Download Congenital abnormalities of aortic artery. Assessment in neonates

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Original article
Congenital abnormalities of aortic artery.
Assessment in neonates and early childhood
with multislice tomography
The multislice angiotomography (MDCTA) presents several properties and is utilized with more frequency every time by childhood cardiovascular surgeons as a chosen method of imaging in the anatomic and
pathological scan of different abnormalities of the aortic arch. The multiplanar capacity and three-dimensional reconstructions in particular are easily to interpret,
as well as being extremely fast studies, with short anesthetic time and low contrast volume to administer.
The objective of the present study has been to
review the findings enabled by multislice computer
tomography (MDCT) of the aortic arch’s development
most frequently found in our institution.
MATERIALS AND METHODS
Angiotomography studies (CTA) performed between October 2006 and November 2008 to patients who
presented congenital abnormalities of aortic artery, were
evaluated in a retrospective way. Total patients included
in this study were neonates and little children in an age
range between 6 days and 11 months. Echocardiography
and/ or thorax X ray, together with the symptoms, enabled a presumptive diagnosis (Table 1).
1
2
Servicio de Diagnóstico por Imágenes.
Cirugía Cardiovascular Infantil. Fundación Favaloro. Avenida
Belgrano 1746. Ciudad de Buenos Aires. República Argentina.
Correspondencia: Dr. Diego Haberman: [email protected]
The studies were performed with a 64 detector CT
(Aquilion , Toshiba Medical Systems, Tokio, Japan)
using a software of radiation modulation doses (Sure
Exposure) with effective doses between 0.6 to 1.4 mSv.
Images taken every 0.5 mm thickness were obtained
with a 0.3 mm reconstruction interval, a factor pitch
of 0.828 and 0.5 sec rotation tube.
Between 1 and 1.5 ml/kg of non ionic intravenous
contrast (Iopamiron 370®, Schering) were injected
under anesthesia and cardiac monitoring, with a 1.5 to
2.5 ml/sec flow injection pump (Medrad , Stellant).
Three-dimension reconstructions were performed
in a workstation (Vitrea, Vital Images). All patients
were symptomatic and had surgery, which enabled to
correlate the information observed by different imaging methods with intra-surgery findings, assumed as
certain definitive diagnosis.
In 11 cases, the presumptive diagnosis was confirmed by angiotomography. In one patient, the angiocomputed tomography showed a non suspected
pathology that was further confirmed by surgery.
Embryology review
Blood vessels start to develop from the mesoderm
that covers the yolk sac at 18 days of the embryo’s age.
On day 24th, the cardiovascular system is composed
Recibido: abril 2009; aceptado: julio 2009
Received: april 2009; accepted: july 2009
©SAR-FAARDIT 2009
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INTRODUCTION
Número 3
Congenital abnormalities of aortic artery.
Assessment in neonates and early childhodd with
multislice tomography
In the evaluation of aortic artery congenital abnormalities,
the echocardiography and the plain X ray are the traditionally used imaging methods. Multislice angiotomography
appears as an important method in diagnosis of these different diseases allowing evaluate these entities in a non invasive,
fast and accurate form, giving to cardiovascular surgeons
very important information to delineate the surgical strategy.
In this article, we review the applications of multislice angiotomography in the evaluation of most frequent congenital anomalies of aorta artery, performed in neonates and early childhood.
Key words: Aorta artery. Congenital anomalies. Neonates.
Multislice angiotomography.
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Abstract
Para la evaluación de los defectos congénitos de la arteria aorta, los métodos de imágenes utilizados tradicionalmente son la ecocardiografia y la radiología simple.
La angiotomografía computada multicorte aparece como
un método de relevancia en el diagnóstico de estas entidades, permitiendo evaluar en forma no invasiva, rápida
y precisa las distintas anormalidades, otorgando de esta
manera información sumamente útil a los cirujanos cardiovasculares para definir la estrategia quirúrgica.
En el presente artículo revisamos las aplicaciones de la
angiotomografia computada multicorte en la evaluación
de las malformaciones congénitas de la arteria aorta más
frecuentes en el periodo neonatal y en la primera infancia
Palabras clave: Arteria aorta. Anomalías congénitas.
