Journal of Cardiology and Cardiovascular Research
Chinawa JM, et al., 2022- J Cardiol Cardiovasc Res
ISSN: 2583-259X
Research Article
Aortic Valve Repair Following
Ventriculo-septal Defect (VSD) and
Severe Aortic Regurgitation: Surgical
Techniques,
Complications
and
Follow up Events
Josephat M Chinawa1*, Agarwal Vijay2, Sarang Gaikwad2 and
Bhadra Trivedi2
Abstract
Background
Aortic regurgitation (AR) is a common sequel resulting from
prolapsed of right coronary cusp (RCC), non-coronary cusp (NCC)
or both, in the repair of ventricular septal defects (VSD).
Objectives
The purpose of presenting these cases is to highlight the fact that
aortic valve repair does not only end in addressing the cusp as this
will result in moderate aortic regurgitation after surgery.
1College
of Medicine, Department of
pediatrics, University of Nigeria, University
of Nigeria Teaching Hospital (UNTH),
Ituku-Ozalla, Enugu State, Nigeria
2Fortis
Hospital limited, Mulund goregaon
Link Road, Bhandup (West), Mumbai, India
*Corresponding
Author: Josephat M
Chinawa, College of Medicine, Department
of pediatrics, University of Nigeria,
University of Nigeria Teaching Hospital
(UNTH), Ituku-Ozalla, Enugu State, Nigeria.
Received Date: 06-07-2022
Accepted Date: 09-22-2022
Published Date: 09-27-2022
Copyright© 2022 by Chinawa JM, et al. All
rights reserved. This is an open access article
distributed under the terms of the Creative
Commons Attribution License, which
permits unrestricted use, distribution, and
reproduction in any medium, provided the
original author and source are credited.
Results
Our first case is a two and half year old male with a large peri-membranous VSD with RCC prolapse causing
co-optation failure and severe AR .The second case is a 12 year old male a large peri-membranous ventriculoseptal defect with RCC prolapsed with severe AR .The third case is a 12 year old female with a large perimembranous VSD with a significant right coronary cusp prolapsed, severe aortic regurgitation due to noncooptation of RCC with the remaining cusps with flow reversal in the descending aorta. The last case is a four
year, 6-month-old male with a large peri-membranous VSD and severe AR from NCC and RCC prolapse. All
had repair of aortic valve with mild regurgitation after several follow up.
Conclusion
Aortic valve repair resulting from prolapse of RCC and NCC in children with VSD is expedient as it reduces
morbidity and mortality post-operation.
Chinawa JM | Volume 3; Issue 3 (2022) | Mapsci-JCCR-3(3)-067 | Research Article
Citation: Chinawa JM, Agarwal V, Gaikwad S, Trivedi B. Aortic Valve Repair Following Ventriculo-septal defect (VSD) and Severe Aortic
Regurgitation: Surgical Techniques, Complications and Follow up Events. J Cardiol Cardiovasc Res. 2022;3(3):1-7.
DOI: https://doi.org/10.37191/Mapsci-JCCR-3(3)-067
Keywords: Ventricular septal defects; Aortic regurgitation; Coronary cusp (RCC); Non coronary cusp (NCC).
Introduction
Aortic regurgitation (AR) with ventricular
septal defects (VSD) is an anatomic anomaly
resulting from prolapsed of right coronary
cusp (RCC), non-coronary cusp (NCC) or
both and presents in 4.5-11% of cases [1].
Aortic valves prolapse with ventricular septal
defect (VSD) are seen in over 5% of children
[2]. This anomaly is common with Subarterial VSDs and it occurs about five times
compared with peri membranous VSDs [2].
Aortic
regurgitation
resulting
from
underdevelopment of the aortic valvular
commissure, and that from deficiency of the
canal musculature are grouped into type 1 and
2 respectively [3]. There are several theories
proposed as the cause of aortic prolapsed in
VSDs. This ranges from deformity of the
cusps, structural defects of leaflets support,
weakness of the commissures and the issues
arising from the Venturi effect.
Venturi effect has been documented as a
single potent factor that could give rise to
development
of
aortic
regurgitation.
Identifying Venturi effect as an important
pathological mechanism in AR will help in
screening children with VSD who are at risk
of AR. This will predict both those patients in
whom surgery is indicated as well as the
timing of surgical intervention.
