Received: 4 January 2021 | Accepted: 4 February 2021

DOI: 10.1111/jocs.15492

ORIGINAL ARTICLE

Modified technique for coarctation of aorta with hypoplastic

distal aortic arch

Ivan A. Kozyrev1 | Nikolai A. Kotin1 | Igor I. Averkin1 |

1 1
Andrey A. Ivanov | Alexander A. Latypov | Mikhail L. Gordeev3 |
Elena S. Vasichkina2 | Tatyana M. Pervunina2 | Evgeny V. Grekhov1

1
Pediatric Cardiac Surgery Department,
Almazov National Medical Research Center, Abstract
St. Petersburg, Russia
Background: A combination of coarctation of aorta with various severity of distal
2
Pediatric Cardiology Department, Almazov
National Medical Research Center, St.
arch hypoplasia frequently occurs in newborns. Traditional techniques in the neo-
Petersburg, Russia natal period such as extended end‐to‐end anastomosis or inner curve patch are
3
Cardiac Surgery Department, Almazov controversial. Arch geometry has a marked role in long‐term outcomes. We in-
National Medical Research Center, St.
Petersburg, Russia troduce a modified Amato technique of distal aortic arch enlargement with native
tissue‐to‐tissue reconstruction.
Correspondence
Methods: Neonatal patients with coarctation of aorta and distal aortic arch hypo-
Evgeny V. Grekhov, Department of Pediatric
Cardiac Surgery, Almazov National Medical plasia who underwent surgical reconstruction using this technique between January
Research Center, 2 Akkuratova St, St.
2016 and December 2019 in our center were included. Patients with concomitant
Petersburg 197341, Russia.
Email: egrehov1@mail.ru complex heart defects were excluded. Data were obtained from echo protocols, CT
scans before and after repair. The dimensions of the arch were assessed using
Z‐score, arch geometry was evaluated with height/width ratio.
Results: Thirty‐two patients (22 males, 10 females) were included. Median age and
weight were 7 days (5; 18) and 3.5 kg (3.1; 4.0), respectively. The Z‐score of distal
part of the arch before and after procedure was significantly different (<0.01). No
mortality, recoarctation, or bronchial compression was found during 18 (6–38)
months of follow‐up.
Conclusion: Modified technique for coarctation of aorta with hypoplastic distal
aortic arch provides favorable geometry of the aorta with a low risk of morbidity.
The proper selection and accurate technique could minimize potential risks. This
method is relatively safe and might improve long‐term outcomes associated with
the geometry of aorta.

KEYWORDS
aortic arch, coarctation of aorta, congenital heart defect, distal hypoplastic arch

1 | BACKGROUND coarctation of aorta with various degree of aortic arch hypoplasia

especially its distal part is a frequent finding. Optimal surgical
Coarctation of the aorta is a common congenital anomaly which management for repair of isolated coarctation with hypoplastic distal
occurs up to 8% of all congenital heart malformations with approx- arch remains controversial. Several techniques were proposed for
imate incidence three cases per 10,000 births.1 Combination of correction, such as arch patch reconstruction through sternotomy

J Card Surg. 2021;36:2063–2069. wileyonlinelibrary.com/journal/jocs © 2021 Wiley Periodicals LLC | 2063

2064 | KOZYREV ET AL.

