REVIEW ARTICLE
Imaging of Pulmonary Atresia With Ventricular Septal Defect
Ahmed Abdel Khalek Abdel Razek, MD,* Hala Al-Marsafawy, MD,† and Maha Elmansy, MD*
Abstract: The aim of this article was to review computed tomography an-
giography and magnetic resonance angiography of pulmonary atresia with
ventricular septal defect. This disorder is a rare complex congenital heart
disease. Preoperative imaging of pulmonary atresia with ventricular septal
defect with computed tomography angiography and magnetic resonance
angiography is important for complete anatomical delineation and planning
for treatment. Preoperative imaging used for assessment of the main pul-
monary artery (its size, valve, and confluence), aortopulmonary collaterals
(its origin, insertion, course, and size), presence of patent ductus arteriosus,
other sources of collaterals as bronchial and coronary arteries, and pattern
Downloaded from http://journals.lww.com/jcat by BhDMf5ePHKbH4TTImqenVIu1/FN/J4kOqexKRYD0zCVnufb9bsK6z4I6kqFpaMMQ on 01/18/2020
of pulmonary arborization. Imaging can detect associated aortic, pulmo-
nary venous and coronary anomalies, and other congenital heart disease.
Postoperative imaging after unifocalization and stent is for assessment of
patency, stenosis, and occlusion of stent or perivascular lesions as seroma.
Key Words: pulmonary atresia, MR angiography, CT angiography,
congenital, cardiac
(J Comput Assist Tomogr 2019;43: 906–911)
P ulmonary atresia with ventricular septal defect (PA-VSD) is a
rare complex cyanotic congenital heart disease that associated
with high rate of mortality and morbidity. Pulmonary atresia
means an undeveloped pulmonary valve or pulmonary artery.
As the pulmonary valve is completely closed, the blood flow from
the right ventricle to the PA is obstructed.1–3 The central pulmonary
artery may be absent or present and the branch pulmonary arteries
may be confluent or nonconfluent. The lungs can derive their blood
supply from various sources including aortopulmonary collaterals
(APCs), patent ductus arteriosus (PDA), and other. It is important
to map the source of supply to each part of the lung. The septal de-
fects in these patients are large subaortic VSD, which is usually of
membranous type.3–6
METHODS OF EXAMINATION
Echocardiography
Echocardiography is a good modality for both the anatomical
evaluation of the central pulmonary arteries and functional assess-
ment of the right and left ventricles, and estimation of pressure
gradients through right ventricular outflow tract (RVOT), but it
is limited in evaluation of pulmonary arties and pulmonary vascu-
lature by poor acoustic windows and it also does not provide the
surgeon with a precise anatomic road map.3–5
From the *Department of Diagnostic Radiology, Mansoura Faculty of Medicine;
and †Pediatric Cardiology, Pediatric Hospital, Mansoura Faculty of medicine,
Mansoura, Egypt.
Received for publication August 19, 2019; accepted August 26, 2019.
Correspondence to: Ahmed Abdel Khalek Abdel Razek, MD, Department of
Diagnostic Radiology, Mansoura Faculty of Medicine, Elgomheria St,
Mansoura, DK, Egypt 13351 (e‐mail: arazek@mans.edu.eg).
The authors declare no conflict of interest.
Copyright © 2019 Wolters Kluwer Health, Inc. All rights reserved.
DOI: 10.1097/RCT.0000000000000938
906 www.jcat.org J Comput Assist Tomogr • Volume 43, Number 6, November/December 2019
Copyright © 2019 Wolters Kluwer Health, Inc. All rights reserved.
Computed Tomography Angiography
Computed tomography angiography (CTA) is capable of
accurate assessment of the pulmonary vascularity, the intracar-
diac abnormalities, and pulmonary collateral vessels as PDA or
APCs.6,7 It has the potential to replace conventional angiogra-
phy for the noninvasive evaluation of infants with PA-VSD.
