OUTCOME OF AORTIC VALVE REPAIR FOR AORTIC...

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OUTCOME OF AORTIC VALVE REPAIR FOR AORTIC

REGURGITATION COMPLICATING VENTRICULAR

SEPTAL DEFECT – A RETROSPECTIVE OBSERVATIONAL

STUDY

Thesis submitted for the partial fulfilment for the

requirement of the

Degree of MCH (Cardiovascular and thoracic Surgery)

BY

DR. S.P.MAHENDRANATH

MCH CARDIOVASCULAR AND THORACIC SURGERY RESIDENT

2015-2017

DEPARTMENT OF CARDIOVASCULAR AND THORACIC

SURGERY

SREE CHITRA TIRUNAL INSTITUTE FOR MEDICAL

SCIENCES AND TECHNOLOGY, TRIVANDRUM

KERALA, INDIA - 695011

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DECLARATION

I hereby declare that this thesis entitled “OUTCOME OF

AORTIC VALVE REPAIR FOR AORTIC REGURGITATION

COMPLICATING VENTRICULAR SEPTAL DEFECT– A

RETROSPECTIVE OBSERVATIONAL STUDY” has been

prepared by me under the capable guidance of Dr.Baiju S

Dharan, Professor, Department of Cardiothoracic and

Vascular Surgery, Dr.Sabarinath Menon, Associate

Professor, Department of Cardiothoracic and Vascular

Surgery, Dr. K. Jayakumar, Senior Professor, Head of the

department of Cardiothoracic and Vascular Surgery, at Sree

Chitra Tirunal Institute for Medical Sciences & Technology,

Thiruvananthapuram.

DR. S.P. MAHENDRANATH

MCH Cardiovascular &

Thoracic Surgery Resident

Department of Cardiovascular

& Thoracic Surgery

Place : Thiruvananthapuram

Date :

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CERTIFICATE This is to certify that the thesis entitled “OUTCOME OF AORTIC

VALVE REPAIR FOR AORTIC REGURGITATION

COMPLICATING VENTRICULAR SEPTAL DEFECT–A

RETROSPECTIVE OBSERVATIONAL STUDY” for the degree of

MCH(CARDIOVASCULAR AND THORACIC SURGERY) has been

carried out by Dr. S.P.MAHENDRANATH under our direct

supervision and guidance.

The work done in connection with this dissertation has been

carried out by the

candidate himself and is genuine.

GUIDE:

Dr. BAIJU S DHARAN

Professor

Department of Cardiovascular and

Thoracic Surgery,

SCTIMST, Thiruvananthapuram, India

CO-GUIDE: CO-GUIDE:

Dr. SABARINATH MENON Dr. K. JAYAKUMAR

Associate Professor Senior Professor and Head

Department of Cardiovascular Department of Cardiovascular

and Thoracic Surgery and Thoracic surgery.

SCTIMST, Thiruvananthapuram, India SCTIMST

Thiruvananthapuram, India

iv

SREE CHITRA TIRUNAL INSTITUTE FOR MEDICAL

SCIENCES

AND TECHNOLOGY

Thiruvananthapuram, India

CERTIFICATE

This is to certify that this thesis entitled “OUTCOME OF

AORTIC VALVE REPAIR FOR AORTIC REGURGITATION

COMPLICATING VENTRICULAR SEPTAL DEFECT – A

RETROSPECTIVE OBSERVATIONAL STUDY” has been prepared by

Dr. S.P. MAHENDRANATH, MCH Cardiovascular and thoracic

surgery Resident, Division of Cardiovascular and thoracic Surgery

at Sree Chitra Tirunal Institute for Medical Sciences & Technology,

Thiruvananthapuram. He has shown keen interest in preparing

this project.

Dr. K JAYAKUMAR

Senior Professor, Head of the department

Department of Cardiovascular

and Thoracic Surgery

SCTIMST, Thiruvananthapuram

Place: Thiruvananthapuram Date:

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ACKNOWLEDGEMENT

I take this opportunity to express my deep sense of gratitude and

thanks to all those who have been instrumental in the successful

completion of this work. I am deeply indebted to my esteemed teacher

and guide, Dr.Baiju S Dharan, Professor, Department of Cardiothoracic

and Vascular Surgery, Sree Chitra Tirunal Institute for Medical Sciences

and Technology for her unremitting encouragement, avid support and

invaluable guidance throughout the course of my study.

I would also like to express my deep gratitude and regards to my co-

guide, Dr.Sabarinath Menon,, Department of Cardiothoracic and

Vascular Surgery at, Sree Chitra Tirunal Institute for Medical Sciences

and Technology. Without his guidance, support and personal

involvement, the work could never have been completed.

I express my sincere thanks to my co-guide Dr.K.Jayakumar for his

constant support and guide throughout the conduct of study.

All my teachers Dr. Vivek Pillai, Dr Vargheese T Panicker, Dr.Bineesh R,

Dr.Sudip Dutta Buruah and Dr.Sowmia Ramanan, have been extremely

helpful during the conduct of the study and have given their invaluable

opinion and constructive criticism.

My sincere thanks to my fellow residents, Dr.Abid Iqbal, Dr.Debabrata

Gohain, Dr.Nikhil. P.K and Dr.Prakash Goura for their help throughout

the study period.

I would like to take this opportunity to show my gratitude to all my

patients who volunteered to be part of this study.

DR. MAHENDRANATH S P

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CONTENTS

SL.NO TOPIC PAGE

NUMBER

1 INTRODUCTION 1

2 AIM AND OBJECTIVE 3

3 REVIEW OF LITERATURE 4

4 MATERIALS AND METHODS 21

5 RESULTS 26

6 TABLES AND CHARTS 35

7 DISCUSSION 43

8 CONCLUSION 50

9 BIBLIOGRAPHY 51

10 ABBREVIATIONS

11 APPENDICES

DATA COLLECTION FORM

IEC APPROVAL FORM

MASTER CHART

PLAGIARISM CHECK FORM

1

INTRODUCTION

Aortic regurgitation (AR) in patients with ventricular septal

defect (VSD) occurs in those anatomical types that are located close

to or in direct contact with the aortic valve1. This subset of patients

form a relatively rare group of patients with an incidence ranging

between 5% and 10%.Perimembranous and doubly committed juxta-

arterial defects are the commonly affected types. Rarely, it can occur

in outlet muscular VSD also2.

Prolapse of the valvular cusps caused by the venturi effect of the

ventricular septal defect jet, abnormal commissural suspension, lack

of appositional forces and lack of continuity between the aortic

media, annulus of aortic valve and the ventricular septum have been

the commonly proposed pathological anatomic mechanisms of

regurgitation3. The right coronary cusp (RCC) is the usually involved

cusp. This is followed in line by the non-coronary cusp whereas, the

involvement of the left coronary cusp is very rare. The prolapsing

cusps render the valve incompetent and cause significant damage to

the valve cusp and annulus.The prolapsing AV may completely close

the VSD so that the shunt may disappear but leaving behind the

regurgitation to progress3.

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Surgical closure of the defect with aortic valve repair is

indicated for these patients irrespective of the grade of preoperative

aortic regurgitation and it is recommended to close the defect at the

earliest4. The technique of valve repair is decided based on the

mechanism(s) of regurgitation5

Despite increasing interest in pediatric aortic valve repair aortic

valve replacement is unavoidable in certain circumstances. With

limited options for valve replacement and lack of prospective studies,

the superiority of one prosthesis over the other cannot be

ascertained6. Metaanalysis has shown that all the currently available

valve substitutes are associated with suboptimal outcome in

children6. Hence a durable aortic valve repair is the need of the hour.

This study aims to evaluate the outcome of patients who

underwent aortic valve repair procedures for aortic regurgitation

complicating VSD and to also to assess the predictors for their

outcomes after surgery.

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AIM AND OBJECTIVE: 1. To identify the demographic parameters affecting a durable

valve repair

2. To assess the effect of preoperative disease severity on disease

recurrence

3. To compare the outcomes of various surgical techniques of

aortic valve repair for AR complicating VSD on short term

follow up

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REVIEW OF LITERATURE

EPIDEMEOLOGY

Ventricular septal defect accounts for 25 % of the acyanotic

congenital heart disease7. The true incidence of ventricular septal

defect in the general population is unpredictable because most of

them close spontaneously in utero. The incidence ranges from 5

to 50 per 1000 live births and 0.3 per 1000 adults8.