F I G U R E 1 (A) Schematic illustration of

technical aspects of the repair. (B) Completed
view of reconstruction

with cardiopulmonary bypass (CPB), extended end‐to‐end anasto- included. Patients with isolated coarctation (Z‐score of proximal
mosis, reverse subclavian flap technique, end‐to‐side anastomosis and distal arch greater than 2, tubular arch hypoplasia, moderate
etc.2–4 All mentioned techniques have their own pros and cons. For to severe proximal arch hypoplasia (Z‐score of proximal
example, patch repair provides good geometry of the arch and arch ≤ −3) and concomitant heart defects requiring surgery were
tension‐free reconstruction but require CPB and in some cases deep excluded. Z‐scores were calculated using dimensions of the
hypothermic circulatory arrest, and could have risks related to patch vessels, weight, and height with online calculator (https://zscore.
material. On the one hand, extended end‐to‐end or end‐to‐side chboston.org/). Clinical data, computed tomography (CT) scans,
anastomosis could be “less invasive” but on the long‐term follow‐up, echocardiography protocols were collected from patient records
dimensions of the arch remains relatively small especially in cases at the time of presentation and follow‐up. Left ventricle ejection
with tubular arch hypoplasia.5 Furthermore, combination of end‐to‐ fraction (EF) was estimated with through the Teicholz method.
end anastomosis with modified reverse subclavian flap angioplasty Height/width ratio (H/W) was assessed as described previously.8
shortens the transverse segment of the arch which is clearly seen in Briefly, Width of transverse of the aortic arch was defined as the
some patients on the follow‐up visits.6 horizontal distance between the midpoints of the ascending and
Arch geometry and deformation could play a prominent role in descending aorta. Height of the aortic arch was defined as the
distant morbidity leading to adverse vascular remodeling and high maximal vertical distance between W and the highest mid‐point
7
blood pressure. We introduce a Modified technique for distal aortic of the aortic arch.
arch enlargement with native tissue‐to‐tissue reconstruction. This Indication for intervention was systolic pressure gradient on
technique includes distal arch augmentation with the left carotid Echo higher 20 mmHg. Rate of mortality, residual aortic gradient,
artery while providing its continuity, resection of coarctation and all reintervention, any vascular changes at surgery area such as
ductal tissues, and creation of anastomosis of reconstructed distal aneurysm, ostial brachiocephalic stenosis and bronchial com-
aortic arch and proximal part of left subclavian artery with des- pression were reviewed. We consider as a residual gradient a
cending aorta. In our opinion this method preserves whole length of doppler peak velocity over 2.5 m/s or blood pressure gradient
transverse segment of arch thus providing optimal shape of aor- between upper and low extremity over 20 mmHg.
tic arch. Near‐infrared spectroscopy with a cerebral oximetry (INVOS
5100; Medtronic) is used for continuous monitoring of oxygen sa-
turation in the both hemispheres of the brain, during the brachio-
2 | METHODS cephalic arteries cross clamp.

Local ethics committee approved the Study (Committee meeting No.

170420, 14.12.2015). Informed consent was signatured by official 2.1 | Surgical technique
representatives of patients.
This is a retrospective study. Patients younger than one After entering the left chest through the third intercostal space,
month old with coarctation of aorta and distal arch hypoplasia 100 U/kg of heparin was administrated and the patient cooled
(Z‐score ≤ −2) who underwent surgical repair with modified down to 34°C during dissection. It is very important to perform
technique between January 2016 and December 2019 were extensive mobilization of the arch, left brachiocephalic arteries
KOZYREV ET AL. | 2065

and descending aorta to avoid any tension of anastomosis. TABLE 1 Characteristics of the patients and operative data
Schematic illustration shows lines of incisions and directions of Variables Range
anastomoses (Figure 1A). No. of patients 32
After placing marking holding sutures, proximal arch just
Age, days 7 (5; 18)
beyond innominate artery, distal part of left brachiocephalic
vessels and aortic isthmus are clamped. During the first stage, Weight, kg 3.5 (3.1; 4.0)

distal perfusion is provided through ductus arteriosus (PDA), Male, n 22

which is patent in the vast majority of patients. The incision of Female, n 10
superior aspect of the distal arch between left carotid artery
Associated anomalies, n
(LCA) and left subclavian artery (LSA) is made. The incision is
started from proximal parts of contiguous walls of LCA and LSA ‐ 3
ASD
and then is carried down on superior surface of distal aortic arch.
It is important to mention that the length of incision on LCA is ‐ 5
VSD
longer than the one on LSA. Then anastomosis is carried out with
starting point on posterior wall in‐between LCA and distal aortic ‐ 12
BAV
arch until the upper incision points on LCA and LSA are reached.
After that the anastomosis line is proceeded to the anterior as- ‐ 2
Other
pect of vessels, down to the end point in‐between LCA and distal
aortic arch. After clamps removal surgical hemostasis within Time first anastomosis, min 25 (23; 28)
several minutes of reperfusion of all brachiocephalic arteries is Time second anastomosis, min 19 (17; 23)
perform. At the second stage after PDA ligation, resection of
Abbreviations: ASD, atrial septal defects; BAV, bicuspid aortic valve; EF,
coarctation with all ductus tissue is made. The incision of the ejection fraction; VSD, ventricular septal defects.
leftward part of the isthmus is carried up to the left side of LSA,
no incision is made on the inner (inferior) curve of the arch to End‐to‐side oblique anastomosis between lateral (leftward) side
save all the length of the transverse segment of aortic arch. To of isthmus/LSA and descending aorta is completed (Figure 1B).
prevent LSA stenosis the incision on its left side should be Intraoperative photos demonstrate step‐by‐step procedure
2–3 mm higher than the anastomosis end point of the first stage. (Figure 2A–H).