The merits of CTA are high temporal and spatial resolution,
short scan times, decreased need for anesthesia, and generation
of high-quality 2- and 3-dimensional images.6–10
Magnetic Resonance Angiography
Contrast magnetic resonance angiography (MRA) is good in
delineation of the pulmonary arteries anatomy, PDA, and APCs. It
is capable of estimation of gradient through stenosed vessels. Time-
resolved MRA improved image quality of contrast MRA.11–15
Cardiac Catheterization
Cardiac catheterization is the criterion standard for evalua-
tion of infants with PA-VSD, but it has limitations, because it is
an invasive method with hazards of radiation exposure and com-
plications of iodinated contrast media.1,2
PREOPERATIVE ASSESSMENT OF PA-VSD
Table 1 shows the role of imaging in preoperative assessment
of infants with PA-VSD.
Pulmonary Arteries
The most important step in preoperative evaluation in cases
of PA-VSD is the anatomical evaluation of the pulmonary artery.
Pulmonary atresia defined as absence of luminal continuity and
blood flow through the pulmonary artery, which is the hallmark
of PA-VSD. Computed tomography angiography and MRA are
accurate tools in assessment of pulmonary artery and can evaluate
length of the pulmonary atresia; presence of pulmonary artery
confluence; and size of main, right, and left pulmonary arteries
at the origin and at the hilum (Figs. 1–3). Analysis of the pulmo-
nary artery anatomy is the single most important aspect of diag-
nostic imaging of patients with pulmonary atresia. The surgical
treatment is determined by the morphology of the pulmonary ar-
teries. Pulmonary artery size may affect the conduct of surgery
if augmentation of the pulmonary arteries is necessary, and it
may be a predictor of successful outcome. Pulmonary artery ab-
normalities may be severe, such as atresia and hypoplasia, or mild,
such as isolated stenosis. Pulmonary artery hypoplasia may be as-
sociated with unilateral or segmental hypoplasia of the lungs.16–19
Aortopulmonary Collaterals
Aortopulmonary collaterals represent fetal primitive inter-
segmental arteries that originate from the descending aorta and
have not been involuted. They help in pulmonary blood supply
in approximately 30% to 40% of patients with PA-VSD, and
sometimes, they are the only source for blood supply. They usu-
ally arise from the descending thoracic aorta but may from the
subclavian or coronary arteries, and abdominal aorta and anasto-
mose with the intrapulmonary arteries usually occurs close to
J Comput Assist Tomogr • Volume 43, Number 6, November/December 2019 Imaging of PA-VSD

TABLE 1. Role of Preoperative Imaging of PA-VSD

Pulmonary artery
Size
Confluence
Valve
APC
Size
Number
Origin FIGURE 2. Magnetic resonance angiography of hypoplastic
Insertion pulmonary artery in PA-VSD. A, Hypoplastic right pulmonary
Course artery (black arrow) with interruption of the left pulmonary artery
(white arrow). B, Nonconfluent hypoplastic pulmonary arteries
Stenosis
(asterisks).
Relationship between APCs
and tracheobronchial tree number, origin, course, distribution, stenosis, and the relationship
PDA with the tracheobronchial tree, so it helps in planning for surgical
Presence treatment (Figs. 4, 5).20–26
Location
Size Patent Ductus Arteriosus
Relationship to
Patent ductus arteriosus is a fetal connection between the dis-
adjacent structures
tal arch of aorta, and left pulmonary artery may be present in cases
Bronchial arteries
of PA-VSD and contributes to pulmonary blood supply. Com-
Origin puted tomography angiography helps in accurate evaluation of
Size its site, size, calcification, and relation to surrounding structures.