Among the patients with VSD the incidence of aortic cusp

prolapse may range between 4% and 9 % and the incidence of AR is 2-

6 %9 . The reported incidence of cusp prolapse in subarterial VSD is

40 % and in perimembranous VSD is 12 %10 . The incidence of AR is

related to the age . It is rarely present at birth but usually develops

after 2 years of age and progresses over the second decade to severe

AR

MORPHOLOGY

THE INTERVENTRICULAR SEPTUM

The morphology of right ventricle in patients with normal

atrioventricular connection is usually divided into three segments

using a tripartite approach. It includes the inlet, apical (trabecular)

and outlet components. The inlet (atrioventricular) septum is located

immediately beneath the septal leaflet of tricuspid valve. The

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membranous septum is located immediately beneath the anteroseptal

commissure. It is divided into the atrioventricular part between the

left ventricle and the right atrium and the interventicular part located

between the right and the left ventricles. This division of the

membranous septum is anatomically by the hinge of attachment of

the tricuspid valve. The conal or the outlet septum is located just

beneath the inter leaflet triangle between the RCC and the NCC. It is

wedged between the left anterosuperior and right posteroinferior limbs

of the trabecula septomarginalis (TSM)from below.

PERIMEMBRANOUS VSD

Perimembranous VSDs are the most common type of VSD

accounting for approximately 80% of patients operated on for primary

VSD11. These defects are also called junctional VSDs as they are

located in the junctional area between the trabecular and outlet

(conal) portions of the ventricular septum and between the posterior

and anterior divisions of the trabecula septomarginalis (septal band).

Perimembranous VSDs can be juxtatricuspid (abutting the

tricuspid valve), juxtamitral and juxta-aortic12. These

perimembranous VSDs are conoventricular in nature and they about

the commissural area between the noncoronary and right coronary

cusps of the aortic valve whereas the other VSDs are only

juxtatricuspid. In hearts with these as well as in hearts with

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perimembranous VSDs, the bundle of His passes along the

posteroinferior border of the defect.

OUTLET SEPTAL VSD (DOUBLY COMMITTED SUBARTERIAL VSD)

The defect within the outlet portion of the RV account

for approximately 5% to 10% of patients treated operatively for

VSD. VSDs in this location are also in the outlet portion of the LV. In

contrast to perimembranous VSDs, they are more beneath the right

aortic cusp than the commissure between it and the noncoronary

cusp.

VSDs in this area are bordered by a space over which lie the

pulmonary and aortic valve13. VSDs of this type are more common in

Asians than in white or black races14. Subarterial VSDs may be

circular, diamond shaped, or oval with the long axis lying

transversely. When viewed from the LV aspect, these defects are in the

outflow portion of the ventricular septum beneath the right coronary

cusp (or commissure between it and the left cusp). The aortic and

pulmonary valve cusps are separated by only a thin rim of fibrous

tissue. The right aortic cusp and (less often) noncoronary cusp may

prolapse into the upper margin of the defect, with or without aortic

regurgitation.

The postero inferior margin of RV outlet VSDs are well

separated from the tricuspid valve annulus by a band of muscle. As a

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result, they are well above the bundle of His and the conduction

system is related to such a VSD as it is with the other

perimembranous defects. Outlet VSD may sometimes be associated

with severe overriding of the aorta, and it is then termed double outlet

right ventricle (DORV) with doubly committed VSD. The same type of

VSD may also be seen in double outlet left ventricle (DOLV), in which

the pulmonary artery severely overrides the VSD.

PATHOPHYSIOLOGY OF AR- THE VENTURI EFFECT

Prolapse may result in part from lack of support of the aortic

sinus of Valsalva and “anulus” by the infundibular septum15,

although because most large perimembranous defects are closely

adjacent to the aortic “anulus” and very few have associated

regurgitation, this cannot be the entire explanation.A structural defect

in the base of the sinus may also play a role when the VSD is small.

Loss of continuity of the aortic media from the aortic “anulus” and the

ventricular septum has also been proposed as a mechanism.

Hemodynamic influences can also aggravate the tendencies

towards AR. Such influences are more marked once AR develops,

resulting in progressive prolapse and distortion. The most widely

accepted mechanism of cusp prolapse is the Venturi effect17. It

describes the changes in velocity and pressure that occur when fluid

passes through conduits of varying diameter. When the conduit caliber

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decreases, fluid velocity increases and pressure decreases. Thus, when

there is restriction to flow through a VSD and an aortic valve cusp is

adjacent to the defect, it is vulnerable to being drawn into the high-

velocity, low-pressure jet as blood shunts from the left to right

through the restrictive VSD. This displaces the unsupported “anulus”

outward and downward into the RV.

During late systole, the aortic cusp prolapses into the VSD and

is thus acted upon by direct pressure from the cavity of the LV,

displacing both “anulus” and cusp into the RV. During diastole, the

high pressure in the aortic root distends the dilated sinus, with

further displacement of the aortic “anulus” toward the RV. As a result

the sinus dilates, the distending force becomes greater from increase

in wall tension.

Evidences supporting the Venturi effect as the predominant

cause of aortic cusp prolapse and subsequent AR include:

The Venturi effect requires a restrictive VSD. Previous studies

in the era of near-routine cardiac catheterization indicate that

most of the defects are restrictive with near normal pulmonary

pressure.

A minimum shunt with a Qp/Qs ratio of 1.4 is required to

induce sufficient cusp distorsion and aortic valve prolapse

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Aortic regurgitation complicating VSD is an acquired lesion. AR

or prolapse with VSD is not present at birth and is diagnosed

after infancy

The nadir of the affected leaflet would be expected to lie within

the stream of the shunt. This is consistent with observations

that overriding of the involved cusp is commonly noted. The

override of the aortic cusp in perimembranous VSDs has been

identified as a risk factor for developing cusp prolapse and

subsequent AR.

ANATOMICAL CLASSIFICATION OF VSD - AR SYNDROME

Kawashima et al, proposed the following classification for aortic

incompetence (AI) complicating VSD in 197318. The classification was

according to the location of the VSD, the anatomic type of the right

ventricular outflow tract, and the severity of the aortic cusp herniation

Type I: Supracristal VSD and AI (60%)

o Type IA: Without aortic cusp herniation

o Type IB: Aortic cusp herniation and conal muscular

rim beneath the pulmonic valve

o Type IC: Aortic cusp herniation without a conal

muscular rim beneath the pulmonic valve.

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Type II: Infracristal VSD and AI (20%)

o Type II A: Aortic cusp herniation and conal muscular

rim beneath the pulmonic valve

o Type II B: Aortic cusp herniation without a conal

muscular rim beneath the pulmonic valve

Type III: Infracristal VSD and AI with infundibular pulmonic

stenosis (PS) (14%)

Type IV: Supracristal VSD and AI with infundibular

pulmonic stenosis PS (6%)

In type IA defect, the cause of AI was not related to the existence

of the VSD. A bicuspid aortic valve or abnormal shortening of a

coronary cusp may be the etiology.

In type IB, there was a thin rim of conal musculature beneath

the pulmonic valve when seen from the right side through right

ventriculotomy. Herniation of the aortic cusp is usually mild or

moderate in this type. No significant pressure gradient was

detected across the right ventricular outflow. The herniated

cusp appeared to be the right coronary cusp (RCC) in most

cases.

In type IC, there is no conal muscular rim beneath the pulmonic

valve and therefore, the aortic and pulmonic valves were in

direct apposition. The RCC herniates into the defect and the

degree of cusp herniation is moderate or severe. A systolic

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pressure gradient of 25 mmHg or more may be present at the

right ventricular outflow tract due to the obstruction resulting

from herniation of the aortic cusp. Presence of deformed cusps

may be associated with this type of defects.

The AI in type IIA is not directly related to the existence of a

VSD as it is in type IA.

CLINICAL EXAMINATION

Alexander S Nadas, M.D., Otto G.et al19 had proposed that a

patient with aortic regurgitation complicating VSD can have the

following auscultatory findings

A systolic murmur, usually grade IV (scale of VI) or louder heard

at the left sternal border. The maximal intensity will be at the

lower left sternal border, where assumes the quality of a

murmur of ventricular septal defect.

In many instances this systolic murmur may be transmitted

very well to the pulmonary area, where it assumes a stenotic

character.

A protodiastolic murmur of aortic regurgitation will be present,

by definition, in every case, although in many instances and

this would not be heard until some years after the discovery of

the systolic murmur.

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In contrast to the loud systolic murmur, the aortic blow was

usually of grade-III intensity, or less, and seldom lasted

throughout diastole.

A continuous murmur with maximal intensity just before the

second heart sound as heard in patients with a patent ductus

arteriosus may be observed.

The systolic murmur was loudest in mid-systole, decreased

toward the second sound, and was then followed with increased

intensity by the protodiastolic murmur. Thus, there appeared to

be a hiatus between systolic and protodiastolic murmur,

resulting in a to-and-fro character.

A mid-diastolic rumble, resembling an Austin Flint murmur,

probably caused by increased mitral valve flow, will be heard in

some patients.

The heart sounds will be difficult to judge because of the

murmurs accompany most of the cardiac cycle.

An ejection click may be heard at the apex.

A systolic thrill may be present at the mid lower left sternal

border.

The cardiac impulse is usually hyperkinetic and involved

principally, but not exclusively, the left ventricle.

Left chest prominence was noted frequently, particularly in the

older children with marked aortic regurgitation. At the time of

catheterization, when the auscultatory evidence of aortic

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regurgitation was usually clear-cut, a wide pulse pressure was a

common finding.

