Am J Perinatol 2017; 34(03): 248-252
DOI: 10.1055/s-0036-1586120
Original Article
Thieme Medical Publishers 333 Seventh Avenue, New York, NY 10001, USA.

Does Ventilatory Time Retain Its Validity in Predicting Neurodevelopmental Outcome at Two Years of Age in High-Risk Congenital Diaphragmatic Hernia Survivors?

Francesca Bevilacqua
1   Unit of Clinical Psychology, Department of Neuroscience and Neurorehabilitation, Bambino Gesù Children's Hospital, Rome, Italy
,
Francesco Morini
2   Department of Neonatal Medicine and Surgery, Bambino Gesù Children's Hospital, Rome, Italy
,
Antonio Zaccara
3   Department of Pediatric Surgery, Bambino Gesù Children's Hospital, Rome, Italy
,
Laura Valfrè
2   Department of Neonatal Medicine and Surgery, Bambino Gesù Children's Hospital, Rome, Italy
,
Lelia Rotondi Aufiero
4   Department of Pediatrics, Bambino Gesù Children's Hospital, Rome, Italy
,
Simonetta Gentile
1   Unit of Clinical Psychology, Department of Neuroscience and Neurorehabilitation, Bambino Gesù Children's Hospital, Rome, Italy
,
Pietro Bagolan
2   Department of Neonatal Medicine and Surgery, Bambino Gesù Children's Hospital, Rome, Italy
,
Lucia Aite
1   Unit of Clinical Psychology, Department of Neuroscience and Neurorehabilitation, Bambino Gesù Children's Hospital, Rome, Italy
› Author Affiliations
Further Information

Publication History

08 March 2016

17 June 2016

Publication Date:
25 July 2016 (online)

Abstract

Objective To evaluate if in high-risk non–extracorporeal membrane oxygenation (ECMO)-treated congenital diaphragmatic hernia (CDH) survivors, ventilatory time (VT) is correlated to and can be used as clinical marker of neurodevelopmental delay at 2 years of age.

Study Design Cohort study was conducted between 2008 and 2012. Mental, motor, and language development were assessed by the Bayley Scales of Infant and Toddler Development III. The correlation between VT and neurodevelopmental outcome (NDO) was analyzed using Pearson's test. Receiver operating characteristic (ROC) analysis was performed to determine the accuracy and best cutoff value of VT to predict the risk of neurodevelopmental delay. Statistical significance was set at p < 0.05.

Results A total of 49 patients form the subject of this study. VT during first admission was inversely correlated with cognitive (r = –0.4116; p = 0.0033), motor (r = –0.4241; p = 0.0024), and language development (r = –0.3564; p = 0.0119). Using ROC curve analysis, VT was a significant predictor for neurodevelopmental delay in the cognitive (area under the curve [AUC]: 0.864, sensitivity: 100; specificity: 66.67; p < 0.0001) and motor (AUC: 0.902; sensitivity: 100; specificity: 73.17; p < 0.0001) scales, but not in the language scale. The best cutoff value for both scales was 9 days.

Conclusion Within a population of high-risk non-ECMO-treated CDH survivors, VT appears to retain its validity as a clinical marker of adverse NDO in cognitive and motor domains.