F I G U R E 2 (А) Intraoperative anatomy before the procedure. (B) Marking holding sutures are placed next to origin of LCA and LSA.
(C) The incision of the contiguous walls of the LCA and superior aspect of the distal arch and LSA origin. (D) Anastomosis of the LCA and distal arch
is performed. (E) Brachiocephalic vessels and arch are released to confirm hemostasis. (F) Coarctation site with all ductus tissue resected, PDA
controlled with two ligatures. (G) Incision from lateral (leftward) aspect of the isthmus is carried up on the lateral (leftward) wall of the LSA.
(H) Completed anastomosis. DescAo, descending aorta; PDA, patent ductus arteriosus; LCA, left common carotid artery; LSA, left subclavian artery
2066 | KOZYREV ET AL.

2.2 | Statistics 4 | DISCUSS ION

Data were analyzed with the Graphpad Prism Ver. 5.0 (GraphPad There are several methods to estimate the degree of aortic arch
Software, Inc) and Statistica 10.0 (StataCorp LP). The normal dis- hypoplasia. The first method is to measure the size of transverse
tribution of continuous variables was determined by Shapiro–Wilk arch, and it should be less than minus two standard deviations
test. Data are expressed as median with interquartile range. (Z‐score). This method requires very precise measurements as even
small changes in dimensions may lead to very different values. The
second method describes the diameter of the arch in millimeters. In
3 | RESULTS this method, the arch diameter should be less than half of the dia-
meter of descending aorta.9 The third method measures the di-
Thirty‐two patients (10 females (31%), 22 males), with a median age mension of transverse arch in millimeters and it should be less than
of 7 days were included in the study (Table 1). bodyweight +1.10 The fourth method measures the diameter of
The median Z‐score of distal part of the arch before procedure the”normal” distal aortic arch. This diameter should be at least 50%
−2.5 (−3.1; −2.3) and after procedure 0.1 (−1.1; 1.0) was significantly of the diameter of the ascending aorta measured at a prescribed
different (p < .01). The cross‐clamp time was 25 (23; 28) and 19 (17; fixed distance proximal to the origin of the innominate artery.7 In our
23) for the first and the second anastomosis respectively. In our department, we use the first method to assess the aortic arch.
study, all patients were with preserved EF. The median EF before and During the last decades, several new approaches were proposed
after procedure was 73% (67; 78) and 69% (65; 73), respectively. The to treat patients with coarctation of aorta. One of these methods is
median aortic arch H/W ratio on follow‐up was 0.5 (0.45; 0.57). We balloon dilatation. Few works compared open surgical repair with
did not find any gothic shape of aortic arch on CT scan images after balloon dilatation in neonates. Surgical repair showed better results
surgery (Figure 3). Illustration with 3D reconstruction demonstrate in rate of reintervention and aortic arch growth.11,12 This method
arch shape anatomy before (Figure 4A) and after correction allows to achieve removal of all ductal tissue. This is a crucial benefit
(Figure 4B) and at 6 months after surgery (Figure 4C). of this type of intervention. In patients with associated arch hypo-
No mortality, recoarctation, or bronchial compression was de- plasia, only surgical repair can provide acceptable results.
tected during 18 (6–38) months of follow‐up. There were no such After successful surgery, not only the shape of aorta but also
complications as hemorrhage, chylothorax or neurological impair- dimensions might have influence for a long‐term outcome. Three‐
ment. No evidence of brachiocephalic narrowing was found except dimensional magnetic resonance imaging demonstrates blood flow
one case where the left subclavian artery stenosis was revealed in- distribution with shear stress evaluation assessment in patients with
traoperatively. This patient was at the beginning of learning curve normal and hypoplastic aortic arch after repair of coarctation.13 In
and had LSA of 2 mm in diameter, in whom the upper end point of patients with hypoplastic arch asymmetrically elevated blood flow in
second anastomosis line turned out to be at the same level with the systole and presence of wall shear stress was shown. These findings
first one thus causing waist formation. After performing the proce- suggest that increasing asymmetry of aortic flow distribution in-
dure according to scheme it has required left subclavian artery re- creases risks of aneurysm formation in the most stressed aortic
implantation in the left carotid artery with good result at 16 months areas. Residual arch hypoplasia is often depending on the type of
follow‐up. surgical repair. Relatively close technique of reconstruction of distal

