DOI: 10.1111/jocs.

14479

CASE REPORT

Three‐dimensional printing for surgical planning of a double

aortic arch case

Luigi Arcieri MD1 | Bastien Provost MD1 | Philippe Charbonneau MD1 |

Emmanuelle Fournier MD2 | Sebastien Hascoet MD2 | Emanuel Le Bret MD1

1
Pediatric Heart Surgery Unit,
Centre Chirurgical Marie Lannelongue,
Le Plessis‐Robinson, France
2 Background and Aim: Over the past years, three‐dimensional (3D) models of patient‐
Pediatric Cardiology Unit, Centre Chirurgical
Marie Lannelongue, Le Plessis‐Robinson,
France
Correspondence
Luigi Arcieri, MD, Pediatric Heart Surgery Unit,
Centre Chirurgical Marie Lannelongue,
133 Avenue de la Résistance, 92350
Le Plessis‐Robinson, France.
Email: luigi.arcieri@gmail.com
Abstract
specific anatomical conditions are being used to improve the comprehension and
surgical management of a variety of diseases. It is an additional diagnostic tool that aids
clinical decision‐making. Furthermore, this technology is still not routinely used in the
medical field since its availability is limited by cost and complex process.
Methods and Results: We describe a patient with a balanced‐type double aortic arch
encircling trachea and esophagus. Considering the clinical symptoms, surgical
decompression of these structures and defined aortic arch reconstruction was
indicated. The 3D printed model revealed narrowing of the left aortic arch at the
junction of the descending thoracic aorta that did not clearly appear on the con-
ventional images reconstruction. The left aortic arch was divided and the symptoms
completely disappeared. No immediate or late complications occurred.
Conclusion: 3D printed models can be helpful in surgical planning of congenital
heart malformations. It should be strongly considered as an additional tool in
complex cases.

1 | INTRODUCTION presence of tracheomalacia. Computed tomography (CT), subse-

Double aortic arch (DAA) is a congenital malformation where two
aortic arches form a complete vascular ring (VR) that encircles the
trachea and esophagus. DAA accounts for 40% of all complete rings
and, therefore, it can be considered the most common VR. Re-
spiratory symptoms may vary from exertional dyspnea to apparent
life‐threatening events (ALTE). The right aortic arch with aberrant
left subclavian artery and left ligamentum is another frequent com-
1
plete ring that can be associated with respiratory symptoms.
In common form of DAA, the right arch is larger than the left in
75% of cases. On the contrary, balanced DAA accounts for only 2% to
5% and, in those cases, the two arches are of equal diameter. DAA is
frequently discovered at a younger age during investigations for
dyspnea and dysphagia.2–4 Symptomatic patients require careful
preoperative anatomic evaluation for surgical planning. Over the past
years, fiberoptic bronchoscopy was used as the first instrumental
investigation to assess the degree of tracheal compression or the
quently, was considered the gold standard for the anatomic definition
for its ability to detect differences between the vascular and
respiratory structures.5
Three‐dimensional (3D) printed anatomic models have emerged
in surgery and their utility was demonstrated in several studies and
case reports described in the literature. The printed patient‐specific
models have been demonstrated to be a useful tool for preoperative
planning, surgical decision‐making as well as trainee education.6–8 In
this case report, we describe our experience with the use of 3D
printing in a patient with symptomatic balanced DAA, and how it
influenced our surgical strategy.
2 | CASE REPORT
A 17‐month‐old boy was admitted to our unit for surgical manage-
ment of a complete VR. Clinical history revealed the presence of

J Card Surg. 2020;1–4. wileyonlinelibrary.com/journal/jocs © 2020 Wiley Periodicals, Inc. | 1