Surgical repair of the aortic valve from aortic
regurgitation (AR)in children with VSD has
gained ground over the last decade. Besides,
aortic valve repair still poses a serious
challenge especially in resource limited
countries like ours. Several surgical
maneuvers abound in the repair of the aortic
valve. This includes direct repair of damaged
leaflets, commissurotomy, re-suspension,
thinning and extension of the prolapsed
leaflet. However, complex repair often require
more innovative and advanced techniques.
The first surgical repair was done by Starr, et
al in 1960 [4]. Later, Trusler, et al. [5] and
Spencer, et al. [6] introduced the plication of
prolapsed leaflet using pledgeted mattress
suture. Chauvaud, et al. [7] and Carpentier
introduced triangular valvular resection with
very good outcome [8]. Transaortic repair of
prolapsed RCC with pledged sutures was
done by Yacoub, et al. [ 9].
The report of these case series is important to
enable the cardiologists and cardiac surgeons
have a high index of suspicion of possibilities
of VSDs especially sub-pulmonic VSDs to
present with aortic regurgitation and to be
equipped with skills to tackle the
management and understand the numerous
complications that follow this anomaly. The
purpose of presenting these cases is to
highlight the fact that aortic valve repair does
not only end in addressing the cusp as this
will result in moderate aortic regurgitation
after surgery. It is expedient therefore to
address every aspect from the ventriculoarterial junction to STJ in the approach of
aortic valve repair as this will always give a
better result of trivial regurgitation after
surgery. In addition, the size of aortic annulus
to be reduced is also very important as this is
often neglected among adult with AR. Early
identification and appropriate intervention
can significantly improve the quality of life.
Chinawa JM | Volume 3; Issue 3 (2022) | Mapsci-JCCR-3(3)-067 | Research Article
Citation: Chinawa JM, Agarwal V, Gaikwad S, Trivedi B. Aortic Valve Repair Following Ventriculo-septal defect (VSD) and Severe Aortic
Regurgitation: Surgical Techniques, Complications and Follow up Events. J Cardiol Cardiovasc Res. 2022;3(3):1-7.
DOI: https://doi.org/10.37191/Mapsci-JCCR-3(3)-067
Case presentation
Our first case is a 2 and half year-old male
who presented with frequent cough.
Examination showed pansystolic murmur 3/6
at mid sternal boarder while Echo showed
large peri membranous VSD with RCC
prolapse causing cooptation failure and
severe AR. The second case is a 12-year-old
male presented with recurrent respiratory
tract infections at infancy and dyspnoea on
exertion, NYC classification 2, PSM 4/6 in the
mid sterna boarder. Echo showed peri
membranous ventricular septal defect with
RCC prolapsed with severe AR. The third case
is a 12-year-old female admitted with a failure
to thrive and recurrent RTIs. Examination
showed pansystolic murmur at mid sternal
boarder. Echo showed large peri membranous
VSD with a significant right coronary cusp
prolapsed, severe aortic regurgitation due to
non-cooptation of RCC with the remaining
cusps with flow reversal in the descending
aorta. The last case is a four year, 6-monthold male who presented with recurrent
respiratory tract infection and failure to
thrive. Examination revealed a pan-systolic
murmur at the mid sternal boarder.
Echo showed a large peri membranous VSD
and severe AR from NCC and RCC prolapse.
The AR jet is along the line of cooptation of
the LCC with the NCC. There is also a jet
across the annulus at the RCC-NCC
cooptation line. Table 1-3 showed detailed
events of the subjects.
Surgical procedure
Standard midline sternotomy, thymectomy
and pericardiotomy were done. PDA was
dissected and ligated. Cardio-pulmonary
bypass was established with aorto-bicaval
cannulation after systemic heparinisation.
Both cavae snugged. Transverse aortotomy
was done. The heart was arrested by giving
delnido cardioplegia directly into both the
coronary ostia.
Main pulmonary artery opened, left heart
vented through left atrium. A patch closure of
VSD with 6 zero proline 10 mm predigested
suture was instituted. Main pulmonary artery
was closed using proline 6 zero sutures. Root
of aorta was dissected and released from
surrounding tissues. Semi-circumferential
annuloplasty was done from NCC to LCC
using a strip of Teflon. RCC and NCC were
suspended to their respective commissures
and RCC and NCC leaflet edges were plicated
using 7 zero proline 10mm pledgeted sutures.
Saline test done was found to be satisfactory.
Immediate intra operative trans-thoracic
echo showed moderate AR.
In case two however, Bovine pericardium was
then anastomosed onto annulus using 6 zero
proline sutures.