Neonatos. Angiotomografia computada multicorte.
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Resumen
2009
Diego Haberman1, Enrique Gurfinkel1, Alejandro Beresñak1, Adriana Martínez1, Rosa Emsani1, Rubén Toledo2
Congenital abnormalities of aortic artery
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Table 1
The heart begins a process of folding and septation. Subsequently, the cardiac bulb divides in three
parts: proximal, one intermediate called truncus arteriosus and distal, called aortic sac. The two big arteries
that are born in the heart are not originated from preexisting vessels, but from the truncus arteriosus,
which first forms a partition and then divides to form
two independent vessels: the ascending aortic artery
and the pulmonary artery trunk.
From the aortic sac there 6 pairs of aortic arches
connecting the ventral and dorsal aortic sketches are
born. Some of them usually regress with development, persisting at the end of the fifth week only
third, fourth and sixth arcs, which will later originate
the great vessels (1).
Patient
Gender Age
Diagnosis
DV
M
5 months
Incomplete aortic ring
LI
M
3 weeks
Truncus arteriosus type II
MM
F
2 weeks
Coarctation
PM
F
11 months
Aberrant subclavian
right artery
PS
F
6 days
Interruption of aortic
arch type B
SA
F
2 months
Coarctation
RM
F
11 months
Coarctation
VG
M
7 days
Coarctation
LC
F
5 months
Interruption of aortic
arch type A
RESULTS
PM
F
6 months
Interruption of aortic
arch type A
SN
M
1 month
Coarctation
PA
M
1 month
Truncus arteriosus type III
In the present study, twelve symptomatic patients
were included, all of them with a previous echocardiography. One patient with coarctation diagnosis by
echocardiography, was interpreted by CTA as an interruption of aortic arch type A, which was confirmed
by surgery. In the other 11 patients, there was a coincidence between the echo-cardiography and the CTA.
None of our patients underwent MRI.
SN
M
1 mes
Coartación
PA
M
1 mes
Tronco arterioso tipo III
Coarctation
This is the most common entity in our series, with
5 patients studied with preoperative CTA (41.6%).
Fig. 1. Male patient, with 6 days of life. 3D reconstruction. Sagital
plane. Coarctation of typical localization in descending aorta, distal to
the appearance of left subclavian artery (arrow). Relative hipoplasia of
transverse arch portion (arrowhead).
Fig. 2. Female patient, with 14 days of life. Signs of heart failure, pulse
discrepancies and dorsal systolic murmur. Sagital plane. Maximum
intensity reconstruction. Severe coarctation (arrow) with continuity to
descending aorta through a filiform flow and intercostals collateral circulation (arrowhead). A.M.I: Internal mammal artery. In surgery, the
diagnosis is proved. Resection was performed for coarctation with end
to end anastomosis and ligation of the ductus, which was permeable
(not visible in ACT).
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Clinical cases
of two cardiac tubes, arterial and venous vessels.
Subsequently, both cardiac tubes fuse to form a tubular heart that develops four dilatations named cavernous sinus, atrium, venticulum and cardiac bulb.
Diego Haberman et al.
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Fig. 3. a) Female patient, 2 months old. Coronal plane: Situs inversus
totalis plus coarctation (arrow). A.S.D.: Right subclavian artery. A.I:
Left atrium. A right torocotomia is performed, coarctation section and
end to end anastomosis . b) 3D reconstruction. Posterior PLANE.
Coarctation (arrow). A.S.D: Right subclavian artery. T: trachea.
Scheme 1: Illustrative scheme of the interruption of aortic arch type A.
VD: Right ventricle.
VI: Left ventricle.
AP: Pulmonary artery.
A Ao Asc: Ascending aortic artery.
A Ao Desc: Descending aortic artery.
Tr Bc: Brachiocephalic arterial trunk.
ACPI: Left primitive carotid artery.
ASI: Left subclavian artery.
Other cardiovascular abnormalities are usually
presented, among them, bicuspid aortic valve, hipoplastic transverse arch portion, dilatation of the ascending aorta, persistent ductus and aberrant right subclavian artery (4) (Fig.1).
