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1071
Late Complications at Repair Site of Operated Coarctation of Aorta .
Robert David Elizabeth
A. Clark1 P. Colley Siedlecki
Late complications at the repair site of coarctation of the aorta include aneurysms and persistent coarctation. Aneurysms are usually due to failure of the surgical anastomosis and can occur many years after repair. Continued aortic coarctation may be due to either repair failure Three cases are presented
or failure of the anastomotic and the literature reviewed.
site to grow
with the patient.
Successful connective surgery for coanctation of the aorta has been performed since 1 945 [1 , 2]. The immediate and short-term effects and complications of surgery have been discussed [3, 4], but the long-term effects and late complications have appearances 8], but
been recognized of the postoperative
the appearances
of this
Case
Reports
Case
of late
aortic
complications
Similarly, have have
the radiographic been described
received
three
little
cases
of the
[7,
attention
of late aortic
aorta
forms
the
report.
1
H. M., a 59-year-old and 13
recently [5, 6]. repaired coanctation
literature [9, 1 0]. Our experience with at the repair site of operated coanctation
in the nadiologic complications basis
only
3 weeks years
coarctation
man,
was admitted
of nonproductive
earlier
using
cough.
a Dacron
to the hospital
An
aortic
prosthetic
for evaluation
coarctation
graft.
was
He had been
of hypertension
diagnosed
otherwise
and
well
repaired
since
his
repair.
His blood pressure was 1 80/1 1 0 mm Hg in the right arm, 1 76/ 1 1 0 mm Hg in the left arm, and 1 50 mm Hg palpable systolic in the right leg. Physical examination, including the arterial Received March 1 3, 1 979; accepted vision July 1 7, 1 979. All authors: Department of Radiology, .
nati,OH45267Addressrepiintrequests AJR
133:1071
-1075,
December
1979
$00.00 Ray Society
ne
CincintoR.A
Clark. 0361 -803X/79/1336-1071 © American Roentgen
after
pulses,
was
otherwise
normal
-Preoperative chest radiography left subclavian artery as well as radiography (fig. 1 8) showed pleural
years later (fig. 1 C) demonstrated the coarctation repair site. There revealed a 5 cm aneurysm (fig. 10). The ascending gradient across the repair
except showed
prominent and
for
a healed
typical lung
incision
dilatation
rib notching fibrotic
changes
over
only.
left
chest.
Chest
radiography
13
a large mass in the left chest adjacent to the region of were no calcifications or cavities. Thoracic aortography
of the aorta distal to the left subclavian aorta and aortic valve were normal. site.
the
of the prestenotic aorta and (fig. 1 A). Postoperative chest
artery at the repair site There was no pressure
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1072
Fig. Diffuse thoracic
CLARK
1 -Case postsurgical aontognam.
Surgery proach.
was
performed
A false
subclavian Dacron
were
replaced patients
through
aneurysm
artery.
vious
The
1 . A, Marked rib notching pleural and parenchymal Lange aneurysm (arrow)
graft
Within
of the
prosthesis.
postoperative
a left aorta
Resected
Dacron course
AL.
AJA:133,
(small arrows) and dilatation of aontic arch and left subclavian artery (large fibrotic changes. C, 1 3 years after repair. Soft tissue mass (arrow) adjacent distal to left subclavian artery at repair site.
the aneurysm
with a knitted
ET
lateral
was
thoracotomy
found
distal
to the
was incorporated aorta
Debakey was
and
ap-
tory
left
managed
and
discharged
4 months
after
surgery.
the pre-
aneurysm
(9 cm)
graft.
complicated
difficulties,
Case
atelectasis,
in preoperative
site. D, Right
hypoxia,
3 weeks
after
but
coarctation.
posterior
he was
surgery.
