Abstract
Objective
The long-term morbidity among children with severe bronchopulmonary dysplasia who require tracheostomy (tBPD) relative to those without tracheostomy (sBPD) is not well characterized. We compared childhood lung function and neurodevelopmental outcomes in tBPD and sBPD.
Study design
Retrospective case-control study of N = 49 tBPD and N = 280 sBPD subjects in Boston Children’s Hospital Preterm Lung Patient Registry and medical record. We compared NICU course, childhood spirometry, and neurodevelopmental testing.
Result
tBPD subjects were more likely than sBPD to be Black, have pulmonary hypertension, and have subglottic stenosis. tBPD subjects had lower maximal childhood FEV1 % predicted (β = −0.14) and FEV1/FVC (β = −0.08); spirometry curves were more likely to suggest fixed extrathoracic obstruction. tBPD subjects had greater cognitive and motor delays <24 months, and greater cognitive delays >24 months.
Conclusion
Compared to subjects with sBPD who did not require tracheostomy, tBPD subjects suffer from increased long-term impairment in respiratory function and neurodevelopment.
This is a preview of subscription content, access via your institution
Access options
Subscribe to this journal
Receive 12 print issues and online access
$259.00 per year
only $21.58 per issue
Buy this article
- Purchase on SpringerLink
- Instant access to the full article PDF.
USD 39.95
Prices may be subject to local taxes which are calculated during checkout

Similar content being viewed by others
References
Abman SH, Collaco JM, Shepherd EG, Keszler M, Cuevas-Guaman M, Welty SE, et al. Interdisciplinary care of children with severe bronchopulmonary dysplasia. J Pediatr. 2017;181:12–28. e11
Sakai M, Kou YF, Shah GB, Johnson RF. Tracheostomy demographics and outcomes among pediatric patients ages 18 years or younger-United States 2012. Laryngoscope. 2019;129:1706–11.
Koltsida G, Konstantinopoulou S. Long term outcomes in chronic lung disease requiring tracheostomy and T chronic mechanical ventilation. Semin Fetal Neonatal Med. 2019;24:101044.
Pereira KD, MacGregor AR, McDuffie CM, Mitchell RB. Tracheostomy in preterm infants: current trends. Arch Otolaryngol Head Neck Surg. 2003;129:1268–71.
Higgins RD, Jobe AH, Koso-Thomas M, Bancalari E, Viscardi RM, Hartert TV, et al. Bronchopulmonary dysplasia: executive summary of a workshop. J Pediatr. 2018;197:300–8.
Wu KY, Jensen EA, White AM, Wang Y, Biko DM, Nilan K, et al. Characterization of disease phenotype in very preterm infants with severe bronchopulmonary dysplasia. Am J Respir Crit Care Med. 2020;201:1398–406.
Keller RL, Feng R, DeMauro SB, Ferkol T, Hardie W, Rogers EE, et al. Bronchopulmonary dysplasia and perinatal characteristics predict 1-year respiratory outcomes in newborns born at extremely low gestational age: a prospective cohort study. J Pediatr. 2017;187:89–97. e83
Fawke J, Lum S, Kirkby J, Hennessy E, Marlow N, Rowell V, et al. Lung function and respiratory symptoms at 11 years in children born extremely preterm: the EPICure study. Am J Respir Crit Care Med. 2010;182:237–45.
Vom Hove M, Prenzel F, Uhlig H, Robel-Tillig E. Pulmonary outcome in former preterm, very low birth weight children with bronchopulmonary dysplasia: a case-control follow-up at school age. J Pediatr. 2014;164:40–45. e44
Verheggen M, Wilson A, Pillow J, Stick S, Hall G. Respiratory function and symptoms in young preterm children in the contemporary era. Pediatr Pulmonol. 2016;51:1347–55.
Kaslovsky R, Sadof M. Spirometry for the primary care pediatrician. Pediatrics Rev. 2014;35:465–75.
Brumbaugh JE, Bell EF, Grey SF, DeMauro SB, Vohr BR, Harmon HM, et al. Behavior profiles at 2 years for children born extremely preterm with bronchopulmonary dysplasia. J Pediatr. 2020;219:152–9.
Cammack B, Noel-MacDonnell J, Cuna A, Manimtim W. Impact of tracheostomy on language and cognitive development in infants with severe bronchopulmonary dysplasia. J Perinatol. 2020;40:299–305.
Cristea AI, Carroll AE, Davis SD, Swigonski NL, Ackerman VL. Outcomes of children with severe bronchopulmonary dysplasia who were ventilator dependent at home. Pediatrics. 2013;132:e727–734.
Levin JC, Sheils CA, Gaffin JM, Hersh CP, Rhein LM, Hayden LP. Lung function trajectories in former premature infants: identifying risk factors for abnormal lung growth. Respir Res. 2021;22:143.
Goryachev S, Sordo M, Zeng QT. A suite of natural language processing tools developed for the I2B2 project. AMIA Annu Symp Proc. 2006;2006:931.
Jobe AH, Bancalari E. Bronchopulmonary dysplasia. Am J Respir Crit Care Med. 2001;163:1723–9.
HoundDog. Research computing. Boston: Boston Children’s Hospital; 2018.
Miller MR, Hankinson J, Brusasco V, Burgos F, Casaburi R, Coates A, et al. Standardisation of spirometry. Eur Respir J. 2005;26:319–38.
Quanjer PH, Stanojevic S, Cole TJ, Baur X, Hall GL, Culver BH, et al. Multi-ethnic reference values for spirometry for the 3-95-yr age range: the global lung function 2012 equations. Eur Respir J. 2012;40:1324–43.
Bayley N. Bayley scales of infant and toddler development. 3rd ed. San Antonio, TX: Harcourt Assessment; 2006.