There is hypertension of superior extremity, which
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It is a congenital abnormality characterized by
narrowing of the aortic lumen. Stenosis is often found
in the proximal descending aorta after the emergence
of left subclavian artery. Considering its connection to
the ductus arteriosus, they are classified in preductal,
postductal or yuxtaductal (2, 3).
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c
Fig. 4. a) Female patient. Interruption of aortic arch type A. Sagital plane. Maximum
intensity reconstruction. Thoracic artery is
interrupted in the posterior sector of the arch
(arrow), in distal position to the left subclavian artery (ASI). The descending aorta receives flow from the pulmonary aorta (AP)
through a permeable ductus (arrowhead). b)
3D reconstruction. Anterior oblique left plane.
Ao. A.: Ascending aorta. Ao. D.: Descending
aorta. AP: Pulmonary artery. ASI: Left subclavian artery. Interruption of aortic arch
(arrow). c) 3D reconstruction. Posterior oblique plane. APD: Right pulmonary artery.
API: Left pulmonary artery. ACPI: Left primitive carotid artery. ASI: Left subclavian
artery. Ao. D.: Descending aorta. In the surgery, the ductus is joint. En la cirugía se liga el ductus, se reseca la zona interrumpida con posterior anastomosis que involucra el arco aórtico, la
boca de la arteria subclavia y la aorta descendente.
Congenital abnormalities of aortic artery
b
c
d
c
Fig. 5. a) Female patient, with 5 days of life. Respiratory distress. Cardiogenic shock. Aortic interruption type B. 3D reconstruction. There is a dissociation between ascending and descending
aorta, which has a continuity with the trunk of pulmonary artery. The interruption is between
carotid artery and left subclavian, which emerges from the descending aorta. Ao. A.: Ascending
aorta. A.C.P.I: Left primitive carotid artery. ASI: Left subclavian artery. b) 3D reconstruction.
Posterior plane. Ao. A.: Ascending aorta. Ao. D.: Descending aorta. A.P.D: Right pulmonary
artery. ASI: Left subclavian artery. c) Sagital plane. The pulmonary artery (AP), dilated, continues with the descending aorta (Ao. D), and from it, the left subclavian artery (ASI) is originated.
d and e) Coronal plane. The aorta and pulmonary artery arise from one of the corresponding ventricles. V.I.: Left ventricle. V.D.: Right ventricle. A.P.: Pulmonary artery. Ao.A.: Ascending aorta.
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tends to be offset by collateral development of intercostals, epigastrics and mediastinals (Fig. 2).
Infants with coarctation, particularly when a persisting ductus arteriosus exists, presented with signs
of congestive heart failure (tachycardia and dyspnea).
A dorsal systolic murmur is often listened, pulses are
verified and differential pressures in the lower limbs.
Diagnosis is based on the clinical findings and on
imaging methods (echocardiography, angiography,
angiotomography and magnetic resonance).
The CT scan gives us information about the site
and extension of stenosis, aspect of collaterals and the
presence of associated abnormalities, currently an
extremely useful modality in the following of postpercutaneous or surgery treatment (Fig 3).
Scheme 2: Illustrative scheme of the disruption
of aortic arch type B.
VD: Right ventricle.
VI: Left ventricle.
AP: Pulmonary artery.
A Ao Asc: Ascending aortic artery.
A Ao Desc: Descending aortic artery.
Tr Bc: Brachiocephalic arterial trunk.
ACPI: Left primitive carotid artery.
ASI: Left subclavian artery.
Interruption of aortic arch
It is defined as the loss of anatomic continuity between the ascending and the descending aorta, and it is considered as an extreme form of coarctation. The flow
towards the descending aorta is given by the ductus,
which remains permeable and provides continuity between the trunk of the pulmonary artery and the distal aorta
to the interruption. Despite this continuity, the blood that
arrives to the abdomen and lower limbs is only partially
oxygenated. In most of the cases, other cardiovascular
abnormalities are associated, such as interventricle communication (IVC), obstruction in the exit tract of left ventricle, bicuspid aortic valve and persisting arterial ductus.
Patients evolve fast after birth with severe congestive heart failure, shock tendency and, if not treated,
they often pass away in the neonatal period at few
days of birth (5).
Diego Haberman et al.