Hounding carotid pulses with a low dicrotic notch and a pistol-

shot sound may heard over the femoral artery in patients with

significant aortic run off.

RADIOLOGICAL FINDINGS

shows cardiac enlargement. While the left ventricle is almost

uniformly dilated, additional right ventricular enlargement may be

present. The pulmonary vessels are within normal limits or only

mildly engorged. This absence of marked pulmonary vascular

engorgement was helpful in differentiating ventricular septal defect

with aortic regurgitation from patent ductus arteriosus.

ELECTROCARDIOGRAM FINDINGS

The chest leads of the electrocardiogram shows left ventricular

preponderance with left ventricular hypertrophy. Left ventricular

hypertrophy with ST and T-wave changes may be present.

Biventricular hypertrophy may be an associated finding in some

patients. The, frontal plane mean QRS axis, in the majority, is

between +30 and +90°. In an occasional patient, incomplete right

bundle-branch block or first degree atrioventricular block may be

observed.

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ECHOCARDIOGRAPHIC EVALUVATION OF AORTIC

REGURGITATION20

A systemic evaluation of aortic regurgitation includes the following

steps:

Confirm diagnosis

Quantify the degree of AR

Define mechanism of AR

Define etiology of AR

Evaluate LV response to p/v overload

Identify associated abnormalities

ESTABLISHING THE DIAGNOSIS

The prolapsed aortic cusp is almost always the right coronary

cusp (RCC) and prolapse is diagnosed when there was a distinct

projection of any part of the RCC from the natural curve of the sinus

of Valsalva. In the long axis view of the aortic valve, we measured the

length of the prolapse of the RCC into the VSD at end-systole.

QUANTIFYING THE DEGREE OF AR

Severity of AR18 was graded according to the level to which the narrow

jet reached as follows:

GRADE 1(trivial): Jet reaches just beneath the aortic valve

GRADE 2(slight): Jet is confined to the left ventricular outflow tract

GRADE 3 (moderate): Jet reaches the mid portion of the left ventricle

GRADE 4(severe): Jet reaches the left ventricular apex.

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MECHANISM OF AORTIC REGURGITATION

El khoury et al20 had proposed a classification, based on the

mechanism of aortic regurgitation in 2000. According to them the

functional aortic root was divided into two components, namely

ventriculo aortic junction (VAJ) and the sinotubular junction (STJ).

TYPE I: Normal valve cusps mobility

TYPE IA: Dilation of the sinotubular junction and ascending

aorta

TYPE IB: Dilation of aortic sinuses, sinotubular junction and

ascending aorta

TYPE IC: Dilation of ventriculoaortic junction, aortic

sinuses, sinotubular junction and ascending aorta

TYPE ID: cusp perforation

TYPE II: Increased valve movement

TYPE III: Restrictive valve movement

The type of aortic regurgitation can be made out based on the

nature of the regurgitant jet

Central jet: TYPE IA, TYPE IB, TYPE IC

Paracentral jet: TYPE ID

Eccentric jet:

Away from the prolapsing cusp – TYPE II

Towards the more restricted cusp –TYPE II

The reference value for aortic root dilation on TEE long axis-view are:

Annulus (≥1.6 cm/m²)

Sinus (≥2.1 cm/m²)

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ST junction (≥1.9 cm/m²)

Predictors of a durable repair according to Kindermann et al20:

Absence of more than mild residual AR

Effective height ≥ 9 mm

Coaptation length ≥ 4 mm

SEVERITY OF CUSP PROLAPSE21

RCC deformity index (RCCD) is obtained by dividing the

prolapse length by the ring diameter of the aortic valve. On the short-

axis view of the aortic valve, we measured the length between each

commissure at end-diastole just before valve opening.

RCC imbalance index (R/L) is calculated as the length

between the commissure of the right and left coronary cusps and that

of the right and non-coronary cusps divided by the length between the

commissure of the right and left coronary cusps and that of the left

and non-coronary cusps

SURGICAL TECHNIQUES FOR AORTIC VALVE REPAIR

TRUSLER PROCEDURE22

One or more leaflets can be repaired by the Trusler method of

plication. This procedure is carried out at the commissure adjacent to

the prolapsed cusp (usually the right or noncoronary cusp). A 5-0 or

6-0 polypropylene suture is placed through the fibrous lacunae at the

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midpoint of each cusp. Cusps can then be assessed for elongation and

attenuation. Cusp plication is performed at the elongated free edges of

the aortic valve cusps. A 5-0 or 6-0 PTFE suture is woven between the

right and noncoronary cusps to adjust the excessive length of the

prolapsed cusp to the adjacent aortic wall. Repair may be reinforced

by pledgets (pericardial or felt) or a small cap of polyester secured as a

pledget over both affected cusps adjacent to the commissure. Hisatomi

and colleagues proposed pledget stitch aortoplasty to make the aortic

cusps protrude for even greater aortic cusp coaptation29. The stitch at

the center of the valve is removed after VSD repair. Approach to the

VSD is appropriate to the location of the defect and may be transaortic

if the defect is accessible. Any infundibular stenosis is relieved by

excising trabecular muscle bands between the infundibular septum

and free wall of the RV and, when necessary, by mobilizing and

excising parietal and septal extensions of the infundibular septum

and portions of the moderator bands.

An entirely different technique has been successfully used by

Carpentier, Chauvaud and colleagues. Its basic feature is triangular

excision and reconstruction of the prolapsing cusp. Combined with

this is an anuloplasty of the left ventriculo aortic junction. They also

recommend that the VSD be repaired through the aortic root, using a

glutaraldehyde treated pericardial patch.

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2. YACOUB PROCEDURE23

Yacoub and colleagues have proposed another alternative repair

that addresses the basic morphologic defect more completely. All

anatomic components of defect are corrected by a simple transaortic

repair. A transverse aortotomy is made. Extent of dilatation of the

right coronary sinus and exact definition of the thin area of sinus

resulting from discontinuity between aortic valve “anulus” and media

of aorta are accurately defined. A series of pledget-reinforced mattress

sutures are placed through the crest of the ventricular septum slightly

on the RV side to avoid injuring the conduction system. Sutures are

passed through the “anulus” of the aortic valve and then used to the

plicate the thin portion of the sinus of Valsalva and continued until

strong aortic tissue supported by aortic media is reached. Tying of

sutures results in closing the VSD, elevating the right coronary

anulus and cusp, and reducing the size of the right coronary sinus

and RV outflow tract bulge.

Repair of the defect is possible in all cases regardless of VSD

size, which is usually slitlike, because there are always redundant

tissues in the septum and aortic sinus in the vertical plane. Plication

of redundant tissues toward the media of the aortic sinus elevates the

aortic valve cusp and anulus, displacing them centrally towards the

aortic lumen. This results in increased aortic valve coaptation and

restored aortic valve competence. This operation can be applied to

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patients with doubly committed subarterial VSDs with AR and thus

may apply to the Asian population, in whom these types of defects are

common. If the valve requires replacement, the RV (or pulmonary

trunk) should be opened before valve insertion, the VSD repaired, the

RV closed, and then the aortic valve replaced. This sequence is

advised because occasionally it may be necessary to place sutures

from the prosthetic valve ring across the upper margin of the VSD

patch where it extends between the base of the right and noncoronary

cusps (in the region normally occupied by the membranous septum).

Sutures in this area should be securely buttressed with pledgets.

Although a freehand allograft has been used successfully for valve

replacement under such circumstances, degree of distortion of the

aortic sinuses often makes accurate placement difficult.

Allograft aortic root replacement or aortic valve replacement

with a pulmonary autograft (Ross procedure) is probably a better

choice. When there is also a true thin-walled aneurysm of the sinus of

Valsalva at the base of the right aortic cusp and a VSD, repair is more

difficult because the sinus must also be repaired. If the aortic valve

requires excision and replacement, excision should include the base of

the cusp and the thinned area of the sinus wall, which becomes

continuous with the VSD and is incorporated into its closure. Again,

under such circumstances, the prosthetic valve suture line will cross

the polyester patch. Aortic root replacement may be a simpler

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solution. When the valve is suitable for plication, the base of the cusp

is preserved and sutured back to the patch.

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STUDY METHODOLOGY

The study was a hospital-based retrospective non-randomised

observational study involving human subjects. It was done in 50

patients who have undergone aortic valve repair for aortic

regurgitation complicating ventricular septal defect in the congenital

heart disease wing of the Department of Cardiovascular and thoracic

Surgery between January 2010 and December 2014 after obtaining

clearance from the Institute Research Committee and Institute Ethics

Committee.

INCLUSION CRITERIA

All patients who have undergone aortic valve repair for aortic

regurgitation complicating ventricular septal defect during the study

period were included in the study.

EXCLUSION CRITERIA

Patient undergoing Aortic valve repair for aetiology other than

aortic regurgitation complicating Ventricular septal defect

Patients who had lost to follow up during the follow up period.

