Advertisement

Pediatric Radiology

, Volume 48, Issue 12, pp 1745–1754 | Cite as

Lung and airway shape in neuroendocrine cell hyperplasia of infancy

  • Emily J. Mastej
  • Emily M. DeBoerEmail author
  • Stephen M. Humphries
  • Marlijne C. Cook
  • Kendall S. Hunter
  • Deborah R. Liptzin
  • Jason P. Weinman
  • Robin R. Deterding
Original Article
  • 389 Downloads

Abstract

Background

Neuroendocrine cell hyperplasia of infancy (NEHI) is a rare lung disease associated with significant air trapping. Although chest CT is crucial in establishing a diagnosis, CT and biopsy findings do not reveal airway abnormalities to explain the air trapping.

Objective

We compared lung and airway morphology obtained from chest CT scans in children with NEHI and control children. In the children with NEHI, we explored relationships between lung and airway shape and lung function.

Materials and methods

We performed a retrospective review of children with NEHI who underwent clinical chest CT. We identified control children of similar size and age. We created lung masks and airway skeletons using semi-automated software and compared them using statistical shape modeling methods. Then we calculated a logistic regression model using lung and airway shape to differentiate NEHI from controls, and we compared shape model parameters to lung function measurements.

Results

Airway and lung shapes were statistically different between children with NEHI and controls. We noted a broad lung apex in the children with NEHI and a significantly increased apical anterior–posterior lung diameter. A logistic regression model including lung shape was 90% accurate in differentiating children with NEHI from controls. Correlation coefficients were significant between lung function values and lung and airway shape.

Conclusion

Lung and airway shapes were different between children with NEHI and control children in this cohort. Children with NEHI had an increased anteroposterior diameter of their lungs that might be useful in the diagnostic criteria.

Keywords

Children Computed tomography Interstitial lung disease Lungs Neuroendocrine cell hyperplasia of infancy Shape modeling 

Notes

Acknowledgments

The authors would like to acknowledge the help of Elin Towler and Brandie Wagner in collecting and standardizing the data. ShapeWorks is provided online through support by the National Institute of General Medical Sciences of the National Institutes of Health under grant number P41 GM103545-18.

Compliance with ethical standards

Conflicts of interest

None

Supplementary material

247_2018_4189_MOESM1_ESM.docx (116 kb)
ESM 1 (DOCX 115 kb)

References

  1. 1.
    Deterding RR (2010) Infants and young children with children's interstitial lung disease. Pediatr Allergy Immunol Pulmonol 23:25–31CrossRefGoogle Scholar
  2. 2.
    Deterding RR, Fan LL, Morton R et al (2001) Persistent tachypnea of infancy (PTI) — a new entity. Pediatr Pulmonol 23:72–73CrossRefGoogle Scholar
  3. 3.
    Fan LL, Deterding RR, Langston C (2004) Pediatric interstitial lung disease revisited. Pediatr Pulmonol 38:369–378CrossRefGoogle Scholar
  4. 4.
    Deutsch GH, Young LR, Deterding RR et al (2007) Diffuse lung disease in young children: application of a novel classification scheme. Am J Respir Crit Care Med 176:1120–1128CrossRefGoogle Scholar
  5. 5.
    Kuo CS, Young LR (2014) Interstitial lung disease in children. Curr Opin Pediatr 26:320–327CrossRefGoogle Scholar
  6. 6.
    Deterding RR, Pye C, Fan LL et al (2005) Persistent tachypnea of infancy is associated with neuroendocrine cell hyperplasia. Pediatr Pulmonol 40:157–165CrossRefGoogle Scholar
  7. 7.
    Brody AS, Guillerman RP, Hay TC et al (2010) Neuroendocrine cell hyperplasia of infancy: diagnosis with high-resolution CT. AJR Am J Roentgenol 194:238–244CrossRefGoogle Scholar
  8. 8.
    Gomes VC, Silva MC, Maia Filho JH et al (2013) Diagnostic criteria and follow-up in neuroendocrine cell hyperplasia of infancy: a case series. J Bras Pneumol 39:569–578CrossRefGoogle Scholar
  9. 9.
    Houin PR, Deterding RR, Young LR (2016) Exacerbations in neuroendocrine cell hyperplasia of infancy are characterized by increased air trapping. Pediatr Pulmonol 51:E9–12CrossRefGoogle Scholar
  10. 10.
    Young LR, Brody AS, Inge TH et al (2011) Neuroendocrine cell distribution and frequency distinguish neuroendocrine cell hyperplasia of infancy from other pulmonary disorders. Chest 139:1060–1071CrossRefGoogle Scholar
  11. 11.
    Soares JJ, Deutsch GH, Moore PE et al (2013) Childhood interstitial lung diseases: an 18-year retrospective analysis. Pediatrics 132:684–691CrossRefGoogle Scholar
  12. 12.
    Humphries SM, Hunter KS, Shandas R et al (2016) Analysis of pediatric airway morphology using statistical shape modeling. Med Biol Eng Comput 54:899–911CrossRefGoogle Scholar
  13. 13.
    Kerby GS, Wagner BD, Popler J et al (2013) Abnormal infant pulmonary function in young children with neuroendocrine cell hyperplasia of infancy. Pediatr Pulmonol 48:1008–1015CrossRefGoogle Scholar
  14. 14.
    Stocks J, Godfrey S, Beardsmore C et al (2001) Plethysmographic measurements of lung volume and airway resistance. ERS/ATS task force on standards for infant respiratory function testing. European Respiratory Society/American Thoracic Society. Eur Respir J 17:302–312CrossRefGoogle Scholar
  15. 15.
    Jones M, Castile R, Davis S et al (2000) Forced expiratory flows and volumes in infants. Normative data and lung growth. Am J Respir Crit Care Med 161:353–359CrossRefGoogle Scholar
  16. 16.
    Cates J, Fletcher PT, Styner M et al (2008) Particle-based shape analysis of multi-object complexes. Med Image Comput Comput Assist Interv 11:477–485PubMedPubMedCentralGoogle Scholar
  17. 17.
    Yushkevich PA, Piven J, Hazlett HC et al (2006) User-guided 3D active contour segmentation of anatomical structures: significantly improved efficiency and reliability. Neuroimage 31:1116–1128CrossRefGoogle Scholar

Copyright information

© Springer-Verlag GmbH Germany, part of Springer Nature 2018

Authors and Affiliations

  • Emily J. Mastej
    • 1
  • Emily M. DeBoer
    • 1
    Email author
  • Stephen M. Humphries
    • 2
  • Marlijne C. Cook
    • 3
  • Kendall S. Hunter
    • 3
  • Deborah R. Liptzin
    • 1
  • Jason P. Weinman
    • 4
  • Robin R. Deterding
    • 1
  1. 1.Department of Pediatrics and Breathing Institute, Children’s Hospital ColoradoUniversity of Colorado Anschutz Medical CampusAuroraUSA
  2. 2.Department of RadiologyNational Jewish HealthDenverUSA
  3. 3.Department of BioengineeringUniversity of Colorado Anschutz Medical CampusAuroraUSA
  4. 4.Department of Radiology, Children’s Hospital ColoradoUniversity of Colorado Anschutz Medical CampusAuroraUSA

Personalised recommendations