepha L.
        • Nguyen D.V.
        • Jancelewicz T.
        • Stein J.
        • et al.
        Predictors of long ECMO runs for congenital diaphragmatic hernia.
        J Pediatr Surg. 2020; 55: 993-997
        • Alore E.A.
        • Fallon S.C.
        • Thomas J.A.
        • Vogel A.M.
        Outcomes after extracorporeal life support cannulation in pediatric patients with trisomy 13 and trisomy 18.
        J Surg Res. 2021; 257: 260-266
        • Coleman R.D.
        • Goldman J.
        • Moffett B.
        • Guffey D.
        • Loftis L.
        • Thomas J.
        • et al.
        Extracorporeal membrane oxygenation mortality in high-risk populations: an analysis of the pediatric health information system database.
        Am Soc Artif Intern Organs J. 2020; 66: 327-331
        • Furlong-Dillard J.M.
        • Amula V.
        • Bailly D.K.
        • Bleyl S.B.
        • Wilkes J.
        • Bratton S.L.
        Use of extracorporeal membrane oxygenation and mortality in pediatric cardiac surgery patients with genetic conditions: a multicenter analysis.
        Pediatr Crit Care Med. 2017; 18: 850-858
        • Reeve N.H.
        • Kahane J.B.
        • Spinner A.G.
        • TJ OL
        Ex utero intrapartum treatment to extracorporeal membrane oxygenation: lifesaving management of a giant cervical teratoma.
        J Laryngol Otol. 2020; 134: 650-653
        • Yeh Y.T.
        • Liu C.
        • Tsai H.L.
        • Wu F.Y.
        • Soong W.J.
        • Lee Y.S.
        • et al.
        A combination of tracheoplasty and tracheal stenting is an acceptable method of treating severe congenital tracheobronchial stenosis under extracorporeal membrane oxygenation.
        J Pediatr Surg. 2019; 54: 2492-2497
        • Mychaliska G.B.
        • Bryner B.S.
        • Nugent C.
        • Barks J.
        • Hirschl R.B.
        • McCrudden K.
        • et al.
        Giant pulmonary sequestration: the rare case requiring the EXIT procedure with resection and ECMO.
        Fetal Diagn Ther. 2009; 25: 163-166
        • Kunisaki S.M.
        • Fauza D.O.
        • Barnewolt C.E.
        • Estroff J.A.
        • Myers L.B.
        • Bulich L.A.
        • et al.
        Ex utero intrapartum treatment with placement on extracorporeal membrane oxygenation for fetal thoracic masses.
        J Pediatr Surg. 2007; 42: 420-425
        • Chapman R.L.
        • Peterec S.M.
        • Bizzarro M.J.
        • Mercurio M.R.
        Patient selection for neonatal extracorporeal membrane oxygenation: beyond severity of illness.
        J Perinatol. 2009; 29: 606-611
        • Cuevas Guaman M.
        • Lucke A.M.
        • Hagan J.L.
        • Kaiser J.R.
        Bleeding complications and mortality in neonates receiving therapeutic hypothermia and extracorporeal membrane oxygenation.
        Am J Perinatol. 2018; 35: 271-276
        • Celik I.H.
        • Demirel G.
        • Canpolat F.E.
        • Erdeve O.
        • Dilmen U.
        Surfactant therapy for maternal blood aspiration: an unusual cause of neonatal respiratory distress syndrome.
        Indian J Pediatr. 2012; 79: 1358-1359
        • Finer N.N.
        Surfactant use for neonatal lung injury: beyond respiratory distress syndrome.
        Paediatr Respir Rev. 2004; 5: S289-S297
        • Mammas I.N.
        • Thiagarajan P.
        Water aspiration syndrome at birth - report of two cases.
        J Matern Fetal Neonatal Med. 2009; 22: 365-367
        • Carpenter L.
        • Weston P.
        Neonatal respiratory consequences from water birth.
        J Paediatr Child Health. 2012; 48: 419-423
        • Davis R.P.
        • Mychaliska G.B.
        Neonatal pulmonary physiology.
        Semin Pediatr Surg. 2013; 22: 179-184
        • Cotten C.M.
        Pulmonary hypoplasia.