In our third case, Truslers repair done on the
prolapsed NCC leaflet by placating the NCC
leaflet edges to the respective commissures
with 6 zero 13mm pledgeted sutures. Truslers
repair of RCC was done with 6 zero proline
sutures. Saline test was found to be
satisfactory. The right and left coronary
buttons were excised from respective
coronary sinuses. The aortic root was
dissected and released from surrounding
tissues. Annuloplasty was done with 5 zero
proline sutures.
Coronary buttons were reattached to
respective sinuses with 6 zero proline sutures.
Chinawa JM | Volume 3; Issue 3 (2022) | Mapsci-JCCR-3(3)-067 | Research Article
Citation: Chinawa JM, Agarwal V, Gaikwad S, Trivedi B. Aortic Valve Repair Following Ventriculo-septal defect (VSD) and Severe Aortic
Regurgitation: Surgical Techniques, Complications and Follow up Events. J Cardiol Cardiovasc Res. 2022;3(3):1-7.
DOI: https://doi.org/10.37191/Mapsci-JCCR-3(3)-067
coronary below the RCA channel. A portion of
the aortic annulus below the LMCA was
excluded from the annuloplasty. Immediate
intra-operative transthoracic echo showed
trivial AR.
The sinu-tubular junction plasty was done
using Teflon felt to buttress the end-to-end
anastomosis of the transected aorta with 6
zero proline sutures.
In our fourth series, a sub-coronary channel
was created below RCA and LMCA for aortic
annuloplasty. A partial circumferential
annuloplasty was done using a strip of Teflon
placed all around the aortic root and sub-
The mean cross clamp time, cardiopulmonary
bypass time and mean stay in intensive care
unit are 124 ± 16.51, 142.25 ± 28.19 mins and
6.66 ± 0.57 days respectively.
Characteristics
Frequency (%); and mean (SD)
Gender
Male
3(75%)
Female
1(25%)
Age in years
7.75 (4.97)
CPB and cross clamping time
Mean (SD) in (mins)
Cross clamp time
124 ± 16.51
CPB time
142.25 ± 28.19
Mean stay in intensive care unit
6.66 ± 0.57 days.
Table 1: Characteristics and socio-demographic features of subjects and surgical events.
No of
cases
Features
Cough (PSM 3’6)
Failure to thrive (NYC 2,
4’6)
3
Echo report
Large PM VSD, RCC prolapse and Coapt failure sAR,
4
Large PM VSD, RCC prolapse, sAR, Coap failure
Recurrent RTIs (PSM 3’6)
4
Large PM VSD, NCC and RCC prolapsed, sAR,
Table 2: Showing clinical features and summary of preoperative Echo results.
2nd follow
up
3rd follow
up
Case 1
First Follow
up
Moderate
AR
Mild AR
Mild AR
Case 2
Mild AR
Mild AR
Mild AR
Case 3
Mild AR
Mild AR
Mild AR
Case 4
Mild AR
Mild AR
Mild AR
4th follow
up
5th Follow
up
Mild AR
Mild AR
Table 3: Showing follow up events of patient with respect to Aortic regurgitation.
Chinawa JM | Volume 3; Issue 3 (2022) | Mapsci-JCCR-3(3)-067 | Research Article
Citation: Chinawa JM, Agarwal V, Gaikwad S, Trivedi B. Aortic Valve Repair Following Ventriculo-septal defect (VSD) and Severe Aortic
Regurgitation: Surgical Techniques, Complications and Follow up Events. J Cardiol Cardiovasc Res. 2022;3(3):1-7.
DOI: https://doi.org/10.37191/Mapsci-JCCR-3(3)-067
Discussion
Closure of VSD does not suffice in children
with moderate to severe aortic regurgitation.
Re-operation may be expedient in many
children because of progressive aortic
regurgitation [10]. If the surgery is deferred till
adulthood, repair may be difficult. It is
pertinent to do an intra-operative assessment
of the aortic valve in all children suspected
with aortic regurgitation.
Valve repair is the treatment of choice in
children with aortic valve regurgitation.
However, this may not be successful. The
plication of prolapsed leaflet on the aortic
wall has been proposed by Trusler, et al. [10]
However, recurrence has been noted as a
common sequel in this type of surgery. This
may cause residual regurgitation [5].
Nevertheless, our reportage showed mild or
no regurgitation. This remained the same
after a long follow up. Besides, Kalangos, et al.