Truncus arteriosus
The truncus arteriosus (TA) is characterized by the
presence of a single large artery which originates in
the base of the heart with a single annulus, that originates coronary, pulmonary and systemic arteries.
In most of the cases there is an interventricular
communication and the trunk usually originates over
this septal defect. It is the result of a failure in the early
embryonic development due to a lack of division of
the primary arterial precursor (truncus arteriosus) in
aortic and pulmonary arteries, eventuality that must
happen in the normal fetus at approximately 8 weeks
of gestation. It is a pathology with low incidence, and
represents approximately the 0.7% of cardiovascular
congenital defects. It can be associated to other alterations, among them, the aortic arch to the right, coarctation and interruption of the aortic arch (association
which is present in the 14% of TA), and also to extracardiac abnormalities, particularly Di George syndro-
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Vascular rings
They belong to abnormal configurations of aortic
arch, which are the result of an incomplete regression
of one of the six present bronchial arches during the
embryonic development. Vascular structures or arterial ligament surround and compress the trachea and
esophagus, generating symptoms such as stridor, breathing difficulty, air trapping and apnea; the esophagus compression causes vomits, swallowing difficulty
and dysphagia. They predispose to the appearance of
aspiration pneumonias (12).
They can be complete, forming a continuous ring
around the trachea and the esophagus, or incomplete,
in such cases the symptoms are usually minor or even
absent (13).
There are several forms of vascular rings:
Syndrome of the anomalous innominate artery.
The brachiocephalic arterial trunk originates from the
aortic arch but more to the left than usual, being the
reason for which it crosses diagonally ahead the trachea and can have certain degree of compression on
its anterior side.
Double aortic arch. It can exist as complete aortic
arch (functional), in which there is persistence and
permeability of right and left arch, or as double arch
with partial atresia of one of them (usually, the left
one), where the continuity is given by a remaining
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Its incidence is low, 3 cases at a million of born
alive approximately, and represents the 1% of all cardiovascular abnormalities. In our study, there were 3
of the 12 studied patients.
They have been classified in three types according
to the place where the interruption is,:
Type A: Distal to the left subclavian artery.
Type B: Between the common left carotid artery
and the left subclavian artery.
Type C: Between the brachiocephalic arterial trunk
and the left subclavian artery.
Type B is the most frequent one (53%), followed by
type A (43%) and type C (4%) (6).
The ACT clearly shows the interruption, its connection to the supra-aortic trunks, as well as the presence of the ductus, that provides continuity between
the pulmonary artery and the descendent aorta
(Schemes 1 and 2) (Fig. 4, 5, 6).
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Figure 6. Surgical photography. A.P.: dilated pulmonary artery. Ao.A.:
Ascending aorta.
Collet-Edwards classification
Type I. There is a small trunk that is separated.
Right and left branches of the pulmonary artery are
originated from it.
Type II. Both arteries are born from the posterior
side of TA, separated by a short distance.
Type III. Each artery is born independently of the
TA’s lateral side, one away from the other.
Type IV. One branch is born from the trunk and
the other, from the descendent aorta or the ductus.
Van Praagh questions Collet’s classification, specially type IV, and introduces some modifications considering the presence or absence of interventricular
communication and interruption of aortic arch.
He uses the prefix A to indicate the presence of IVC,
or B to indicate the intact septum. On the other hand,
he includes in type IV the TA associated to the interruption of the arch (10). There is a pulmonary flow increased
with a mixture of systemic and pulmonary blood.
Most of the children arrive with cyanosis in early
stages due to the mixture of oxygenated and deoxidized blood and with signs of congestive heart failure.
The prognosis, if a surgery correction is not performed, is bad, with a survival of 25% at 6 months (11)
(Schemes 3 and 4) (Fig. 7, 8).
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me (facial anomalies, cleft palate hypoplastic thymus
and hipocalcemia) (7).
There are two anatomic classification systems that
basically consider the origin of pulmonary arteries:
Collet-Edwards and Van Praagh (8, 9).
Congenital abnormalities of aortic artery
a
b
c
d
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Scheme 3: Illustrative scheme of truncus arteriosus type III.
VD: Right ventricle.
VI: Left ventricle.
CIV: Ventricular septal defect.
