Takashi Fujiwara1, LaDonna Malone1, Kelly Jarvis2, Kathryn C Chatfield3, Lorna P Browne1, and Alex J Barker1,4
1Department of Radiology, Children's Hospital Colorado, University of Colorado Anschutz Medical Campus, Aurora, CO, United States, 2Department of Radiology, Northwestern University, Chicago, IL, United States, 3Department of Pediatrics, University of Colorado Anschutz Medical Campus, Children’s Hospital Colorado, Aurora, CO, United States, 4Department of Bioengineering, University of Colorado Anschutz Medical Campus, Aurora, CO, United States
Synopsis
Keywords: Flow, Velocity & Flow, 4D flow MRI
Bicuspid aortic valve is associated with
progressive aortic dilatation, which is seen both in pediatric and adult populations.
Although several studies have found a connection between aortic hemodynamics,
vessel wall architectural abnormalities, and progressive dilation, the role of
hemodynamics in pediatric aortopathy remains unclear. We used 4D flow MRI data in
pediatric bicuspid aortic valve patients to identify potential hemodynamic
biomarkers associated with aortic diameter. Despite no clear association with
aortic diameter, different flow features between BAV patients with/without
coarctation were found.
Introduction
Bicuspid aortic valve (BAV) is one of the
most common congenital heart defects with a prevalence of 1-2%1. The
aorta of BAV patients is more likely to be dilated compared to the general
population, with monitoring and surgical guidelines predicated on the risk for
aortic aneurysm and dissection. Routine monitoring for progression of aortic
dilation is recommended by consensus guidelines, but no evidence or predictors
have been identified in children to predict aortic dilatation or assess risk
for dissection. 4D flow MRI (time-resolved, three-dimensional phase-contrast
MRI) is a promising tool for risk stratification in pediatric patients with BAV.
Several 4D flow studies in adult BAV patients have shown unique hemodynamic
characteristics predictive of growth or abnormal tissue biomechanics2.
Wall shear stress (WSS) is one such blood flow feature, as its close
association with elastic fiber degradation in the aorta was recently reported3.
Although progressive aneurysms are also observed in pediatric BAV patients4,
4D flow investigations in young patients are limited and possibly etiologically
different compared to adults5,6. In prior studies, WSS has not been conclusively
predictive of aortic dilatation, possibly due to small sample sizes. Another
challenge in pediatric BAV study is a wide variety of comorbidities, such as aortic
coarctation, which may result in variability in obtained hemodynamic quantities.
Here, we conduct a retrospective 4D flow analysis in pediatric/young adult BAV
patients to investigate if there are hemodynamic indicators of aortic
dilatation. We hypothesize that hemodynamic biomarkers will correlate with
aortic diameter (aortic Z-score) and comorbidities seen in BAV patients affect
hemodynamics in the aorta.Methods
This IRB-approved retrospectively study investigated 47 BAV patients who
underwent cardiac 4D flow MRI and were less than 20 years old at the time of
scan from our hospital database (mean age, 14.3 ± 4.2 years; Table 1). Patients
were scanned with Philips Ingenia (Philips Healthcare, Best, Netherlands; scan
parameters in Table 2). Patients were divided into 4 subgroups: BAV, BAV with
aortic coarctation, BAV with Turner syndrome, and BAV with both aortic
coarctation and Turner syndrome. The 4D flow MRI analysis and methods of these
patients are illustrated in Fig. 1. 4D flow MRI post-processing was performed to
correct eddy currents, mask noise, anti-alias velocity, and to create
phase-contrast MR angiography (PC-MRA) images using custom MATLAB scripts7.
Based on the PC-MRA, the aorta was segmented automatically using a deep learning
framework,8,9 followed by
manual correction. WSS, helicity, vorticity, and viscous energy loss were
computed using previously described approaches10,11. Here, 5
temporal phases centered at peak flow systole were averaged to obtain the
peak-systolic quantities and the obtained values were further averaged for
three subdomains: ascending aorta (AAo), aortic arch, and descending aorta.
Aortic pulse wave velocity was also obtained by cross-correlation approach12. Pearson correlation between the obtained hemodynamic metrics and AAo Z-score, as obtained
from medical records, were evaluated. Subgroup comparison for hemodynamic
quantities excluded two subgroups (BAV with Turner syndrome, n=6; BAV with both
aortic coarctation and Turner syndrome, n=4) due to the small number of
subjects. A Kruskal-Wallis test and post-hoc analysis with Bonferroni
correction were employed for more than three group comparison with p=0.05 as a
significance level, while an Wilcoxon rank sum test was used for two group
comparisons. Results
The BAV subgroup showed larger AAo
Z-score than those with coarctation (2.5 ± 1.6 in BAV vs. 0.8 ± 1.7 in BAV with
coarctation, p=0.03; Table 1). For the entire cohort (N=47), no significant
correlation was found between AAo Z-score and hemodynamic quantities at the AAo
(R=-0.25-0.13, p=0.09-0.84; Fig.2A-E). Overall, no strong correlation was found
between Z-score and the obtained quantities (Fig.2F). Comparison between BAV
and coarctation subgroups found significant differences in vorticity in the
aortic arch (p=0.03), helicity in the AAo (p=0.01), and viscous energy loss in the
aortic arch (p=0.04) (Table 3). Correlation analysis in each subgroups showed
different tendency (Fig.2G,H).Discussion
Correlation analysis did not find a strong
hemodynamic association with aortic Z-score, contrary to our hypothesis. The lack
of correlation between pulse wave velocity and AAo diameter suggests inherent aortic
wall property may not be a dominant factor for progressive aortic dilatation in
early stage of life; however, more study is needed given the limited temporal
resolution of 4D flow MRI. The results agree with a previous study in adults,
reporting altered wall stiffness is seen at older age, rather than adolescence13.
On the other hand, lesion-specific hemodynamic features were found; BAV
patients with coarctation had larger vorticity and viscous energy loss at the
aortic arch, and larger helicity at the AAo than those without coarctation. A previous
study found altered hemodynamics (WSS, peak velocity) at the arch and descending aorta in BAV patients with
coarctation5. Our study implies altered hemodynamics can also be
seen for the proximal aorta. Although significantly different AAo Z-score confound
these results, it suggests BAV combined with coarctation may produce unique
flow features.Conclusion
A clear association between aortic Z-score
and hemodynamic quantities were not found in this study while lesion-specific
flow features were found for the proximal aorta. Considering large variability in comorbidity and lesion-specific hemodynamic differences of these pediatric patients, proper
subgroup analysis, larger study sizes and longitudinal monitoring is necessary.Acknowledgements
NIH R01HL133504 (AJB)
NIH NIA P30AG059988 (KJ)
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