Corp. I IBM SPSS Statistics for Macintosh. 26.0 ed. Armonk, NY: IBM Corp.; 2018.
Carron JD, Derkay CS, Strope GL, Nosonchuk JE, Darrow DH. Pediatric tracheotomies: changing indications and outcomes. Laryngoscope. 2000;110:1099–104.
Levit OL, Shabanova V, Bazzy-Asaad A, Bizzarro MJ, Bhandari V. Risk factors for tracheostomy requirement in extremely low birth weight infants. J Matern-Fetal Neonatal Med. 2018;31:447–52.
Grundfast KM, Felizardo S, Camilon J, Barber CS, Pransky S, Fink R. Prospective study of subglottic stenosis in intubated neonates. Ann Otol Rhinol Laryngol. 1990;99:390–5.
Lee JH, Smith PB, Quek MBH, Laughon MM, Clark RH, Hornik CP. Risk factors and in-hospital outcomes following tracheostomy in infants. J Pediatr. 2016;173:39–44.
Lewis CW, Carron JD, Perkins JA, Sie KCY, Feudtner C. Tracheotomy in pediatric patients: a national perspective. Arch Otolaryngol Head Neck Surg. 2003;129:523–9.
Stark AR, Eichenwald EC. Bronchopulmonary dysplasia: management. UpToDate 2019 February 7, 2019 [cited 2020 June 18]. Available from: https://www.uptodate.com.
Guaman MC, Gien J, Baker CD, Zhang H, Austin ED, Collaco JM. Point prevalence, clinical characteristics, and treatment variation for infants with severe bronchopulmonary dysplasia. Am J Perinatol. 2015;32:960–7.
Thoma ME, Drew LB, Hirai AH, Kim TY, Fenelon A, Shenassa ED. Black-White disparities in preterm birth: geographic, social, and health determinants. Am J Prev Med. 2019;57:675–86.
Kavvadia V, Greenough A, Dimitriou G, Hooper R. Influence of ethnic origin on respiratory distress syndrome in very premature infants. Arch Dis Child Fetal Neonatal Ed. 1998;78:F25–28.
Ryan RM, Feng R, Bazacliu C, Ferkol TW, Ren CL, Mariani TJ, et al. Black race is associated with a lower risk of bronchopulmonary dysplasia. J Pediatr. 2019;207:130–5. e132
Han SM, Watters KF, Hong CR, Edwards EM, Knell J, Morrow KA, et al. Tracheostomy in very low birth weight infants: a prospective multicenter study. Pediatrics. 2020;145:e20192371.
Sigurdson K, Mitchell B, Liu J, Morton C, Gould JB, Lee HC, et al. Racial/ethnic disparities in neonatal intensive care: a systematic review. Pediatrics. 2019;144:e20183114.
Cristea AI, Ackerman VL, Swigonski NL, Yu Z, Slaven JE, Davis SD. Physiologic findings in children previously ventilator dependent at home due to bronchopulmonary dysplasia. Pediatr Pulmonol. 2015;50:1113–8.
Contecin P, Narcy P. Size of endotracheal tube and neonatal acquired subglottic stenosis. Arch Otolaryngol Head Neck Surg. 1993;119:815–9.
Ehrenkranz RA, Walsh MC, Vohr BR, Jobe AH, Wright LL, Fanaroff AA, et al. Validation of the National Institutes of Health consensus definition of bronchopulmonary dysplasia. Pediatrics. 2005;116:1353–60.
Doyle LW, Davis PG, Morley CJ, McPhee A, Carlin JB. Outcome at 2 years of age of infants from the DART study: a multicenter, international, randomized, controlled trial of low-dose dexamethasone. Pediatrics. 2007;119:716–21.
Litt JS, Minich N, Taylor HG, Tiemeier H. The inter-relationships of extremely low birth weight, asthma, and behavior: a study of common cause, mediation, and moderation. Acad Pediatr. 2020;20:975–82.
Acknowledgements
Lawrence M. Rhein, MD MPH, Department of Pediatrics, University of Massachusetts, Worcester, MA, United States.
Funding
Supported by the National Institutes of Health (NHLBI K23 HL136851 [to L.P.H.], NICHD K23 HD088695 [to J.S.L.], NLM T15LM007092 [to N. Gehlenborg])
Author information
Authors and Affiliations
Contributions
C.A.A. worked on the design of the study, conducted the analysis and drafted the initial manuscript. J.C.L. aided with the conceptualization of the study, assisted with statistical analysis, and reviewed the manuscript. J.S.L. helped design the methodology and reviewed the manuscript. C.A.S. aided with investigation, provided resources, and reviewed the manuscript. L.P.H. provided supervision for the design, methodology, and analysis and provided multiple manuscript revisions.
Corresponding author
Ethics declarations
Competing interests
The authors declare no competing interests.
Additional information
Publisher’s note Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Rights and permissions
About this article
Cite this article
Annesi, C.A., Levin, J.C., Litt, J.S. et al. Long-term respiratory and developmental outcomes in children with bronchopulmonary dysplasia and history of tracheostomy. J Perinatol 41, 2645–2650 (2021). https://doi.org/10.1038/s41372-021-01144-0
Received:
Revised:
Accepted:
Published:
Version of record:
Issue date:
DOI: https://doi.org/10.1038/s41372-021-01144-0
This article is cited by
-
Neonatal multimorbidity and the phenotype of premature aging in preterm infants
Pediatric Research (2025)
-
The impact of early tracheostomy on neurodevelopmental outcomes of infants with severe bronchopulmonary dysplasia exposed to postnatal corticosteroids
Journal of Perinatology (2024)
-
In-hospital mortality and length of hospital stay in infants requiring tracheostomy with bronchopulmonary dysplasia
Journal of Perinatology (2024)