F I G U R E 3 Aortic shape in six different

patients after repair
KOZYREV ET AL. | 2067

F I G U R E 4 3D reconstruction (A) before, (B)

after surgery and (C) six months follow‐up

arch with coarctation of aorta was described by Amato et al.14 In this Another technique addresses the problem of distal arch hypo-
technique, the first stage was creation of end‐to‐end anastomosis plasia with aorta coarctation is reverse subclavian flap with ligation16
between inner curve of aortic arch and descending aorta and after or reimplantation of LSA.17 While it provides tissue‐to‐tissue repair,
that anastomosis of proximal parts of contiguous walls of LCA and it hasn't become a widespread method. The one reason for that
LSA. The arch shape after reconstruction looks almost the same as might be technical complexity together with lack of physiologic flow.
extended end‐to‐end anastomosis with more favorable distal aortic This method might look close to some extent to the concept of our
arch size. In our modified technique, not only the order of the steps, procedure however has some significant differences. Reimplantation
which gives advantage of providing perfusion of the lower part of the of LSA with circular anastomosis in reverse subclavian flap repair
body through PDA during the first stage of correction, is different could have a risk of stenosis while our modified technique includes
from the original technique, but also the aortic shape after repair due distal arch augmentation with left carotid artery while providing its
to preserving whole the length of aortic arch transverse segment. In continuity. Another difference point could be geometry of the arch
our opinion, this is the main difference in compare with original after repair, which in reverse subclavian flap repair tends to be more
Amato's technique. We strongly believe that this modification pro- gothic due to descending and ascending aorta approximation be-
vides smoother arch geometry and minimize risk of angulated de- cause of undersewing descending aorta to the inner curve of the arch
formity after surgery. thus making its shape relatively close to extended end‐to‐end ana-
Presence of tubular arch or moderate or severe hypoplasia of stomosis type.
proximal arch could be challenging for repair. The most widely used One of the methods that full eliminate the presence of hy-
type of correction as extended end‐to‐end anastomosis via thor- poplastic aortic arch segments is end‐to‐side anastomosis. The
acotomy might be performed, but residual hypoplasia of the proximal results of this type repair through sternotomy in patients with
arch could be the reason for the long‐term morbidity. One study aortic arch coarctation and interruption with long‐term follow‐up
15
showed that the arch can increase after resection of coarctation. have been reported. 18 During follow‐up period, 18‐year survival
This could be a reason why midterm outcome showed a low rate of was 93%. No reoperation was required in 87% of patients and
reintervention for recoarctation. This theory was also discussed by lack of any intervention was reported for 85% patients. Almost
Kotani et al.5 They found that after repair the segment of proximal one fifth of patients (17%) of late survivals had prehypertension
arch grows significantly but still remained smaller in patients with and 10% had hypertension. More than a quarter of all patients
previous moderate to severe tubular arch hypoplasia. Extended (27%) had increased blood pressure which could be partially
end‐to‐end anastomosis via left thoracotomy has a low risk of explained by the specific aortic arch shape after repair. The ob-
reintervention for patients with proximal arch hypoplasia. Never- vious disadvantage of this approach is the need to use CPB.
theless, this method leads to approximation of descending aorta to One more approach to treat patients with tubular arch or
the ascending one thus usually resulting in more gothic aortic arch moderate or severe proximal arch hypoplasia and without con-
shape after surgery. Besides, from our experience, there are some traindications for CPB is “Norwood type” reconstruction. This
patients in which extended end‐to‐end anastomosis wouldn't be method is carried out through median sternotomy with homo-
successful for distal arch hypoplasia relief but our modified techni- graft patch for inner curve of the arch.19 This type of repair is a
que can solve this problem due to less space requiring for clamp method of choice for cases with intracardiac defects that re-
application. quired surgery.
2068 | KOZYREV ET AL.