[11] noted the use of thin pericardial strip
before plication at free margin of the leaflet,
this technique provides a balance to the stress
at
commissural
site
with
reduced
incompetence in a long term. Two of the
patients were given same technique and good
outcome was attained. This method of repair
highlighted above will help in preventing the
growth of leaflets in children. Long term
results are reported to be satisfactory despite
different results of aortic valvuloplasty in
different centers [12-14]. This is also seen in
our series.
The exact measurement of the effective leaflet
height suspension is a very important during
surgery
and
this
determines
valve
competence. In this report, the release of the
cross-clamp during repair determines the real
test of competence.
The association of ventricular septal defect
and aortic regurgitation connotes a poor
prognosis, however, with current trends in
open heart surgery, total correction is being
more widely advocated.
Nevertheless, total repair is difficult, and a
good knowledge of the structure and function
of the aortic valve is crucial. We attained
complete success in all the series, all
presenting with mild to no aortic
regurgitation at follow up.
The age of presentation of our cases is
between 2 to 12 years. The exact age
prevalence of AR is unknown; however, this is
rare before the age of 2. Once AR develops, it
worsens in severity within ten years. There
may be episodes of Aneurysm of the sinuses
of Valsalva in sub-pulmonic VSD.
Over two and half years, the unit has done
1000 pediatric surgical case, of which 400
were VSD closures, out of which only 4 had
VSD with severe regurgitation giving a
prevalence rate of 1%. This was less than that
of Hasan, et al. [15] which revealed that out of
314 patients with VSD closure, 9 patients had
severe AR giving a prevalence of 2.86%. Our
finding is also less than 4.5-11% seen in the
general population.
There is male dominance as seen in this study.
This was in tandem with Hasan’s study but
however different from that of Francois, et al.
who noted no difference in gender. The
reason for this preponderance could be the
general reason seen among males been more
Chinawa JM | Volume 3; Issue 3 (2022) | Mapsci-JCCR-3(3)-067 | Research Article
Citation: Chinawa JM, Agarwal V, Gaikwad S, Trivedi B. Aortic Valve Repair Following Ventriculo-septal defect (VSD) and Severe Aortic
Regurgitation: Surgical Techniques, Complications and Follow up Events. J Cardiol Cardiovasc Res. 2022;3(3):1-7.
DOI: https://doi.org/10.37191/Mapsci-JCCR-3(3)-067
afflicted with congenital heart disease than
females.
Postoperative course in all our cases were
good with all having mild aortic regurgitation
on follow up with no post operative morbidity
and mortality. This finding is also noted by
Hasan, et al. [15] who neither found any
mortality nor atrioventricular block. There
was no significant aortic regurgitation seen in
this study post-operation. There were no
complications from mechanical valve after a
long follow up.
Regarding children with VSD who had aortic
regurgitation, the route of surgery and the
technique of VSD closure are crucial. Our
cases were repaired by the aortotomy or via
right atrial route in the first place. There were
no reported residual VSD in our cases on
follow-up. Aortic valve should be replaced
without hesistance if aortic valve repair is not
successful. Literature had shown that on a
long-term
follow-up,
left
ventricular
functions and functional capacity may show
significant improvement after aortic valve
repair and replacement. This also akin to our
series.
Echo showed that all our cases, but one was
peri membranous VSD. This conflicts with
earlier report that sub arterial VSDs are more
liable to aortic regurgitation compared to peri
membranous VSD.
Conclusion
Aortic valve repair resulting from prolapse of
RCC and NCC in children with VSD is
expedient as it reduces morbidity and
mortality post-operation.
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Chinawa JM | Volume 3; Issue 3 (2022) | Mapsci-JCCR-3(3)-067 | Research Article
Citation: Chinawa JM, Agarwal V, Gaikwad S, Trivedi B. Aortic Valve Repair Following Ventriculo-septal defect (VSD) and Severe Aortic
Regurgitation: Surgical Techniques, Complications and Follow up Events. J Cardiol Cardiovasc Res. 2022;3(3):1-7.
DOI: https://doi.org/10.37191/Mapsci-JCCR-3(3)-067
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Chinawa JM | Volume 3; Issue 3 (2022) | Mapsci-JCCR-3(3)-067 | Research Article
Citation: Chinawa JM, Agarwal V, Gaikwad S, Trivedi B. Aortic Valve Repair Following Ventriculo-septal defect (VSD) and Severe Aortic
Regurgitation: Surgical Techniques, Complications and Follow up Events. J Cardiol Cardiovasc Res. 2022;3(3):1-7.
DOI: https://doi.org/10.37191/Mapsci-JCCR-3(3)-067