        Semin Fetal Neonatal Med. 2017; 22: 250-255
        • Rescorla F.J.
        • West K.W.
        • Vane D.W.
        • Engle W.
        • Grosfeld J.L.
        Pulmonary hypertension in neonatal cystic lung disease: survival following lobectomy and ECMO in two cases.
        J Pediatr Surg. 1990; 25: 1054-1056
        • Ben-Ishay O.
        • Nicksa G.A.
        • Wilson J.M.
        • Buchmiller T.L.
        Management of giant congenital pulmonary airway malformations requiring pneumonectomy.
        Ann Thorac Surg. 2012; 94: 1073-1078
        • Attar M.A.
        • Donn S.M.
        Congenital chylothorax.
        Semin Fetal Neonatal Med. 2017; 22: 234-239
        • Mettauer N.
        • Agrawal S.
        • Pierce C.
        • Ashworth M.
        • Petros A.
        Outcome of children with pulmonary lymphangiectasis.
        Pediatr Pulmonol. 2009; 44: 351-357
        • Resch B.
        • Sever Yildiz G.
        • Reiterer F.
        Congenital chylothorax of the newborn: a systematic analysis of published cases between 1990 and 2018.
        Respiration. 2022; 101: 84-96
        • Don M.
        • Orsaria M.
        • Da Dalt E.
        • Tringali C.
        • Sacher B.
        Rapidly fatal "congenital lung dysplasia": a case report and review of the literature.
        Fetal Pediatr Pathol. 2014; 33: 109-113
        • Bishop N.B.
        • Stankiewicz P.
        • Steinhorn R.H.
        Alveolar capillary dysplasia.
        Am J Respir Crit Care Med. 2011; 184: 172-179
        • Szafranski P.
        • Gambin T.
        • Dharmadhikari A.V.
        • Akdemir K.C.
        • Jhangiani S.N.
        • Schuette J.
        • et al.
        Pathogenetics of alveolar capillary dysplasia with misalignment of pulmonary veins.
        Hum Genet. 2016; 135: 569-586
        • Towe C.T.
        • White F.V.
        • Grady R.M.
        • Sweet S.C.
        • Eghtesady P.
        • Wegner D.J.
        • et al.
        Infants with atypical presentations of alveolar capillary dysplasia with misalignment of the pulmonary veins who underwent bilateral lung transplantation.
        J Pediatr. 2018; 194: 158-164 e1
        • Chelliah B.P.
        • Brown D.
        • Cohen M.
        • Talleyrand A.J.
        • Shen-Schwarz S.
        Alveolar capillary dysplasia--a cause of persistent pulmonary hypertension unresponsive to a second course of extracorporeal membrane oxygenation.
        Pediatrics. 1995; 96: 1159-1161
        • DeBoer E.M.
        • Keene S.
        • Winkler A.M.
        • Shehata B.M.
        Identical twins with lethal congenital pulmonary airway malformation type 0 (acinar dysplasia): further evidence of familial tendency.
        Fetal Pediatr Pathol. 2012; 31: 217-224
        • Ryan G.
        • Somme S.
        • Crombleholme T.M.
        Airway compromise in the fetus and neonate: prenatal assessment and perinatal management.
        Semin Fetal Neonatal Med. 2016; 21: 230-239
        • Gatzweiler E.
        • Hoppe B.
        • Dewald O.
        • Berg C.
        • Muller A.
        • Reutter H.
        • et al.
        Extracorporeal membrane oxygenation support in a newborn with lower urinary tract obstruction and pulmonary hypoplasia: a case report.
        J Med Case Rep. 2018; 12: 210
        • Caesar R.E.
        • Packer M.G.
        • Kaplan G.W.
        • Dudell G.G.
        • Guerrant A.L.
        • Griswold W.R.
        • et al.
        Extracorporeal membrane oxygenation in the neonate with congenital renal disease and pulmonary hypoplasia.
        J Pediatr Surg. 1995; 30: 1560-1563
        • Bagdure D.
        • Torres N.
        • Walker L.K.
        • Waddell J.
        • Bhutta A.
        • Custer J.W.
        Extracorporeal membrane oxygenation for neonates with congenital renal and urological anomalies and pulmonary hypoplasia: a case report and review of the extracorporeal life support organization registry.