TA: Truncus arteriosus.
APD: Right pulmonary artery.
API: Left pulmonary artery.
Figure 7. a) Male patient, with 3 weeks of life. Dyspnea, tachycardia and cyanosis. Truncus arteriosus type III. Coronal plane. Extensive ventricular septal defect (C.I.V) above which originates a single large artery called truncus arteriosus (TA). V.I.: Left ventricle. V.D.: Right ventricle.
A.D.: Right atrium. b) Coronal plane. Picture at 5 mm posterior to previous photo. From the
truncus arteriosus (TA) is fromed a small common trunk that will form the brachiocephalic arterial trunk (Tr.B.) and left primitive carotid artery (A.C.P.I.). c) The right pulmonary artery originates from the lateral side of the truncus arteriosus (T.A.). d) Plane cut at 5 mm posterior to
the previous image. Left lung artery (API) is born far away from and at different heights of the
right pulmonary artery. At the same level, the truncus is continuous with the descending aorta.
fibrous cord that completes the ring. This last option is
the most frequent one.
Supra-aortic trunks appear separately, without a
brachiocephalic arterial trunk, this is, subclavia artery
and right carotid, subclavia artery and left carotid,
which is named sign of the four vessels. When the
minor arch is atresic, the fibrous cord generally appears distal to the appearance of left subclavian artery.
In these cases, it can sometimes be misinterpreted
as an aortic arch to the right; however, the sign of the
four vessels is usually seen. On the other hand, on the
atresic side, a small permeable segment previous to the
fibrous cord usually remains, manifested as a diverticulum posterior to the esophagus (Scheme 5) (Fig. 9).
Aortic arch to the left with aberrant right subclavian artery. In the literature it is not considered as a true
ring. However, in some occasions, it causes considerable symptoms, similarly to true rings. In these patients,
the right subclavian artery is not originated from the
brachiocephalic arterial trunk, since it branches off as
the last vessel of the aortic arch and courses posterior to
the esophagus upwards to the right (Fig. 10).
Aortic arch to the right with aberrant left subclavian artery. The aberrant left subclavian artery is the
last trunk to branch off. The presence of the arterial
ligament completes a true ring. They usually are
symptomatic at early age.
DISCUSSION
Traditionally, congenital abnormalities of aortic
arch were evaluated with echo-cardiography or conventional radiology and, in some instances, with conventional angiography. In recent years, magnetic resonance (MR) and ACT, specially with multislice technology, gained relevance in the diagnosis and pre-surgery scan of different abnormalities (14-16).
Each one of these modalities presents advantages
and disadvantages that must be considered in choosing one.
Neonate patients and little infants present a heart
rate that oscillates between 100 and 160 heart beats a
minute. Therefore, it is essential to count with equip-
Diego Haberman et al.
a
b
c
d
ment of high temporal resolution in order to reduce
the movements per heart beat, decreasing also the
time exposure to radiation and the anesthetic time,
and also with high space resolution due to the small
size of the organs to study.
Multislice computer tomography achieves these
specifications. In 64 detector units the space resolution
is of 0.35mm and the temporal resolution is of 0.4
seconds, enabling to obtain, in an apnea of 3 to 5
seconds, an acquisition of the complete thorax of a
neonate or a little infant. It only takes one volumetric
acquisition to subsequently do multiplanar and three
dimensional reconstructions.
With ACT it is possible to perform “triggered” or
synchronized exams, obtaining images in different
phases of the EKG, which allows to reconstruct them
in the lowest movement phase and to reduce the artifacts per beat. However, triggered acquisition adds
more radiation, for what it is used in a few occasions,
when it is absolutely necessary to define the aortic root’s anatomy (where there usually is more movement).
The disadvantage of ACT is the utilization of
radiation and eventual adverse reactions to iodine.
With high field MR units and fast sequences, images of high quality and millimeter space resolution are
obtained. Planar sequences are obtained in different
orientations, and also images at up 20 phases during
the cardiac cycle that are visualized as film and 3D
angiographic sequences (17).