DEFINITION

Disease recurrence : appearance of AR of Grade 2 or more

during any time in the follow up period.

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Treatment failure: Need for valve replacement during any time

in the study period.

SAMPLE SIZE CALCULATION

The minimum sample size required will be 30.The exact sample

size was calculated with an expected power of 80% and to detect a

small difference with an effect size of 0.50 and alpha error of 0.025.

The sample size required was estimated to be 38. The study was

begun to analyse at least a minimum of 38 patients.

METHODOLOGY

The list of patients who have undergone aortic valve repair for

AR complicating VSD was obtained from the electronic medical

records using relevant terminologies in the preoperative,

postoperative diagnosis and the procedure executed.

The list obtained was screened and the patients falling within

the inclusion criteria were included in the study.

The medical records were reviewed for demographic data and for

clinical data including the pre op echo findings, intraoperative

findings, operation record, anaesthesia record, intraoperative

transoesophageal ECHO findings and the follow up details. The

data collected were recorded in the observation chart

Demographic data on the age of the patient a surgery, sex,

height (in meter), weight (in kilogram) and body surface area

were collected.

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Preoperative transthoracic echo was analysed for the following:

Anatomic type of VSD

Anatomy of the aortic valve

Severity of AR

Mechanism of AR

Cusps‟ prolapsing

Degree of cusp deformity

Size of the aortic annulus

Operation record was analysed for the following:

Intraoperative findings on the anatomy of aortic

valve (bicuspid/tricuspid/others), the type of VSD,

Cusps prolapsingMechanism of AR

Nature of cusps

Type of aortic repair performed

Annulus dimensions and type of annuloplasty

Route(s) of VSD approach

Additional procedures performed (if any)

Intraoperative transoesophageal echo findings were

analysed for the following:

Anatomic type of VSD

Anatomy of the aortic valve

Severity of AR

Mechanism of AR

24

Cusps‟ prolapsing

Degree of cusp deformity

Size of the aortic annulus

Residual VSD after procedure

Leaflet coaptation height

Degree of regurgitation

Immediate postoperative transthoracic echo was analysed

for the

Presence of AR

Severity of AR

Mechanism of AR

The patients follow up records at the first month, sixth

month and at the end of first year. It was analysed for the

transthoracic echo findings of

Presence of AR

Severity of AR

Mechanism of AR

METHOD OF ANALYSIS

Statistical analysis was performed using SPSS version 22 for

Windows.

25

All categorical data related to patients characteristics, clinical,

laboratory and surgical parameters were recorded and

presented as frequencies and percentages.

Continuous variables like age, height, weight, BSA were

presented as mean with standard deviation and compared using

Independent Student t test

All the categorical data between the various groups were

compared using Chi-Square or Fischer‟s exact test.

The trend of aortic regurgitation from discharge till the final

view was represented as grades. The mean of ranks during each

review was computed and compared using Friedman test.

All statistical analysis was carried out for two tailed significance

and a „p‟ value ≤ 0.05 was considered statistically significant.

26

RESULTS

I.0 IMPACT OF THE DEMOGRAPHIC PARAMETERS ON THE

DISEASE RECURRENCE

1.1 SEX

Table 2.1 shows, 9(75%) male patients and 3(25%) female

patients had disease recurrence compared to 24 (63.2%) male patients

and 14 (36.8%) female patients had no recurrence during follow up.

When the recurrence was compared between boys and girls using Chi-

squared test, the difference was not statistically significant.

The²value was 0.570 and the p value was 0.450.

1.2 AGE

Table 2.2 shows, the mean age of the patients at surgery, who

had disease recurrence was 7.9 ± 5.3 years and that of who did not

have recurrence was 9.4 ±5.8 years. When the recurrence was

compared between the two groups using student t-test, the difference

was not statistically significant. The t value was 0.770 and the p value

was 0.445.

1.3 BODY WEIGHT

Table 2.2 shows, the mean weight of the patients at surgery,

who had disease recurrence was 20.5 ± 8.8 kg and that of who did not

have recurrence was 25.2 ± 13.8 kg. When the recurrence was


was not statistically significant. The t value was 1.099 the p value

27

was 0.277.

1.4 HEIGHT OF THE PATIENT

Table 2.2 shows, the mean height of the patients at surgery,

who had disease recurrence was 1.0 ± 0.1 m and that of who did not

have recurrence was 1.0 ± 0.3 kg. When the recurrence was compared

between the two groups using student t-test, the difference was not

statistically significant. The t value was 0.968the p value was 0.338.

1.5 BODY SURFACE AREA

Table 2.2 shows, the mean body surface area of the patients at

surgery, who had disease recurrence was 0.7 ± 0.2 m² and that of who

did not have recurrence was 0.8 ± 0.3kg. When the recurrence was


was not statistically significant. The t value was 0.968 the p value was

0.338.

2.0 IMPACT OF ANATOMICAL FACTORS ON THE DISEASE

RECURRENCE

2.1 VALVE ANATOMY

Table 2.3 shows, 10 (83.3%) of patients with tricuspid valve and

2 (16.7%) with bicuspid valve had disease recurrence. When the

recurrence was compared between the two groups, using Chi- squared

test, the difference was not statistically significant. The ² value was

0.326 and the p value was 0.568.

28

2.2 ANATOMICAL LOCATION OF VSD

From table 2.4, it is known that, of the 12 patients who had

disease recurrence, 8 (66.7%) had subaortic type of VSD and 4(33.7%)

patients had doubly committed VSD. None of the patients with outlet

muscular VSD had disease recurrence.When the recurrence was

compared between the various types of VSD, using Chi- squared test,

the difference was not statistically significant. The ² value was 2.210

and the p value was 0.331.

2.3 SURGICAL APROACH TO VSD CLOSURE

We infer from table 2.5 that, 8 (66.7%) of the 12 patients with

recurrence had their VSD closed from the right atrium (trans-RA).

Whereas, 4 (33.3%) of the 12 patients with recurrence had their VSD

closed from the pulmonary artery (trans-PA). Five patients (13.2%)

whose VSD were closed from the aorta and 2 (5.3%) patients whose

VSD was closed via right ventricle did not have any disease

recurrence.When the recurrence was compared between the various

types of surgical approach, using Chi- squared test, the difference was

not statistically significant. The ² value was 3.150 and the p value

was 0.359.

29

3.0 IMPACT OF THE PREOPERATIVE GRADE OF AORTIC

REGURGITATION ON THE DISEASE RECURRENCE

Table 2.6 shows, a total of 17 (345) patients had moderate grade

of aortic regurgitation and 33 (66%) had severe grade of aortic

regurgitation prior to surgery. Three (25%) patients with moderate

grade of aortic regurgitation and9 (75%) patients with severe grade of

aortic regurgitation had disease recurrence. When the recurrence was

compared between preoperative grades of aortic regurgitation, using

Chi- squared test, the difference was not statistically significant. The

² value was 0.570 and the p value was 0.45

4.0 EFFECT OF THE SURGICAL TECHNIQUES ONRECURRENCE

RATES

We draw the following inferences from the data in Table 2.7.

4.1 Central plication

A total of 11 patients had undergone this procedure accounting

for 22 % of the repairs in this study.

Of the 11 patients who underwent the procedure, 5 patients had

disease recurrence. Hence, the recurrence rate was 45.45% for

this technique.

Considering all the procedures, 5 out of the 12 cases who had

recurrence belong to this group, amounting to 41.7% of disease

recurrence in this study.

30

4.2 Triangular resection



Of the 13 patients who underwent the procedure, 3 patients had

disease recurrence. Hence, the recurrence rate was 23.08% for

this technique.

Considering all the procedures, 3 out of the 12 cases who had

recurrence belong to this group, amounting to 25 % of disease


4.3 Trusler repair


for 40% of the repairs in this study.

Of the 20 patients who underwent the procedure, 4 had disease

recurrence. Hence, the recurrence rate was 20 % for this

technique.

Considering all the procedures,4 out of the 12 cases who had

recurrence belong to this group, amounting to 33.3% of disease


4.4 Pericardial patch repair



31

Of the 2 patients who underwent the procedure, none had

disease recurrence. Hence, the recurrence rate for this

technique was nil.

Considering all the procedures, none of the 12 cases who had

recurrence belong to this group, amounting to zero percent

disease recurrence in this study.

4.5 Yacoub repair



Of the 4 patients who underwent the procedure, none had

disease recurrence. Hence, the recurrence rate for this

technique was nil.

Considering all the procedures, none of the 12 cases who had

recurrence belong to this group, amounting to zero percent

disease recurrence in this study.