        J Pediatr Intensive Care. 2017; 6: 188-193
        • Wen J.X.
        • Feldenberg L.R.
        • Abraham E.
        • Sadiq F.
        • Christensen K.M.
        • Braddock S.R.
        Continuous venovenous hemodialysis via extracorporeal membrane oxygenation pump for treatment of hyperammonemia secondary to propionic acidemia in monochorionic diamniotic twin boys.
        J Pediatr. 2016; 175: 231-232
        • van Heijst A.F.
        • de Mol A.C.
        • Ijsselstijn H.
        ECMO in neonates: neuroimaging findings and outcome.
        Semin Perinatol. 2014; 38: 104-113
        • Tierradentro-Garcia L.O.
        • Saade-Lemus S.
        • Freeman C.
        • Kirschen M.
        • Huang H.
        • Vossough A.
        • et al.
        Cerebral blood flow of the neonatal brain after hypoxic-ischemic injury.
        Am J Perinatol. 2021;
        • Massaro A.N.
        • Govindan R.B.
        • Vezina G.
        • Chang T.
        • Andescavage N.N.
        • Wang Y.
        • et al.
        Impaired cerebral autoregulation and brain injury in newborns with hypoxic-ischemic encephalopathy treated with hypothermia.
        J Neurophysiol. 2015; 114: 818-824
        • Pakvasa M.A.
        • Winkler A.M.
        • Hamrick S.E.
        • Josephson C.D.
        • Patel R.M.
        Observational study of haemostatic dysfunction and bleeding in neonates with hypoxic-ischaemic encephalopathy.
        BMJ Open. 2017; 7e013787
        • Shankaran S.
        • Laptook A.R.
        • Ehrenkranz R.A.
        • Tyson J.E.
        • McDonald S.A.
        • Donovan E.F.
        • et al.
        Whole-body hypothermia for neonates with hypoxic-ischemic encephalopathy.
        N Engl J Med. 2005; 353: 1574-1584
        • Shankaran S.
        • Pappas A.
        • Laptook A.R.
        • McDonald S.A.
        • Ehrenkranz R.A.
        • Tyson J.E.
        • et al.
        Outcomes of safety and effectiveness in a multicenter randomized, controlled trial of whole-body hypothermia for neonatal hypoxic-ischemic encephalopathy.
        Pediatrics. 2008; 122: e791-e798
        • Lakshminrusimha S.
        • Shankaran S.
        • Laptook A.
        • McDonald S.
        • Keszler M.
        • Van Meurs K.
        • et al.
        Pulmonary hypertension associated with hypoxic-ischemic encephalopathy-antecedent characteristics and comorbidities.
        J Pediatr. 2018; 196: 45-51 e3
        • Agarwal P.
        • Altinok D.
        • Desai J.
        • Shanti C.
        • Natarajan G.
        In-hospital outcomes of neonates with hypoxic-ischemic encephalopathy receiving extracorporeal membrane oxygenation.
        J Perinatol. 2019; 39: 661-665
        • Agarwal P.
        • Natarajan G.
        • Sullivan K.
        • Rao R.
        • Rintoul N.
        • Zaniletti I.
        • et al.
        Venovenous versus venoarterial extracorporeal membrane oxygenation among infants with hypoxic-ischemic encephalopathy: is there a difference in outcome?.
        J Perinatol. 2021; 41: 1916-1923
        • Samies N.L.
        • James S.H.
        • Kimberlin D.W.
        Neonatal herpes simplex virus disease: updates and continued challenges.
        Clin Perinatol. 2021; 48: 263-274
        • Prodhan P.
        • Bhutta A.T.
        • Gossett J.M.
        • Stroud M.H.
        • Rycus P.T.
        • Bratton S.L.
        • et al.
        Extracorporeal membrane oxygenation support among children with adenovirus infection: a review of the Extracorporeal Life Support Organization registry.
        Am Soc Artif Intern Organs J. 2014; 60: 49-56
        • Prodhan P.
        • Wilkes R.
        • Ross A.
        • Garcia X.
        • Bhutta A.T.
        • Rycus P.
        • et al.
        Neonatal herpes virus infection and extracorporeal life support.
        Pediatr Crit Care Med. 2010; 11: 599-602