One of the most serious factors of long‐term risk is hyperten- odified procedure. Second, in cases of combination of coarctation
sion. It is common complication after repair of aortic coarctation with tubular arch or moderate proximal arch hypoplasia, presence of
even in cases of “successful” repair when no residual gradient is contraindication for modified procedure we perform extended end‐
revealed postoperatively. Even mild aortic arch hypoplasia is asso- to‐end anastomosis via thoracotomy. Third, in cases with coarctation
ciated with predisposition to arterial hypertension, despite the lack with intracardiac or great vessels defects or severe proximal arch
of significant arm‐leg gradient at rest.20 Geometry of the arch might hypoplasia we chose central approach via sternotomy with CPB. In
21
have a strong influence on progression of arterial hypertension. our opinion it is the appropriate approach to decrease long‐term
Three types of aortic arch were defined based on arch geometry morbidity.
after successful repair of aortic coarctation: gothic, crenel and nor- It is not generally accepted practice to use heparin during repair
mal.8 Gothic arch was represented by an acute angulation between aortic coarctation via thoracotomy in neonatal period. We usually
ascending and descending aorta with shortened or absent of the administrate 100 U/kg of heparin in cases with novel technique
horizontal part of the aorta, and the H/W gradient was around 0.8. because of the risks of double aortic clamping for the first and sec-
Data of analysis confirmed fact that the hypertension was more ond anastomosis. We believe this prophylaxis dose of heparin, on one
frequent in patients with Gothic arch geometry (45.5%). Although hand, minimizes the risk of thrombotic complication which were
the pathophysiology of hypertension in this condition is not fully completely absent in our cases, on the other hand, we did not detect
understood.7 In our study we use the parameter H/W in infants. In either any hemorrhagic events.
our cohort, the median H/W ratio was 0.5. Apparently, this could be In conclusion, this modified technique for aortic coarctation with
related to the young age of our patients. Despite age, we tried to hypoplastic distal aortic arch provides favorable geometry of the
compare our data with data from previously mentioned studies. We aorta. The main risks associated with compromising of left arch
did not find any gothic arch in our patient during follow‐up period. vessels. The proper selection and accurate technique could minimize
Nevertheless, limited follow‐up does not guarantee lack of devel- potential risks. This method is rather safe and we believe might
oping of this condition in the future. Not only hypertension but also improve long‐term outcomes related to the geometry of the aorta.
22 23
left ventricular function and clinical vascular dysfunction could Further studies with long‐term follow‐up are needed.
be associated with the aortic arch shape on the long‐term follow‐up
after surgery. In other words, aortic arch shape has a prominent role
in long‐term morbidity. 5 | LIM IT A TIO N
It is worth to say that our method requires precise technique
and has a risk of disturbance of left arch vessels. In our cohort, we An important limitation of the study is lack of control group to
found two cases of mild dilatation of the arch at the level of LSA and compare different techniques in relatively close anatomic conditions.
LCA 24 months after surgery, which were in our understanding due Another limitation concerns retrospective nature, small number of
to excessive cut of LSA on the rightward side during the first stage patients, short follow‐up time and single center approach.
of procedure, which could take place in the beginning of the
learning curve. Now we don't make incision on the LSA longer than CO N FLI CT O F I N TER E S TS
3‐4 mm to avoid potential excessive augmentation of this segment. The authors declare that there are no conflict of interests.
The main contraindication for this technique is presence of tubular
or proximal arch hypoplasia, which we considered when Z‐score of OR C ID
proximal arch was less than −3. Other contraindication in our ex- Ivan A. Kozyrev http://orcid.org/0000-0002-2533-5339
perience was the weight of the patient less than 2 kg. In addition, Nikolai A. Kotin https://orcid.org/0000-0003-1124-701X
following anatomic features were contraindications for application Igor I. Averkin https://orcid.org/0000-0002-6443-1796
of our technique: Alexander A. Latypov https://orcid.org/0000-0002-5459-6765
Mikhail L. Gordeev https://orcid.org/0000-0002-8199-0813
• Aberrant RSA Elena S. Vasichkina https://orcid.org/0000-0001-7336-4102
• Small size of the arch vessels (LCA less then 2.5 mm and LSA less Tatyana M. Pervunina https://orcid.org/0000-0001-9948-7303
then 2.0 mm) Evgeny V. Grekhov https://orcid.org/0000-0002-7636-1983

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