The disease recurrence was compared between the various

techniques of aortic valve repair, using Chi- squared test, the

difference was not statistically significant. The ²alue was 4.468 and

the p value was 0.346

32

5.0 EFFECT OF ANNULOPLASTY ON DISEASE RECURRENCE

From table 2.8, it is known that, 14 (28%) of the patients had

undergone annuloplasty along with the aortic valve repair in this

study. Six out this 14 patients had disease recurrence. Whereas, six

out of the 36 patients who had not undergone annuloplasty also had

disease recurrence.When the recurrence was compared between the

two groups, using Chi- squared test, the difference was not

statistically significant. The ² value was 3.791 and the p value was

0.052.

6.0 THE EFFECT OF ADDITIONAL SURGICAL PROCEDURES ON

THE DISEASE RECURRENCE

From table 2.9, it is known that, a total of 14 (28%) patients had

undergone additional surgical procedures.

ICR was done in 11(22%) of the patients of which 2 (4%) of the

recurrence. This accounted for 16.7% of recurrence in the

study.

SOV repair was done in 1 (2%) patient who did not have

recurrence.

Mitral valve repair was done in 1(2%) who had recurrence. This

accounted for 4 % of recurrence in the study.

When the recurrence was compared between the groups, using

Chi- squared test, the difference was not statistically significant. The

² value was 8.059 and the p value was 0.153.

33

7.0 TREND OF THE GRADES OF AORTIC REGURGITATION OVER

THE FOLLOW UP PERIOD

From table 3.1 we infer,

At the time of discharge, 11 (22%) had no aortic regurgitation,

33 (66%) had mild grade of aortic regurgitation, 6 (12%) had

moderate grade of regurgitation and none had severe grade of

aortic regurgitation. The mean rank (grade of aortic

regurgitation) at the time of discharge was 2.98.

At the time 1st month follow up, 9 (18%) had no aortic

regurgitation, 35 (70%) had mild grade of aortic regurgitation, 6

(12%) had moderate grade of regurgitation and none had severe

grade of aortic regurgitation. The mean rank (grade of aortic


At the time of 6th month follow up, 8 (16%) had no aortic


(6%) had moderate grade of regurgitation and none had severe



At the time of 1st year follow up, 9 (18%) had no aortic


(14%) had moderate grade of regurgitation and 1 (2%) had

severe grade of aortic regurgitation. The mean rank (grade of

aortic regurgitation) at the time of discharge was 3.07.

34

At the time of 2nd year follow up, 9(18%) had no aortic


(12%) had moderate grade of regurgitation and 1(2%) had severe



The mean of ranks during each of the visit was compared using

Friedman test to assess any significant change in the trend of the

grade of AR during the follow up period. The p value was 0.749 and

the difference was not statistically significant.

35

Table 1.1

Demographic Parameters of the study population

1. Sample size 50 (100%)

2. Gender

Male 33(66%)

Female 17(34%)

3. Age (in years) 9.02±5.67

4. Weight (in kilograms) 24.04±12.82

5. Body Surface Area (metre²) 0.82±0.30

6.Valve anatomy

Tricuspid 44(88%)

Bicuspid 06(12%)

7. Type of VSD

SAVSD 35(70%)

DCVSD 11(22%)

OUTLET MUSCULAR VSD 04(08%)

TABLE 2.1

Comparison of the sex with disease recurrence

Sex

Recurrence

Total

χ2 p Yes No

N % N % N %

Male 9 75.0 24 63.2 33 66.0

0.570 0.450 Female 3 25.0 14 36.8 17 34.0

Total 12 100.0 38 100.0 50 100.0

36

TABLE 2.2

Comparison of demographic parameters with disease

recurrence

Recurrence t

p value Yes No

Mean SD Mean SD

Age ( in years) 7.9 5.3 9.4 5.8 -0.770 0.445

Weight ( in Kg) 20.5 8.8 25.2 13.8 -1.099 0.277

Height (in meter) 1.0 0.1 1.0 0.3 -0.968 0.338

BSA (in m2 ) 0.7 0.2 0.8 0.3 -1.093 0.280

TABLE 2.3

Comparison of valve anatomy with disease recurrence

VALVE ANATOMY

Recurrence Total

χ2 P

value Yes No

N % N % N %

TAV 10 83.3 34 89.5 44 88.0

0.326 0.568 BAV 2 16.7 4 10.5 6 12.0

Total 12 100.0 38 100.0 50 100.0

TABLE 2.4

Comparison of the type of VSD with disease recurrence

TYPE OF VSD

Recurrence Total

χ2 P Yes No

N % N % N %

SAVSD 8 66.7 27 71.1 35 70.0

2.210 0.331

DCVSD 4 33.3 7 18.4 11 22.0

Outlet

Muscular VSD 0 0.0 4 10.5 4 8.0

Total 12 100.0 38 100.0 50 100.0

37

TABLE 2.5

Comparison of the surgical approach with disease

recurrence

SURGICAL

APROACH TO

VSD

Recurrence

Total

χ2 P Yes No

N % N % N %

Trans Aortic 0 0.0 5 13.2 5 10.0

3.150 0.369

Trans RA 8 66.7 24 63.2 32 64.0

Trans PA 4 33.3 7 18.4 11 22.0

Trans RV 0 0.0 2 5.3 2 4.0

Total 12 100.0 38 100.0 50 100.0

TABLE 2.6

Comparison of preoperative severity of regurgitation with

disease recurrence

PREOP AR

GRADE

Recurrence

Total

χ2 p Yes No

N % N % N %

Moderate 3 25.0 14 36.8 17 34.0

0.570 0.450 Severe 9 75.0 24 63.2 33 66.0

Total 12 100.0 38 100.0 50 100.0

38

TABLE 2.7

Comparison of the surgical technique with disease

recurrence

SURGICAL TECHNIQUE

Recurrence Total

χ2 P Yes No

N % N % N %

1. Central

plication 5 41.7 6 16.2 12 24

4.468 0.346

2. Triangular

resection 3 25.0 10 27.0 13 26

3. Trusler

repair 4 33.3 16 43.2 20 40

4. Pericardial

patch repair 0 0.0 2 5.4 2 4

5. Yacoub

repair 0 0.0 3 8.1 3 6

Total 12 100.0 37 100.0 50 100.0

39

TABLE 2.8

Comparison of annuloplasty with disease recurrence

Recurrence

Total χ2 P ANNULOPLASTY Yes No

N % N % N %

Not done 6 50.0 30 78.9 36 72.0

3.791 0.052 Done 6 50.0 8 21.1 14 28.0

Total 12 100.0 38 100.0 50 100.0

TABLE 2.9

Comparison of additional procedures done with disease

recurrence

ADDITIONAL

PROCEDURE

Recurrence

Total

χ2 P Yes No

N % N % N %

No

additional

procedure

9 75.0 27 71.1 36 72.0

8.059 0.153

ICR 2 16.7 9 23.7 11 22.0

SOV repair 0 0.0 1 2.6 1 2.0

MV repair 1 8.3 0 0.0 1 2.0

Total 12 100.0 38 100.0 50 100

40

TABLE 3.1

TREND OF AORTIC REGURGITATION ON FOLLOW UP

Grade of Aortic

regurgitation

At discharge

First follow

up

Second

follow up

Third

follow up

Fourth

follow up

N % N % N % N % N %

No 11 22.0 9 18.0 8 16.0 9 18.0 9 18.0

Mild 33 66.0 35 70.0 39 78.0 33 66.0 34 68.0

Moderate 6 12.0 6 12.0 3 6.0 7 14.0 6 12.0

Severe 0 0

0 0 0 0 1 2.0 1 2.0

Total 50 100.0 50 100.0 50 100.0 50 100.0 50 100.0

'

Ranks Mean Rank

AR AT DISCHARGE 2.98

POST OP AR AT FOLLOW UP 1 3.04




Friedman test p=0.749

41

TABLE 3.2

Comparison of various surgical techniques and types of VSD on

disease recurrence

SURGICAL

TECHNIQUE

TYPE OF VSD Total

SAVSD DCVSD Outlet VSD

N % N % N % N %

Central Plication 9 75.0 1 8.3 2 16.7 12 100.0

Triangular resection 10 76.9 2 15.4 1 7.7 13 100.0

Trussler repair 14 70.0 6 30.0 0 0.0 20 100.0

Pericardial Patch 1 50.0 1 50.0 0 0.0 2 100.0

Yacoub repair 1 33.3 1 33.3 1 33.3 3 100.0

Total 35 70.0 11 22.0 4 8.0 50 100.0

χ2 =8.854 p=0.354

TABLE 3.3

Comparison of various surgical techniques and the surgical

approach to the VSD closure on disease recurrence

SURGICAL

TECHNIQUE

ROUTE OF VSD REPAIR

Trans Aortic

Trans RA

Trans PA

Trans RV

N % N % N % N % N %

Central Plication 0 0.0 11 91.7 1 8.3 0 0.0 12 100.0

Triangular resection 1 7.7 9 69.2 2 15.4 1 7.7 13 100.0

Trussler 2 10.0 11 55.0 6 30.0 1 5.0 20 100.0

Pericardial Patch 1 50.0 0 0.0 1 50.0 0 0.0 2 100.0

Yacoub 1 33.3 1 33.3 1 33.3 0 0.0 3 100.0

Total 5 10.0 32 64.0 11 22.0 2 4.0 50 100.0

χ2 =13.487 p=0.335

42

FIGURE 1 TREND OF GRADES OF AORTIC RGURGITATION

DURING FOLLOW UP (IN PERCENTAGE)

2218 16 18 18

6670

7866 68

12 126

14 12

0 0 0 2 2

0%

10%

20%

30%

40%

50%

60%

70%

80%

90%

100%

At discharge First follow up Second follow up Third follow up Fourth follow up

No Mild Moderate Severe

43

DISCUSSION

The earliest description of aortic cusp prolapse and

regurgitation complicating ventricular septal defects occurring in the

outlet septal region was in 1921 by Laubry and Pezzi24. Garamella and

Starr et al in 1960 first descibed the surgical technique for this

relatively rare congenital heart disease25. Since then, the surgical

techniques had continued to evolve. Yacoub et al in 199723 was the

most recent to describe such a technique of valve repair. With the

surgical techniques being described more recently, the consensus

regarding the patient selection for each technique has been a grey

area. Similarly, though the natural course of disease has been

elucidated in detail by many authors, the factors predicting surgical

success or failure have not been described, particularly in the Asian

population. Thus, this study aimed at identifying the factors that

predict a durable valve in this rare subset of patients.