One of the advantages of this method is that it provides morphological and functional information, with
the possibility to measure flow and pressure gradients. This can be used as a predictor of severity in
situations such as coarctation (18). In addition, it enables to visualize the anatomy and function of heart
valves, which is useful in the aortic arch anomalies
that are associated to valve malformations (coarctation- bicuspid aortic valve).
Another advantage of MR is that it does not use
ionizating radiation, which is important when control
repetition studies are needed.
One disadvantage is the acquisition time, which
can be a limitation for the study of patients with bad
general condition who cannot tolerate prolonged
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Figure 8. Truncus arteriosus type II. 3D reconstruction. Previous view. From the truncus arteriosus (T.A.) emerges a small common trunk (T.C.) that will give rise to the left primitive carotid artery (ACPI) and the brachiocephalic arterial trunk (Tr.Br.). Truncus arteriosus continues
with the descending aorta (Ao. D.) through permeable ductus (Du). A.C.P.D.: right primitive
carotid artery. A.S.D.: right subclavian artery. A.S.I.: left subclavian artery. b) 3D reconstruction. Anterior oblique view. T.A.: truncus arteriosus. Du.: Ductus. Ao.D.: Descending aorta.
A.S.I.: left subclavian artery. c) 3D reconstruction. Posterior view. Lung arteries originate close
together from the back of the truncus arteriosus. A.P.D.: Right pulmonary artery. A.P.I.: left pulmonary artery. Ao.D.: Descending aorta. d) Surgical photography. T.A.: truncus arteriosus.
T.C.: common trunk. V.C.S.: superior vena cava.
Página 7
VD: Right ventricle.
VI: Left ventricle.
CIV: Ventricular septal defect.
TA: Truncus arteriosus.
APD: Right pulmonary artery.
API: Left pulmonary artery.
2009
Scheme 4: Illustrative scheme of truncus arteriosus type II.
Congenital abnormalities of aortic artery
a
b
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c
Figure 9. a) Male patient, 5 months old.
Progressive respiratory distress and stridor.
Double aortic arch with atresia of the left component. Coronal plane. Diverticulum of the
left atretic arch (arrow). A.S.D.: right subclavian artery. A.S.I.: left subclavian artery. b)
3D reconstruction. Superior oblique view.
Emerging arteries from the aortic arch emerge
each independently (Sign of the four vessels).
A.C.P.I.: Left primitive carotid artery.
A.C.P.D.: right primitive carotid artery.
A.S.I.: left subclavian artery. A.S.D.: right
subclavian artery. Diverticulum of the left atretic arch (arrow). c) Axial plane. Compression of
right main bronchus with signs of air trapping of that lung.
b
c
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a
Scheme 5: Illustrative scheme of incomplete
double aortic arch. Diverticulum corresponding to the permeable sector of the double arch
(long arrow). Fibrous cord that completes the
arch (arrowhead).
Figure 10. a) Female patient, 11 months old. Dysphagia and recurrent pneumonias. Axial plane.
Aberrant right subclavian artery with retroesophageal path. Airspace consolidations on the right
upper lobe with some hypodense sectors with aspiration pneumonia look. A.S.D.: right subclavian artery. T: Trachea. Dilated esophagus (arrow).
b) A.S.D.: right subclavian artery. Ao.D.: Descending aorta. c) 3D reconstruction. Posterior view. A.S.D.: right subclavian artery. A.S.I.: left subclavian artery. A.C.P.D.: right primitive carotid artery. A.C.P.I.: left primitive carotid artery. With surgery, it is performed a right posterolateral
thoracotomy, section of the aberrant artery at the base and its reimplantation in the ipsilateral carotid artery.
Página 8
anesthesia. Studies take between 20 – 40 minutes, longer than ACT.
The decision of which modality is to be used,
depends on the availability and experience of each
particular imaging service.
In our opinion, CT presents the advantage of
being extremely fast and easy to program, requiring
less complex anesthetic procedures.
CONCLUSION
Computed tomography angiography appears as
an extremely useful complementary method in the
diagnosis of congenital abnormalities of the aortic
arch in neonates and early childhood patients. It is a
fast and non invasive procedure, which takes short
anesthetic time and low charge of intravenous contrast. In our series, it enabled to diagnose all the studied anomalies in an efficient and precise way, with
excellent correlation to the surgery findings reported
by surgeons.
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