 
  • References

  • 1 Wynn J, Krishnan U, Aspelund G , et al. Outcomes of congenital diaphragmatic hernia in the modern era of management. J Pediatr 2013; 163 (1) 114-119.e1
  • 2 Benjamin JR, Gustafson KE, Smith PB , et al. Perinatal factors associated with poor neurocognitive outcome in early school age congenital diaphragmatic hernia survivors. J Pediatr Surg 2013; 48 (4) 730-737
  • 3 Lally KP, Engle W ; American Academy of Pediatrics Section on Surgery; American Academy of Pediatrics Committee on Fetus and Newborn. Postdischarge follow-up of infants with congenital diaphragmatic hernia. Pediatrics 2008; 121 (3) 627-632
  • 4 Bagolan P, Morini F. Long-term follow up of infants with congenital diaphragmatic hernia. Semin Pediatr Surg 2007; 16 (2) 134-144
  • 5 Danzer E, Hedrick HL. Neurodevelopmental and neurofunctional outcomes in children with congenital diaphragmatic hernia. Early Hum Dev 2011; 87 (9) 625-632
  • 6 Bevilacqua F, Morini F, Zaccara A , et al. Neurodevelopmental outcome in congenital diaphragmatic hernia survivors: role of ventilatory time. J Pediatr Surg 2015; 50 (3) 394-398
  • 7 Reiss I, Schaible T, van den Hout L , et al; CDH EURO Consortium. Standardized postnatal management of infants with congenital diaphragmatic hernia in Europe: the CDH EURO Consortium consensus. Neonatology 2010; 98 (4) 354-364
  • 8 Bayley N. Bayley Scales of Infant and Toddler Development. 3rd ed. Administration Manual. Florence, Italy: Giunti O.S.; 2010
  • 9 Danzer E, Gerdes M, Bernbaum J , et al. Neurodevelopmental outcome of infants with congenital diaphragmatic hernia prospectively enrolled in an interdisciplinary follow-up program. J Pediatr Surg 2010; 45 (9) 1759-1766
  • 10 Safavi A, Synnes AR, O'Brien K, Chiang M, Skarsgard ED, Chiu PP ; Canadian Pediatric Surgery Network. Multi-institutional follow-up of patients with congenital diaphragmatic hernia reveals severe disability and variations in practice. J Pediatr Surg 2012; 47 (5) 836-841
  • 11 Leeuwen L, Walker K, Halliday R, Fitzgerald DA. Neurodevelopmental outcome in congenital diaphragmatic hernia survivors during the first three years. Early Hum Dev 2014; 90 (8) 413-415
  • 12 Friedman S, Chen C, Chapman JS , et al. Neurodevelopmental outcomes of congenital diaphragmatic hernia survivors followed in a multidisciplinary clinic at ages 1 and 3. J Pediatr Surg 2008; 43 (6) 1035-1043
  • 13 Frisk V, Jakobson LS, Unger S, Trachsel D, O'Brien K. Long-term neurodevelopmental outcomes of congenital diaphragmatic hernia survivors not treated with extracorporeal membrane oxygenation. J Pediatr Surg 2011; 46 (7) 1309-1318
  • 14 Nelson HD, Nygren P, Walker M, Panoscha R. Screening for speech and language delay in preschool children: systematic evidence review for the US Preventive Services Task Force. Pediatrics 2006; 117 (2) e298-e319
  • 15 Peyre H, Bernard JY, Forhan A , et al. Predicting changes in language skills between 2 and 3 years in the EDEN mother-child cohort. PeerJ 2014; 2: e335 DOI: 10.7717/peerj.335.
  • 16 Cauley RP, Potanos K, Fullington N , et al. Pulmonary support on day of life 30 is a strong predictor of increased 1 and 5-year morbidity in survivors of congenital diaphragmatic hernia. J Pediatr Surg 2015; 50 (5) 849-855
  • 17 Cauley RP, Stoffan A, Potanos K , et al; Congenital Diaphragmatic Hernia Study Group. Pulmonary support on day 30 as a predictor of morbidity and mortality in congenital diaphragmatic hernia. J Pediatr Surg 2013; 48 (6) 1183-1189
  • 18 Reuner G, Fields AC, Wittke A, Löpprich M, Pietz J. Comparison of the developmental tests Bayley-III and Bayley-II in 7-month-old infants born preterm. Eur J Pediatr 2013; 172 (3) 393-400
  • 19 Acton BV, Biggs WS, Creighton DE , et al. Overestimating neurodevelopment using the Bayley-III after early complex cardiac surgery. Pediatrics 2011; 128 (4) e794-e800