1979
B,
oblique
successfully
He was
doing
well
2
J. M. , a 55-year-old by respira-
and
arrow)
to graft
December
of cough
and
chest
woman, pain
and
was 1 day
admitted
because
of heinoptysis.
of 6 weeks Coarctation
of
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AJR:133,
December
COMPLICATIONS
1979
AFTER
REPAIRED
A
AORTIC
COARCTATION
1073
B
Fig. 2.-Case 2. A, Admission radiograph. Mass adjacent to aorta in left chest subsequently died, and this nepresented ruptured false aneurysm at repair site.
the aorta was diagnosed and repaired 1 0 years earlier. Her blood pressure was 220/1 40 mm Hg in both arms and her pulse was 128 beats /min. Physical examination, except for a healed left thoracotomy incision, was otherwise normal. Admission chest radiography (fig. 2a) showed a large mass in the left chest adjacent to the aorta. After 24 hr, her chest pain suddenly increased and she became hypotensive. Repeat chest radiography (fig. 2B) showed extension of the mass density into the left chest and a left pleural effusion . Cardiopulmonary collapse ensued, resuscitation maneuvers failed, and the patient died. Autopsy revealed a false aneurysm at the coarctation repair site that had ruptured into the mediastinum and left chest.
Case
3
J. J. , a 40-year-old woman, underwent repair of an aortic coarctation 25 years earlier. Routine chest radiography showed a soft tissue density above the aortic knob presumed to represent the left subclavian artery (fig. 3A). Her blood pressure was 1 60/90 mm Hg in the right arm, 1 40/90 mm Hg in the left arm, and 1 1 0/80 mm Hg in the left leg. Physical examination was otherwise normal. Because of the difference in blood pressure measurements and the abnormal chest film, she was referred for angiography. Aortography (fig. 3B) revealed a three-cusp aortic valve without regurgitaton.
The
aortic
arch
was
slightly
hypoplastic
at the
level
of
the left carotid artery. There was a 2 cm x 1 1 mm stenosis of the aorta distal to an enlarged left subclavian artery. Prominent collateral vessels from the subclavian arteries and internal mammary arteries bridged the stenosis. The aorta distal to the stenosis was dilated to about 6 cm (fig. 3C). Intraarterial
pressure
measurements
were:
prestenotic,
145/85
10 years
mm
Hg
with
after
with
coarctation
a mean
a mean
stenosis,
of 1 25
95
mm
B, 24 hours
mm
Hg;
Hg;
intervention
was
later.
Mass
poststenotic,
and
30 mm Hg. The patient
surgical 1 year
of
repair.
pressure
enlarged.
1 00/85 gradient
and her physician
necessary,
and
she
Patient
was
mm
across
decided still
Hg the
that no
asymptomatic
after angiography.
Discussion In the
several
large
reported
series
ment of postoperative coanctation, usually related to an associated one
[1 1 1’ 68%
series
of the clinical
of the
patients
had
one
associated congenital cardiovascular defects, common being patent ductus anteniosus, with mitral valve lesions following in frequency. There incidence of premature 5.7% [1 1 ] to 1 2% [5] Late aontic aontic aneurysm of operated
patients
coanctation,
which
1 1 ]. Since
the
and
Development aortic
coarctation
4], and
occur
in 6%-i
of patients the
true
death, patients.
are infrequent. which occur
[4, 5, 1 2-1 may
majority
are lost to follow-up, tions may be higher.
Aneurysm
cardiovascular of all operated
complications on dissection,
assess-
late complications were cardiovascular anomaly. In
with
incidence
ranging They in less
residual 0%
or
more
the most aortic and was a high from
include than 1 %
on recurrent of cases
repaired
[5,
7,
coanctation
of these
complica-
Dissection of an aneurysm is unusual
after and
surgical
angiographic
connection documenta-
of
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1074
CLARK
ET
AL.
AJR:133,
Fig. 3.-Case 3. A, Routine film 25 years after coanctation repair. Enlarged left subclavian artery (arrow). B, Thoracic aortogram, right axillary approach. Residual or recurrent coanctation distal to left subclavian artery (straight arrows). Collateral vessels eminate from subclavian arteries (curved arrows). C, Thoracic aortognam. tnansfemoral approach. Extent of stenotic segment (arrows) demarcated. Pressure gradient across stenosis was 30 mm Hg.
tion
has
been
reported
in few
of the patients, or ascending
the aorta
case
it was
associated
aortic
regurgitation.
Angiognaphy
have made
precise
However,
several
aortic
coanctation
effects increase
aneurysm has been
repair
0
since
and
valves
are
of the
wall
and
most
pa-
some dilatation while about 40%
aorta There forces
aortic
hypertension present
aneurysm,
dis-
is unknown.
contribute valve, [1 5].