Hung-Chi Lue, Atsuyoshi Takao et al had shown there is a

tendency for increase in male preponderance in patients with aortic

regurgitation complicating VSD.26 Similar to their observation, male

patients in this study accounted for two third (66%) of the population

with girls forming the rest. Male population also accounted for 75 % of

the recurrence. But when the difference between the two groups was

analysed the difference was not statistically significant (p = 0.450).

44

Komai et al had concluded from their study that “the most

prudent strategy for the patient with VSD and aortic cusp prolapse is

early closure of the VSD before the onset of AR”17. But in our study all

patients had developed aortic regurgitation prior to the procedure. The

age of patients in our study ranged from 1 year to 27 years. The mean

age at surgery being 8.65±5.6 yrs. This is slightly higher to what

Komai et al had speculated from their study which had a median age

of 4.6 years. Hiba Gaafar Salih, Sameh R. Ismail et al in a recent

study, similar to this study employing patients with overt cusp

prolapse prior to surgery also had the mean age of patients 3.8 ± 2.6

years.

The relatively higher age of our patients was because of a

multitude of socioeconomic factors rather than scientific factors. The

poor literacy level and low socioeconomic status of the population our

institution caters to were the major hurdles in getting the patients at

an early age for surgery. This accounts for the considerable level of

delay in diagnosis and referral, withstanding the fact of the long

waiting list of patients for surgery in our institution which is a tertiary

care level hospital.

The comparison of mean age of patients who had disease

recurrence and who did not in this study from table 1.1, had shown

no statistically significant difference between the two groups. We thus

45

infer, aortic valve repair can be safely attempted even in the patients

presenting late in their natural course, with no extra added risk of

recurrence with their younger counterparts. Nevertheless, it is not a

prudent option to delay the surgery.

This study had 6 (12%) patients with bicuspid aortic valve in

contrast to the 1-2% in the general population27. The mechanism of

aortic regurgitation in this group of patients was of El khoury type II

caused by the cusp prolapse unrelated to the occurrence of the VSD18.

All the 6 patients had a perimembranous type of VSD, 5 of which were

repaired by the transatrial approach while in the sixth case, it was

repaired via the transaortic route. Two among these 6 patients

developed disease recurrence by definition, accounting for 16.7 % of

the total recurrences in the study. In both the patients, the severity of

regurgitation declined from grade 2 to grade 1 by the end of first year

and remained the same till the second year follow up. In contrast,

patients with tricuspid aortic valve formed 88% (44/50) of the total

population with 10 recurrences accounting for 83.3% of recurrence.

The comparison of recurrences between the two groups with

anatomically different valve leaflets had shown no statistically

significant difference (p= 0.568). The increased risk of recurrence after

valve repair in patients with bicuspid aortic valve proposed by Okita et

al28 in their study was not established in our study.

46

Most of the patients in this study had perimembranous VSD

accounting for 70 % of population, followed by doubly committed VSD

accounting for 22% and the rest 8% was by outlet muscular VSD. This

was in contrast to the results of previous study by Hisatomi K and

Kosuga K et al29, which had documented increased incidence of

doubly committed VSD in the Asian population.

Okita et al28 had shown increased risk of residual AR with

perimembranous VSD rather than outlet VSDs. When the recurrence

of AR was compared between the various types of VSD in this study,

from table 2.4, the difference was not statistically significant (p=

0.331).

With the echocardiographic evaluation of preoperative severity

of aortic regurgitation, 34% of the study population had moderate

(grade 2) aortic regurgitation and 66% of the population had severe

(grade 3 or more) aortic regurgitation. The incidence of recurrence

was 25% and 75 % for the moderate and the severe grades of AR

respectively. The difference between the two groups on comparison

showed no statistical significance (p= 0.450). The severity of

preoperative aortic regurgitation thus had not precluded a safe valve

repair in patients with AR complicating VSD.

Trusler repair was the most frequently performed procedure in

47

this study with a total of 20 (40 %) cases. Four (20%) out of the 20

patients who underwent this procedure had recurrence. Annuloplasty

was done in 6 of these patients for aortic annulus dilation (≥2 SD).

ICR was done in 3 of these patients for right ventricular outflow

obstruction. Also, from table 3.2 and table 3.3 we know that, the type

of VSD and the route of repair had no bearing on the recurrence. The

recurrence is hence independent of the type of VSD, the surgical

approach to VSD closure, performance of annuloplasty or an

additional procedure. It is probably the growth of the valve per se

accounting for the recurrence of the disease after Trusler repair.

Further, the qualitative analysis of the regurgitant jet by

echocardiogram is essential for elucidating the exact mechanism

involved. But, this analysis was not done in the study.

Similarly, the effect of all the other surgical techniques the

patients had undergone was analysed for the recurrence, including

the Carpenteir technique, Yacoub technique and pericardial patch

repair. Analysis had revealed that there is no difference in terms of

risk of recurrence of one procedure over the other. Subanalysis of the

effect of the various anatomical type of VSD and the surgical approach

to VSD closure on the recurrence of each of the surgical procedure,

had also shown to have no statistically significant difference (table 3.2

and table 3.3).

48

Annuloplasty was done in 14 (28 %) patients with dilated

annulus. This was in addition to addressing the leaflet pathology. Six

out of 14 patients had recurrence, but with no statistically significant

difference on comparison. None of the patients had stenosis during

follow up rather. The addition of subcommissural annuloplasty had

thus found not been associated with an additional risk to patients

undergoing aortic valve repair.

The trend of the grade of aortic regurgitation in the study

population is as shown in table 3.1. The mean of ranks during each

visit was computed and was compared between each visits. But the

difference was not statistically significant. Though there was minor

changes in the number of patients in each of the grades, the mean of

the ranks over the entire study population had not changed

significantly.

Although by the definition in this study,12 patients had

developed recurrence, 11 of those twelve patients had grade 2

(moderate) aortic regurgitation and only 1 patient had severe (grade

3)aortic regurgitation. On analysis of the transthoracic

echocardiographic mechanism of the recurrence, all the recurrence

were intravalvar leaks. All of them were detected on regular follow up

visit and none of the patients were symptomatic for the regurgitation

during the follow up period. Excluding the one patient who required

49

valve replacement in the immediate postoperative period, none of the

other patients either of life in required a replacement or medical

management for their valve leak. The quality of life in all the patients

who had undergone valve repair had definitely improved during follow

up though it was not objectively analysed in the study.

The study was limited by the small sample size and its

retrospective nature though it had analysed a subset of patients with

very low disease incidence. The duration of follow up was also

relatively less to determine the recurrence of the disease considering

the growth potential of the patients in this study who were

predominantly in the pediatric age group. The subjective evidence of

improvement in thequality of life of this population was very evident in

the study. But an objective measurement of the quality of life of this

population would have given the real impact of valve repair in this

rare subset of patients on their clinical as well as socioeconomic

status. Aortic valve repair in patients with aortic regurgitation

complicating ventricular septal defect was thus a safe and durable

procedure considering the short term outcomes. A prospective

randomised trial would be the solution to the questions unanswered

in the study.

50

CONCLUSION

Outcome after aortic valve repair in patients with AR

complicating VSD is not affected by demographic factors like

age, sex, height, weight and the body surface area of the

patients.

Aortic valve repair however, can be safely attempted in patients

presenting with AR late in the course of the disease without

increased risk of recurrence.

Repair of a bicuspid valve associated with VSD can be done

satisfactorily with no added risk of failure of repair.

Anatomical factors like the type of VSD and surgical approach

to VSD closure did not affect the outcome after valve repair.

Additional surgical procedures and annuloplasty during valve

repair have no adverse effect on the outcome of valve repair.