Lung arborization Multiplanar reformatted images are very important in diagnosis
VSD and assessment of PDA. Magnetic resonance angiography sagittal
Size images also can optimally visualize PDA (Figs. 6, 7). When a
Type PDA connects with a central pulmonary artery, the peripheral dis-
Associated anomalies tribution is normal, and there are no aortic pulmonary collaterals
Aortic arch in the hemithorax. Thus, a PDA does not coexist with collaterals
in the same lung.27
Pulmonary veins
Systemic veins
Coronary arteries Bronchial Arteries and Other Collaterals
Extracardiac Most infants with PA-VSD have a diffusely enlarged network
Situs anomalies of bronchial arteries providing a nondiscrete source of the pulmo-
nary circulation. They are usually smaller and more numerous
than APCs. Other sources of pulmonary blood supply also may in-
clude the paramediastinal collateral arteries and the intercostals
the hilum.12,13 In long-standing cases, aneurysmal dilatation or collateral arteries.21–24
stenosis of APCs can occur. The presence of stenosis gives an in-
direct idea that the pulmonary vasculature is protected from future
pulmonary hypertension. Computed tomography angiography Lung Arborization (Distribution)
and MRA are accurate for assessment of APCs with regard to their Preoperative imaging of PA-VSD should include the source
and distribution of blood supply and the extent of distal arboriza-
tion. Aortopulmonary collaterals may supply lung segments not in
communication with branch pulmonary arteries. This information
should be provided to cardiologist, the surgeon, and the intensive
care team, which is essential to optimize care.28,29

FIGURE 1. Computed tomography angiography of hypoplastic

pulmonary artery in PA-VSD. A, Axial image showing large VSD FIGURE 3. Computed tomography angiography of PA-VSD with
(asterisk). B, Three-dimensional volume rendered reconstructed image pulmonary artery atresia. A, Three-dimensional volume rendered
showing atresia of the main pulmonary artery (MPA) (arrow). image. B, Two-dimensional axial image showing atresia of the MPA
Figure 1 can be viewed online in color at www.jcat.org. (arrows). Figure 3 can be viewed online in color at www.jcat.org.

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Abdel Razek et al J Comput Assist Tomogr • Volume 43, Number 6, November/December 2019
FIGURE 4. Computed tomography angiography of an APC in
PA-VSD coronal MIP reformatted image of CTA showing major
collateral artery (arrow) seen arising from the descending thoracic
aorta (A) to supply the right pulmonary artery in a case of
tetralogy of Fallot with pulmonary atresia.
Ventricular Septal Defect
The VSD is typically a large, perimembranous, subaortic de-
fect; less commonly, the defect may be subpulmonic that may be
associated with ostium secundum–type atrial septal defect or pat-
ent foramen ovale.1–3
Associated Anomalies
- Aortic anomalies include right-sided aortic arch, aberrant
right or left subclavian artery, and coarctation of the aorta and
double aortic arch.30
- Pulmonary venous anomalies include partial and total
anomalous pulmonary venous drainage.31,32
- Systemic venous anomalies include left-sided superior vena
cava and interrupted inferior vena cava.33
FIGURE 5. Computed tomography angiography of APC in PA-VSD
(A) sagittal reformatted image showing multiple major collaterals
(asterisks) arising from overriding aorta (AO). B, Three-dimensional
volume rendered image delineating those large collateral arteries
in a case of tetralogy of Fallot with atretic pulmonary valve. Figure 5
can be viewed online in color at www.jcat.org.
908 www.jcat.org
Copyright © 2019 Wolters Kluwer Health, Inc. All rights reserved.
FIGURE 6. Computed tomography angiography of PDA in PA-VSD.
A, Sagittal reformatted image showing large PDA (asterisk) arising
from the under surface of the aortic arch (A) to the left pulmonary
artery (P). B, Three-dimensional volume rendered image showing
the PDA (asterisk) and the atretic MPA (white arrow). Figure 6 can be
viewed online in color at www.jcat.org.
- Coronary anomalies include left anterior descending artery
originating from the right coronary, and coronary artery to
pulmonary-artery fistula.34,35
- Congenital heart disease include double outlet right ventricle,
double outlet left ventricle, and transposition of great arteries.3–6
- Situs anomalies with asplenia, central liver, right-sided
stomach, and situs inversus.20–22
- Extracardiac: trachea-esophageal fistula and scoliosis may
be reported.
POSTOPERATIVE ASSESSMENT OF PA-VSD
Treatment of PA-VSD is challenging and controversial. The
surgical treatment of PA-VSD is variable and depends on patient's
age, the degree of cyanosis, the extent of RVOT obstruction, size
of the VSD, the presence and size of pulmonary arteries, and asso-
ciated findings. If there are multiple APCs, multistage or single-
stage rehabilitation of pulmonary arteries with unifocalization is
needed before intracardiac repair. In cases of large pulmonary
FIGURE 7. Magnetic resonance angiography of PDA in PA-VSD
source image of MRA shows large PDA (asterisk) connecting the
aorta (A) with the left pulmonary artery (P).