‘*a.
to this
dilatation of patients
are usually
associated
is evidence that the dilatation on the aortic wall by turbulent abnormal and
may
valve weaken
[1 8]. the
% ,;.**i
of
Congenital
in up to 85%
[1 7] and
structurally tension
or dissection. Rupnoted [5, 1 1 , 1 5].
aortic factors
of a bicuspid
aorta,
of the
valve
of coanctation
hemodynamic
aortic dilatation. results from abnormal forces
aortic
to interpret
of ascending
after
with
flow
In most
[1 6]. Therefore, diagnosis is best of increased dilatation on succes-
presence
ascending
bicuspid with
be difficult
etiology
or rupture the
a bicuspid
on localized of aneurysm
section, lesion:
9, 1 0, 1 5].
postoperatively demonstrate sinuses and ascending aorta
sive examinations, tune of this type
the
with
aontic regurgitation by documentation
The
[7,
1979
(
aneurysm occurred at the aortic sinuses 4-i 0 years postoperatively, and in each
may
tients studied of the aortic
cases
December
These
structural
,
mechanism. this
In contrast
type
is most
surgical
rysm site
have
dissection
necessary
(similar
on true
but these
other
types
of
aneurysm,
aneurysm reports
due to failure of of mycotic aneu-
aneurysm
[21]
were
related
at the repair
to failure
line. There has been one previous report at the site of a Dacron graft prosthesis
to case 1 ). The for ascending dilatation,
the
a false Occasional
[9],
appeared,
Aneurysms 1 and 2) are
aortic
to
often
anastomosis.
[20],
at the suture false aneurysm
at the coarctation repair site [9, 10, 20] (cases much less common and occur by a different
.
C
of the aorta, favoring the occurrence of aneurysm, dissection, on rupture [1 5]. Hypertension contributes to the propagation of the lesion [19]. integrity
.
do
not
or leak of a [22]
predisposing hemodynamic factors aortic aneurysm, such as bicuspid apply
to aneurysms
at the
repair
AJA:133,
December
COMPLICATIONS
1979
AFTER
REPAIRED
AORTIC
studies
site. However, hypertension may contribute to the development of the lesion. It was present in both cases 1 and 2, and in case 2 it may have led to aneurysm rupture. The time
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period
from
repair
to diagnosis
of the
aneurysm
in this
or Recurrent
This still
type
the patient
24].
years
optimum with
In some failure,
of operation
for elective
and
congestive
site
to the
heart
failure
to
grow
is indicated
the
to failure
patient
[6].
[6,
7].
Almost all repaired ing at the anastomotic most ence
In our
case
mm Hg has for 1 year.
been
residual
managed
pressure
in the presanastomotic
to visualize. Therefore, simultanemeasurements are mandatory in
evaluation
of the
postoperative
chest
film
coanctation
sign is
that
may
coarctation
dilatation
of
help the
in suspecting left
subclavian
artery (case 3). In one series of 1 56 postoperative coanctation chest radiographs, this finding was present in only two cases [1 1 ]. Since its presence is unusual in postopencoanctation,
it may
residual stenosis, hypertension. Continued
to residual majority tation atomic
blood
due to persistent
flow
in coarctation
Hartmann
AF,
Goldring
0, Hernandez
around
A, Behrer
MA,
Schad
N,
from
thoracic Radio!
aortic
surgery-congenital
North
Am
Clln
C, Davignon
9:253-255,
and
acquired
1971
A, Ethier MF: Coarctation
Unger
11.
1 2.
1 3.
EL,
of the aorta in
Marsan
RE:
Ruptured
Cardiovasc
aneurysm
20
after
years
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1 4.
J Thorac
follow-up.
T: Coarctation
Scand
material.
of the
J Thorac
aorta-a
Cardiovasc
follow-up
of op-
Surg (Suppl]
9:1 -
of coarctation
of
1973
Owens
J, Swan
the aorta.