The superiority of one surgical procedure over the other for

aortic valve repair is not evident from our study. A prospective

randomised trial is needed to establish the same.

51

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OBSERVATION CHART

NAME

HOSPITAL NO

AGE AT SURGERY

SEX

HEIGHT

WEIGHT

BSA (DERIVED)

PRE OPERATIVE PARAMETERS

Anatomy of the aortic valve :BAV/ TAV/OTHERS

Mechanism of regurgitation:

Grade of AR at diagnosis :MILD/MODERATE/SEVERE

Aortic annulus dimensions: (in mm)

Type of VSD

INTRAOPERATIVE

Type of procedure executed

Need for additional procedures

POSTOPERATIVE

Grade of AR in the immediate postoperative

period:MILD/MODERATE/SEVERE

Grade of AR at

1st monthly :MILD/MODERATE/SEVERE

Half yearly :MILD/MODERATE/SEVERE

Annual follow up:MILD/MODERATE/SEVERE

REOPERATION

Type of procedure

Time of procedure

S NO NAME SEX AGE WEIGHT HEIGHT BSA TYPE OF VSDROUTE OF VSD REPAIRVALVE ANATOMYSURG TECHANNULOPLASTYADD PROCEDUREPREOP ARAR AT DISCHARGE

FIRST SECOND THIRDFOURTH

1 ABHIMANYU S PILLAI 0 5 15 0.99 0.64 1 2 1 1 1 3 1 1 1 2 2

2 ABHINAV 0 8 30 1.02 0.92 2 3 1 2 0 0 2 2 2 2 2 1

3 ABINAYA 1 6 20 0.95 0.73 1 2 2 2 0 0 3 1 1 1 1 1

4 ADHIN JOSE 0 4 17 0.87 0.64 2 3 1 3 0 0 3 1 1 1 2 2

5 ADITHYA 0 10 25 1.12 0.88 1 2 1 1 1 0 2 0 0 2 2 2

6 AJITHA 1 8 18 0.96 0.69 2 3 1 3 1 0 3 0 0 0 0 0

7 AMAL SHAN 0 9 22 1.2 0.86 1 2 1 1 0 0 3 1 1 1 1 1

8 ANAMIKA 1 3 14 0.85 0.57 1 2 1 2 0 0 3 1 1 1 0 0

9 ANJAH 0 14 40 1.28 1.19 2 3 1 3 0 0 3 1 1 1 1 1

10 ANNA JAMUNA 1 9 28 0.95 0.86 1 2 1 1 1 0 3 1 1 1 1 1

11 ANZIL 0 6 21 0.86 0.71 1 2 1 3 1 0 2 1 1 1 1 1

12 ARJUN P A 0 12 32 1.03 0.96 2 3 1 2 0 0 2 1 1 1 1 1

13 ASWATHY ARUN 1 11 15 0.95 0.63 1 2 2 3 1 3 2 1 1 1 1 1

14 BHAVANA 1 10 24 1.25 0.91 1 2 1 2 0 0 2 1 1 1 1 1

15 EDWIN THOMAS 0 10 25 1.18 0.91 2 3 1 6 0 3 3 1 1 1 1 1

16 FATHIMA FIDA 1 1 9 0.65 0.40 1 2 1 1 0 3 3 0 0 0 0 0

17 GOKUL 0 10 30 1.45 1.10 1 1 1 3 0 0 3 1 1 1 2 2

18 GOUTHAM VIJAY 0 8 24 0.89 0.77 1 2 1 2 0 0 3 0 1 1 3 3

19 IDRIS P Y 0 9 23 0.96 0.78 3 2 1 6 0 5 3 1 1 1 1 1

20 JASMIN SEJI 1 22 40 1.24 1.17 2 3 1 3 0 0 2 0 1 1 2 2

21 JUVAN THOMAS 0 2 11 0.81 0.50 1 2 1 2 0 0 2 0 0 0 0 0

22 JEYA MARY 1 8 18 1.01 0.71 1 2 1 1 1 0 3 2 2 2 2 2

23 LIJO 0 16 40 1.35 1.22 1 2 1 3 1 3 2 1 1 1 1 1

24 MEEVAL THOMAS 1 5 15 0.87 0.60 1 1 1 5 0 0 3 0 0 0 0 0

25 MUHAMMED ALFIN 0 2 10 0.77 0.46 3 1 1 2 0 0 2 0 0 0 0 0

26 NANDANA 1 3 15 0.89 0.61 2 3 1 1 1 6 3 2 2 1 1 1

27 NASEEHA 1 9 28 1.02 0.89 1 2 2 3 0 0 2 1 1 1 1 1

28 NEWJIN 0 7 12 0.85 0.53 3 2 1 1 0 0 2 1 1 1 1 1

29 NEERAJA RAJ 1 3 7 0.79 0.39 1 2 1 3 0 3 2 1 1 1 1 1

30 PRIYADHARSHINI 1 9 17 0.85 0.63 1 2 1 2 1 0 3 1 1 1 1 1

31 RAHUL 0 4 16 0.92 0.64 2 3 1 3 0 0 3 1 1 1 1 1

32 SAHAD 0 17 35 1.15 1.06 1 2 1 1 0 0 3 0 1 1 1 1

33 RIFANA 1 2 14 0.95 0.61 3 2 1 1 1 3 2 1 1 1 1 1

POST OP FOLLOW UP

34 SANTHANA KUMAR 0 20 55 1.6 1.56 1 2 1 2 0 3 2 1 1 1 1 1

35 SHABINA 1 14 45 1.45 1.35 1 2 1 1 0 0 2 1 0 0 0 0

36 SUDHA 1 10 55 1.45 1.49 1 4 1 2 1 3 3 1 1 1 1 1

37 SURESH M 0 27 68 1.71 1.80 1 4 1 3 0 3 3 1 1 1 1 1

38 TINTO MATHEW 0 25 40 1.32 1.21 2 3 1 4 0 4 3 1 1 1 1 1

39 VIGNESH 0 10 23 0.94 0.77 1 2 1 3 0 0 3 1 1 1 1 1

40 VISHNU 0 7 23 0.93 0.77 1 2 1 3 0 0 3 1 1 1 1 2

41 VYSHNAV 0 9 18 0.82 0.64 1 2 1 3 0 0 3 0 0 0 0 0

42 KRISHNA SANGEETHA 0 7 18 0.88 0.66 1 1 1 3 0 0 3 1 1 1 1 1

43 SREESHMA 0 5 15 0.87 0.60 1 2 1 3 0 0 2 1 1 1 1 1

44 THASNI 0 12 24 1.02 0.82 1 2 1 1 0 3 3 2 2 1 1 1

45 SHAMLA SHERIN 0 10 25 0.99 0.83 1 1 2 4 0 0 3 1 1 1 1 1

46 MUHAMMED MIHRAN 0 2 6 0.77 0.36 1 2 1 3 1 0 3 1 1 1 2 2

47 ANJANA 0 6 14 0.8 0.56 1 2 2 3 1 0 3 2 2 1 1 1

48 VINOD 0 10 25 0.95 0.81 2 3 1 3 0 0 3 0 0 0 0 0

49 SELVAKUMAR 0 7 18 1.04 0.72 1 2 2 2 0 0 3 2 2 1 1 1

50 ANITHA 0 8 20 0.95 0.73 1 2 1 2 0 0 3 1 1 1 1 1

1.0274 0.82

12.8237 0.30

CODES USED

1-TAV1-CENTRAL PLICATION0- NOT DONE0- NO ADD PROC

0- MALE 1- SAVSD1- TRANS AORTIC2-BAV2-RCC TRIANGULAR 1-DONE1-ICR

1- FEMALE 2-DCVSD2- TRANS RA3-TRUSSLERSSOV REPAIR1 MILD

3- OUTLET MUSCULAR VSD3- TRANS PA4-PERICARDIAL PATCH3-MV REPAIR2 MODERATE

4-TRANS RVYACOUB 3 SEVERE

0 NO AR

GRADE OF AR

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OUTCOME OF AORTIC VALVE REPAIR FOR AORTIC REGURGITATION COMPLICATING

VENTRICULAR SEPTAL DEFECT-A RETROSPECTIVE OBSERVATIONAL STUDY

INTRODUCTION Aortic regurgitation in patients with ventricular septal defect (VSD)

occurs in those anatomical types that are located close to or in direct contact with the

aortic valve. This subset of patients form a relatively rare group of patients with an

incidence ranging between 5% and 10%.Perimembranous and doubly committed

juxta-arterial defects are the commonly affected types. Rarely, it can occur in outlet

muscular VSD also.

Prolapse of the valvular cusps caused by the venturi effect of the ventricular septal

defect jet, abnormal commissural suspension, lack of appositional forces and lack of

continuity between the aortic media, annulus of aortic valve and the ventricular septum

have been the commonly proposed pathological anatomic mechanisms of regurgitation.