2 |
F I G U R E 1 Conventional preoperative computed tomography
scan three‐dimensional reconstruction showing a balanced double
aortic arch. It is difficult to appreciate the smallest arch in this
picture. AA, ascending aorta; DA, descending aorta; LA, left arch;
LB, left bronchus; RA, right arch; RB, right bronchus; T, trachea
stridor within the first days of life, followed by recurrent respiratory
infections and an episode of ALTE with respiratory distress and
cyanosis. An episode of severe dysphagia was also described by
the parents but misinterpreted by the general pediatrician.
ARCIERI ET AL.
Bronchoscopy demonstrated 50% of tracheal diameter reduction
combined with signs of tracheomalacia. A multidetector‐row CT scan
(64‐slice, SOMATOM Definition; Siemens, Forchheim, Germany) was
performed and demonstrated an anatomic balanced DAA (Figure 1).
CT scan slice thicknesses was 0.6 mm and was electrocardiogram
gated using a rotation time of 330 ms.
The tracheal team discussion highlighted the necessity of a
better definition of the anatomy of the two arches (because not
completely clear with conventional CT images) to decide which one
should be preserved. This choice was primary considered to allow the
least arterial gradient across the aorta after the operation and to
optimize the release from the compression. For these reasons, we
opted to build a 3D printed model based on the patient's anatomy.
The CT scan digital images were imported in a dedicated post-
processing software (Mimics Innovation Suite; Materialise NV,
Leuven, Belgium). Segmentation of the thoracic structures was then
performed, and the model was sliced to be easily visualized for sur-
gical planning. 3‐matic software (Materialise, Leuven, Belgium) was
used to process the digital images, and the model was finally printed
using a 3D printer with HeartPrint flex material (Materialise). After
the model analysis, the differences between the two arches appeared
more evident. The left arch was of slightly smaller diameter at the
junction with the descending aorta, when compared to the right
(Figure 2), and the narrowing was easily visible.
The procedure was performed under general anesthesia and
through a left posterolateral thoracotomy within the fourth inter-
costal space. The ligamentum arteriosus was exposed and divided.
The VR was opened by the division of the left arch between the
F I G U R E 2 A, Frontal; B, right lateral;
C, posterior; D, superior views of the printed
model. It is possible to appreciate the
reduction of the diameter of the left arch at
the point of insertion in the descending
thoracic aorta (dotted line). AA, ascending
aorta; DA, descending aorta; LA, left arch;
LB, left bronchus; RA, right arch; RB, right
bronchus; T, trachea
ARCIERI ET AL.
descending aorta and the origin of the left subclavian artery. The
trachea and esophagus were completely released with careful dis-
section of the surrounding tissue. The child was extubated in the
operating room before transfer to the pediatric intensive care unit.
The postoperative course was uneventful. Dysphagia and other
respiratory symptoms completely disappeared at follow‐up (follow‐
up time was 2 years). The child continued to grow‐up and after tra-
cheal team discussion, no further instrumental examination was
proposed considering his stable clinical status.
3 | D I S C U S SI O N
We presented a patient with balanced DAA where surgical man-
agement was influenced by a 3D printed model. To the best of our
knowledge, this is the first case in which 3D printing technology was
applied in a specific case of balanced DAA. According to the
F I G U R E 3 A comprehensive algorithm for
management of balanced double aortic arch
| 3
literature, DAA represents only 1% of all congenital heart defects
(CHDs). Right dominant, left dominant, and balanced‐type aortic
arches represent the three types of DAA, with the last one corre-
sponding to almost 5% of all DAA. DAA can be asymptomatic in some
patients and, therefore, incidentally discovered later in life during
investigations for other thoracic diseases.1–4,9 In symptomatic
patients, surgical treatment is mandatory. Preoperative anatomic
evaluation is necessary to define the arch diameter, the position of
the descending aorta in relation to both arches, and the degree of
tracheal and esophageal compression. Surgery consists of resection/
division of the nondominant arch, which is the left in 80% of cases,
and sometimes atretic. This information is routinely obtained by a CT
scan and bronchoscopy.5 When balanced‐type DAA is encountered,
the purpose of surgery is to give a more harmonious configuration to
the residual aortic arch, to avoid flow turbulence with the negative
hemodynamic effect on ventricular function (Figure 3).10 Therefore,
division of the arch at the opposite site of the descending aorta is the
4 |
usual surgical strategy if no patent ductus arteriosus is present.
When the descending aorta is apparently medial to both arches in a
symmetric manner (as in our case), it is difficult to define which arch
should be divided to obtain the best hemodynamic result. In this
situation, we propose the use of 3D printed model.
As demonstrated in our case, 3D printed model has clarified all
preoperative concerns about the patient's anatomy, showing a left
arch slightly smaller compared with the right, and this detail influ-
enced surgical decision‐making.
The benefits of 3D models in clinical practice have been demon-
strated by several authors. It is a useful tool for preoperative planning
and trainee education of complex congenital cardiac, thoracic and
abdominal cases.6,8,11 Jones et al7 observed that when 3D models were
printed for anatomic abnormalities (like vascular rings or pulmonary
artery sling) and, subsequently used by young cardiothoracic surgeons for
learning, it was a better comprehension of the surgical anatomy of all
cases. To demonstrate this, the authors submitted the residents to a
survey on the anatomy of vascular rings and observed that the test‐score
improved significantly between pre and post 3D model examinations.7
The creation of the patient‐specific model, the advantages of 3D printing
have been demonstrated in different forms of CHD. This innovative
technology has helped the clinicians by the possibility to personalized
treatments, particularly in complex cases as double outlet right ventricle
or pulmonary venous return malformation.6
A novel application of 3D printing is the building of implantable
custom‐made devices that will be specific for the patient anatomy, and
overcome the high variability frequently encountered in CHDs.11
Furthermore, the use of bioabsorbable materials that can dissolve during
growth could have a high clinical benefit.12 Finally, 3D printed models can
be used by surgeons to improve the patient's or parents comprehension
on their malformation and its treatment, allowing a more valuable pre-
operative informed consent for the surgery.13,14 In conclusion, 3D printed
model demonstrated a clear advantage in the management of our patient
with balanced DAA. 3D printed models can be very helpful in surgical
planning of congenital heart disease. Since this technology is progres-
sively being more available, it should be strongly considered as an addi-
tional comprehensive tool in complex malformations.
A C K N O W L E D GM E N T
The authors would like to thank TJ Contini, MD, for its careful
English revision.
CO NFLICT OF I NTERE STS
The authors declare that there are no conflict of interests.
A UT HO R C ONT RI BU TIO NS
All authors contributed equally in the making of this manuscript.
ARCIERI ET AL.
ORCI D
Luigi Arcieri http://orcid.org/0000-0001-9172-8571
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How to cite this article: Arcieri L, Provost B, Charbonneau P,
Fournier E, Hascoet S, Le Bret E. Three‐dimensional printing
for surgical planning of a double aortic arch case. J Card Surg.
2020;1–4. https://doi.org/10.1111/jocs.14479