H: Complications
in the repair
J Cardiovasc
Surg (Torino) 4:81 6-825, 1963 1 5. White EW, Zoller RP: Left aortic dissection following repair of coarctation of the aorta. Chest 63:573-577, 1973 1 6. Karnell J: Coarctation of the aorta. Circulation 38 [suppl 5]: 1968
7. Edwards JE: The congenital bicuspid aortic valve. Circulation 23:485-488, 1961 1 8. Holman E: The obscure physiology of post-stenotic dilatation: its relation to the development of aneurysms. J Thorac Car1
1 9.
diovasc
Surg
28:1
Prokop
EK,
Palmer
33,
RF,
aneurysms
dissecting
prestenotic
09-1
1954
Wheat
(abstr).
MW:
Hydrodynamic
Circulation
in 6]:1 58,
forces
38 [suppl
1968
systemic
hypertension
of patients etiology
collateral
or dilatation
coarctation,
repair
suggest
pressures
JO, Rowe RD, Mellits ED: Prognosis of coarctation of the aorta: a 20 year postCirculation 47:1 1 9-1 26, 1973
Humphries
corrected appraisal.
35-44,
plain
persistent
ative
1 0.
successfully
[7]. Conversely, repair site, an
BJ,
surgically operative
Pelletier
considered
aortas will demonstrate some narrowsite, as seen by angiognaphy, but
stenosis may be difficult ous intraarterial pressure the angiographic
3, the
been
will produce no gradient of an aneurysm at the
patient. One
have
blood
Surg 57:171-179, 1969 9. Steinberg I, Stein HL, Goldberg HP: Aneurysms complicating the postoperative course of coarctation of the aorta. Report of 3 cases. AJR 93:331 -338, 1965
of the Pressure
residual coarctation [5, 6, 8, 1 1 ], and gradients mm Hg or more are considered an indication for of 30 surgery
Hg on more
8.
in
significant of 40-50
repair
Maron
diseases.
due to repair
due
JO: Postoperative
Br Heart J 26:671 -678, 1964 SR, Gross RE: Surgery for coarctation of the aorta, of 500 cases. J Thorac Cardiovasc Surg 43:54-69,
infancy-postoperative
of 1 0-20
gradient without
5.
suIting
[23,
gradients
surgical
mm
with
of the
[6].
it is recurrent
233:
Ferguson T, Burford T: Recurrent coarctation of the aorta after successful repair in infancy. Am J Cardiol 25:405-410, 1970 7. Fleming RJ, Steinberg I, Baltaxe HA: Radiologic findings re-
is said to be 10-15
is residual
J Med
aorta.
Schuster a review
6.
of
pressure grainitially operated
segments
N EngI
correction.
1962
but age
the status
or childhood
the coarctation
in others
anastomotic
repair
in infancy
patients,
while
aneurysm,
or had long hypoplastic
age
[1 6], but repair
patients
than is related
Most patients with a significant a residual coanctation were
age 6 months The
frequent
Its development
at the time
coanctation. dient across
before
is more
[7].
its surgical
1945
L, Keith
4.
Coarctation
complication
uncommon
Rathi of the
of lesion has ranged from 0.5 [5] to 20 years, and rupture has occurred 20 years after repair of the coanctation [10]. Residual
regarding
287-293, 3.
1075
COARCTATION
have [5,
with ‘
after
repair
but it is not a reliable continued
‘essential’
‘
hypertension hypertension
may
sign. after without
be due
20.
Martin W, Kirklin mal endarteritis
The vast coancan an-
21].
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871-874, 1956 Ross JK, Monro
J, Dushane J: Aortic after resection for
aneurysm coarctation.
and
aneurys-
JAMA
160:
22.
CG: Late complications of surgery Thorax 30:31 -39, 1975 Berroya A, Aleman J, Mannix E: A long-term failure of prosthetic graft in coarctation. J Cardiovasc Surg 1 1 :329-332, 1970
23.
Khoury
24.
infancy Taimes
21
.
for coarctation
GH,
JL, Sbokos
of the
Haines
aorta.
CR:
Recurrent
coarctation
of the
aorta
in
and childhood. J Pediatr 72:801 -806, 1968 RL, Aberdeen E, Waterston DJ, Carter RE: Coarctation of the aorta in infants and children. Circulation 39 [suppl 6]: 173-184, 1969