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J Comput Assist Tomogr • Volume 43, Number 6, November/December 2019
TABLE 2. Postoperative Complications of PA-VSD
1. Unifocalization:
APCs stump aneurysm, anastomotic stenosis of fine APCs
Pulmonary hemorrhage, edema
2. Shunts:
Shunt thrombosis, stenosis
Major or branch pulmonary artery stenosis
Seroma around the graft
3. RVOT patches:
Right ventricle -to-PA conduit: stenosis or regurgitation
RVOT patch: patch aneurysm, pulmonary stenosis
4. Glenn operation:
Narrowing of Glenn anastomotic site.
Stenosis of inferior vena cava-to-pulmonary artery conduit
FIGURE 8. Magnetic resonance angiography of shunt in PA-VSD. A,
Hypoplastic both pulmonary arteries (arrows) with patent left
subclavian pulmonary shunt (asterisk). B, Small multiple collaterals
arising from the left lung to supply the lungs (arrows).
arteries without ABCs, palliative procedures as aortopulmonary
shunts in younger children and corrective surgery (RVOT patch)
in older children can be done after closure of VSD. Imaging of
postoperative PA-VSD is modified by complicated anatomy with
surgical intervention, and interference from the frequently pre-
sents conduits, baffles, stents, and coils.35–43 Table 2 show the
types of surgery and complications in PA-VSD.
FIGURE 9. Computed tomography angiography of PDA stent in patient with PA with APCs and right-sided aortic arch. A, Axial image showing
atretic MPA (arrow). B, Coronal reformatted image showing PDA stent (arrows) and right-sided aortic arch (A) before crossing of descending
aorta to the left side. C, Three-dimensional volume rendering image delineating large PDA (asterisks) connecting left brachiocephalic artery to
the left PA and multiple APCs (arrows). Figure 9 can be viewed online in color at www.jcat.org.
© 2019 Wolters Kluwer Health, Inc. All rights reserved.
Copyright © 2019 Wolters Kluwer Health, Inc. All rights reserved.
Imaging of PA-VSD
Unifocalization
Unifocalization is a surgical procedure that eliminates extra
cardiac sources of pulmonary arterial blood flow and coalition
of largest possible pulmonary arterial segments so that they even-
tually can be incorporated into the RVOT. Complications include
pulmonary hemorrhage, pleural effusion, stump aneurysm, and
anastomotic stenosis of fine APCs.38–41
Aortopulmonary Shunts
Aortopulmonary shunts include modified Blalock–Taussig
shunt (anastomose subclavian artery with ipsilateral branch PA),
Potts shunt (side-to-side anastomosis of the left pulmonary artery
to the descending aorta), and Waterston's shunt (side-to-side anas-
tomosis between the right pulmonary artery and the ascending
aorta). Shunt patency is evaluated by CTA and/or MRA for shunt
occlusion either fully or partially obstructed (Figs. 8, 9). Other
complications include major or branch pulmonary artery stenosis
and seroma around the graft.4–12
Right Ventricular Outflow Tract Patches
Residual flow across patches and patch dehiscence is
assessed by CTA and MRA. It may appear early as irregularity,
thickening, or small defects at the patch site. Other complications
include RVOT patch aneurysm, pulmonary valve stenosis, and/
or regurgitation.35–37
Glenn Operation (Cavopulmonary Shunt)
Glenn operation includes occlusion of flow of superior vena
cava to the right atrium and connection of superior vena cava with
branch pulmonary arteries, augmentation of central pulmonary ar-
teries, and PDA. Complications include narrowing of the Glenn
anastomotic site or stenosis of inferior vena cava-to-pulmonary
artery conduit.42
CONCLUSIONS
We conclude that CTA and MRA are important for preoper-
ative assessment and treatment planning of infants with PA-VSD
as these precise the central pulmonary artery, ABCs, PDA, pulmo-
nary arborization, and associated vascular and congenital heart
disease. For postoperative cases, imaging is important for assess-
ment of stenosis and occlusion of shunts and conduits.
www.jcat.org 909
Abdel Razek et al J Comput Assist Tomogr • Volume 43, Number 6, November/December 2019
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