Nara S. Higano1,2, Alister J. Bates1,3, Erik B. Hysinger4, Robert J. Fleck5, Andrew D. Hahn6, Sean B. Fain6,7, Paul S. Kingma8, and Jason C. Woods1,2,4,5
1Center for Pulmonary Imaging Research, Cincinnati Children's Hospital, Cincinnati, OH, United States, 2Physics, Washington University in St. Louis, St. Louis, MO, United States, 3Upper Airway Center, Cincinnati Children's Hospital, Cincinnati, OH, United States, 4Pulmonary Medicine, Cincinnati Children's Hospital, Cincinnati, OH, United States, 5Radiology, Cincinnati Children's Hospital, Cincinnati, OH, United States, 6Medical Physics, University of Wisconsin - Madison, Madison, WI, United States, 7Radiology, University of Wisconsin - Madison, Madison, WI, United States, 8Neonatology and Pulmonary Biology, Cincinnati Children's Hospital, Cincinnati, OH, United States
Synopsis
Neonatal airway malacia (dynamic larynx,
trachea, and/or bronchi collapse) is a common airway complication often associated
with preterm birth and congenital abnormalities but has not been extensively
studied. This condition is currently diagnosed through visual bronchoscopy,
which can be unreliable and poses increased risks to patients. We address these
issues with an innovative technique using retrospectively respiratory-gated
ultrashort echo time MRI and geometric analysis of moving airway anatomy for
regional, quantitative evaluation of dynamic airway collapse in quiet-breathing,
non-sedated neonates. This method has the potential to yield more accurate and
objective assessment of neonatal airway collapse than current techniques allow.
Purpose
Airway collapse (laryngomalacia [LM], tracheomalacia
[TM], or bronchomalacia [BM]) is a common airway complication occurring in infants
with bronchopulmonary dysplasia (BPD, lung disease of prematurity), congenital
diaphragmatic hernia (CDH), or esophageal atresia/tracheoesophageal fistula (EA/TEF)1-6, but the underlying pathology, impact on
clinical outcome, and response to therapy are not well understood. Infants with
airway abnormalities typically face longer hospitalizations and increased
medical comorbidities due to increased airway compliance during respiration5-7.
The clinical diagnostic standard for collapse assessment is bronchoscopy, which
has multiple potential limitations: only moderate inter-reader reliability; inconsistency
between rigid and flexible scopes; lack of quantitative evaluation; and invasiveness,
posing increased risk to patients. Computed tomography (CT) can be used to
visualize collapse but has a time resolution limited by the gantry rotation and
exposes infants to ionizing radiation. Further, both bronchoscopy and CT typically
require sedation, which can obscure the airway dynamics under evaluation. In
this study, we demonstrate the first dynamic 3D MR images of the neonatal
airway and develop a novel method for non-invasive quantitative regional assessment
of neonatal dynamic airway collapse under restful breathing conditions via
retrospectively respiratory-gated ultrashort echo time (UTE) MRI, without sedation
or ionizing radiation.Methods
Neonatal 1H 3D
radial UTE MR images8-9 were acquired during restful breathing in 14
neonatal subjects recruited from the NICU (40±2 weeks mean post-menstrual age)
who also had clinical bronchoscopy (5 BPD, 4 CDH, 4 EA/TEF, 1 premature non-BPD
with abnormal sleep study) using a quadrature body coil on a neonatal-sized,
NICU-sited 1.5T scanner10-11, with no sedation administered for
scanning. Expanding upon previous techniques12 using the time-course
of k0 as a retrospective respiratory-gating waveform, UTE images
were binned into eight frames throughout the respiratory cycle (Figure-1). Typical UTE parameters were:
TR/TE≈5/0.2ms; FA=5º; FOV=18cm; number of radial projections=200,000; 3D
isotropic resolution≈0.7 mm; and scan time≈16 min.
End-expiration airway anatomy was
segmented from UTE images via ITK-SNAP (3.6.0, Penn Image Computing and Science
Laboratory, USA)13. Airway motion during breathing was generated via
image registration of respiratory-gated images14, yielding surface
renderings throughout respiration (Figure-2). A center-line was defined through each respiratory-gated surface
rendering via VMTK 1.2 (Orobix, Bergamo, Italy) to account for airway
curvature and misalignment with axes15, and a series of disks
bounded by the airway rendering and orthogonal to the center-line were defined
at 1-mm intervals (Figure-3). The cross-sectional area (CSA) of each disk was calculated
regionally along the airway, with degree of collapse defined here as:
$$Z_{collapse}=1-\frac{CSA_{end-expiration}}{CSA_{end-inspiration}}$$
such that Zcollapse=0
with no dynamic collapse and Zcollapse=1
with complete dynamic collapse, under conventional assumptions of intrathoracic
collapse during expiration. We define presence of dynamic collapse when Zcollapsemax>0.25 at any point along the airway.
Results
Dynamic airway collapse was quantified
regionally in all cases, with collapse identified in 12 patients (Zcollapsemax>0.25) and absent in 2 patients (Zcollapsemax<0.25). Representative MRI results from two patients
with collapse (both severe BPD) are presented, with qualitative comparison to
clinical bronchoscopy. Subject A (Figure-4) demonstrated partial dynamic collapse in the middle and lower trachea
(TM) that progressively worsened during exhalation and reached maximum collapse
at end-expiration (maximum Zcollapse≈0.5). Subject B (Figure-5) demonstrated little collapse through the trachea (Zcollapse≈0-0.4), partial collapse above the larynx (Zcollapse≈0-0.4), and complete airway collapse (Zcollapse=1) in the larynx (LM),
occurring dynamically at end-expiration only. These results regionally agree
with qualitative findings from bronchoscopy.
Discussion and Conclusions
This
innovative MRI technique
provides a new platform for quantitative and regional assessment
of dynamic airway morbidities in neonates during restful breathing, without
requiring sedation, invasive procedures, or ionizing radiation, and qualitatively
compares well to clinical bronchoscopy. While
rigorous validation with bronchoscopic findings is required to establish
accuracy, this method overcomes the current barriers associated with existing
diagnostic standards, yielding more objective and quantitative neonatal airway biomarkers
than currently possible. Further, this technique poses no additional risks to
patients and so can be used in serial assessment to guide use of pharmacologic
interventions for airway collapse, such as albuterol. In the future, neonatal
dynamic airway imaging may be used in conjunction with computational fluid
dynamics (CFD) models14 to calculate respiratory effort and predict
increased energy expenditure in infants related to airway collapse.
Looking
forward, regional and dynamic biomarkers from geometric analysis and CFD
simulations may be associated with clinical outcomes, which would allow for
direct translation to the clinical care of neonatal patients with airway
complications. This may have particular impact with BPD, for which the
connection between pulmonary and airway disease is not yet well characterized. Ultimately,
this approach has the potential to significantly advance understanding and
treatment of airway morbidities and trajectories in this vulnerable neonatal population.
Acknowledgements
The
authors thank The Perinatal Institute at Cincinnati Children’s Hospital Medical
Center, The Hartwell Foundation, NIH P01 HD093363, and The Departments of Radiology and
Medical Physics and the School of Medicine and Public Health at University of Wisconson-Madison
for research funding and support.References
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