The right coronary cusp (RCC) is the usually involved cusp.

This is followed in line by the non-coronary cusp whereas, the involvement of the left

coronary cusp is very rare. The prolapsing cusps render the valve incompetent and cause

significant damage to the valve cusp and annulus. The prolapsing AV may completely

close the VSD so that the shunt may disappear but leaving behind the regurgitation to

progress.

Surgical closure of the defect with aortic valve repair is indicated for these patients

irrespective of the grade of preoperative aortic regurgitation and it is recommended to

close the defect at the earliest. The technique of valve repair is decided based on the

mechanism(s) of regurgitation Despite increasing interest in pediatric aortic valve repair

is unavoidable in certain circumstances.

With limited options for valve replacement and lack of prospective studies the

superiority of these one prosthesis over the other cannot be ascertained. Metaanalysis

has shown that all the currently available valve substitutes are associated with

suboptimal outcome in children. Hence a durable aortic valve repair is the need of the

hour.

This study aims to evaluate the outcome of patients who underwent aortic valve repair

procedures for aortic regurgitation complicating VSD and to also to assess the

predictors for their outcomes after surgery. METHODOLOGY The study was a

hospital-based retrospective non-randomised observational study involving human

subjects. It was done in 50 patients who have undergone aortic valve repair for aortic

regurgitation complicating ventricular septal defect in the Department of Cardiovascular

and thoracic Surgery between January 2010 and December 2015 after obtaining

clearance from the Institute Research Committee and Institute Ethics Committee.

INCLUSION CRITERIA All patients who have undergone aortic valve repair for aortic

regurgitation complicating ventricular septal defect during the study period were

included in the study. EXCLUSION CRITERIA Patient undergoing Aortic valve repair for

aetiology other than aortic regurgitation complicating Ventricular septal defect Patients

who had lost to follow up SAMPLE SIZE CALCULATION The minimum sample size

required will be 30.The exact sample size was calculated with an expected power of 80%

and to detect a small difference with an effect size of 0.50 and alpha error of 0.025.

The sample size required was estimated to be 38. The study was begun to analyse at

least a minimum of 38 patients. METHODOLOGY The list of patients who have

undergone aortic valve repair for AR complicating VSD was obtained from the electronic

medical records using relevant terminologies in the preoperative, postoperative

diagnosis and the procedure executed.

The list obtained was screened and the patients falling within the inclusion criteria were

included in the study. The medical records were reviewed for demographic data and for

clinical data including the pre op echo findings, intraoperative findings, operation

record, anaesthesia record, intraoperative transoesophageal ECHO findings and the

follow up details.

The data collected were recorded in the observation chart Demographic data on the age

of the patient a surgery, sex, height (in meter), weight (in kilogram) and body surface

area were collected. Preoperative transthoracic echo was analysed for the following:

Anatomic type of VSD Anatomy of the aortic valve Severity of AR Mechanism of AR

Cusps‟ prolapsing Degree of cusp deformity Size of the aortic annulus Operation record

was analysed for the following: Intraoperative findings on the anatomy of aortic valve

(bicuspid/tricuspid/others), the type of VSD, Cusps prolapsing Mechanism of AR Nature

of cusps Type of aortic repair performed Annulus dimensions and type of annuloplasty

Route(s) of VSD approach Additional procedures performed (if any) Intraoperative

transoesophageal echo findings were analysed for the following: Anatomic type of VSD

Anatomy of the aortic valve Severity of AR Mechanism of AR Cusps‟ prolapsing Degree

of cusp deformity Size of the aortic annulus type Residual VSD after procedure Leaflet

coaptation height and Degree of regurgitation Immediate postoperative transthoracic

echo was analysed for the Presence of AR Severity of AR Mechanism of AR The patients

follow up records at the first month, sixth month and at the end of first year.

It was analysed for the transthoracic echo findings of Presence of AR Severity of AR

Mechanism of AR METHOD OF ANALYSIS Statistical analysis was performed using SPSS

version 22 for Windows. All categorical data related to patients characteristics, clinical,

laboratory and surgical parameters were recorded and presented as frequencies and

percentages.

Continuous variables like age, height, weight, BSA were presented as mean with

standard deviation and compared using Independent Student t test All the categorical

data between the various groups were compared using Chi-Square or Fischer‟s exact

test. The trend of aortic regurgitation from discharge till the final view was represented

as grades.

The mean of grades during each review was computed and compared using Wilcoxin

rank sum test. All statistical analysis was carried out for two tailed significance and a „p‟

value = 0.05 was considered statistically significant RESULTS RESULTS IMPACT OF THE

DEMOGRAPHIC PARAMETERS ON THE DISEASE RECURRENCE SEX Table 2.1

shows, 9 (75%) male patients and 3(25%) female patients had disease recurrence

compared to 24 (63.2%) male patients and 14 (36.8%) female patients had no recurrence

during follow up. When the recurrence was compared between boys and girls using Chi-

squared test, the difference was not statistically significant. The ?² value was 0.570 and

the p value was 0.450. AGE Table 2.2 shows, the mean age of the patients at surgery,

who had disease recurrence was 7.9 ± 5.3

years and that of who did not have recurrence was 9.4 ± 5.8 years. When the recurrence

was compared between the two groups using student t-test, the difference was not

statistically significant. The t value was 0.770 and the p value was 0.445. BODY WEIGHT

Table 2.2 shows, the mean weight of the patients at surgery, who had disease recurrence

was 20.5 ± 8.8

kg and that of who did not have recurrence was 25.2 ± 13.8 kg. When the recurrence

was compared between the two groups using student t-test, the difference was not

statistically significant. The t value was 1.099 the p value was 0.277. HEIGHT OF THE

PATIENT Table 2.2 shows, the mean height of the patients at surgery, who had disease

recurrence was 1.0 ± 0.1

m and that of who did not have recurrence was 1.0 ± 0.3 kg. When the recurrence was

compared between the two groups using student t-test, the difference was not

statistically significant. The t value was 0.968 the p value was 0.338. BODY SURFACE

AREA Table 2.2 shows, the mean body surface area of the patients at surgery, who had

disease recurrence was 0.7 ± 0.2

m² and that of who did not have recurrence was 0.8 ± 0.3kg. When the recurrence was

compared between the two groups using student t-test, the difference was not

statistically significant. The t value was 0.968 the p value was 0.338. IMPACT OF

ANATOMICAL FACTORS ON THE DISEASE RECURRENCE VALVE ANATOMY Table 2.3

shows, 10 (83.3%) of patients with tricuspid valve and 2 (16.7%) with bicuspid valve had

disease recurrence.

When the recurrence was compared between the two groups, using Chi- squared test,

the difference was not statistically significant. The ?² value was 0.326 and the p value

was 0.568. ANATOMICAL LOCATION OF VSD From table 2.4, it is known that, of the 12

patients who had disease recurrence, 8 (66.7%) had subaortic type of VSD and 4(33.7%)

patients had doubly committed VSD. None of the patients with outlet muscular VSD had

disease recurrence.

When the recurrence was compared between the various types of VSD, using Chi-

squared test, the difference was not statistically significant. The ?² value was 2.210 and

the p value was 0.331. SURGICAL APROACH TO VSD CLOSURE We infer from table 2.5

that, 8 (66.7%) of the 12 patients with recurrence had their VSD closed from the right

atrium (trans-RA). Whereas, 4 (33.3%) of the 12 patients with recurrence had their VSD

closed from the pulmonary artery (trans-PA). Five patients (13.2%) whose VSD were

closed from the aorta and 2 (5.3%) patients whose VSD was closed via right ventricle did

not have any disease recurrence.

When the recurrence was compared between the various types of surgical approach,

using Chi- squared test, the difference was not statistically significant. The ?² value was

3.150 and the p value was 0.359. IMPACT OF THE PREOPERATIVE GRADE OF AORTIC

REGURGITATION ON THE DISEASE RECURRENCE Table 2.6 shows, a total of 17 (345)

patients had moderate grade of aortic regurgitation and 33 (66%) had severe grade of

aortic regurgitation prior to surgery.

Three (25%) patients with moderate grade of aortic regurgitation and 9 (75%) patients

with severe grade of aortic regurgitation had disease recurrence. When the recurrence

was compared between preoperative grades of aortic regurgitation, using Chi- squared

test, the difference was not statistically significant. The ?² value was 0.570 and the p

value was 0.45 EFFECT OF THE SURGICAL TECHNIQUES ON RECURRENCE RATES We

draw the following inferences from the data in Table 2.7.

Central plication A total of 11 patients had undergone this procedure accounting for 22

% of the repairs in this study. Of the 11 patients who underwent the procedure, 5

patients had disease recurrence. Hence, the recurrence rate was 45.45% for this

technique. Considering all the procedures, 5 out of the 12 cases who had recurrence

belong to this group, amounting to 41.7% of disease recurrence in this study.

Triangular resection A total of 13 patients had undergone this procedure accounting for

26 % of the repairs in this study. Of the 13 patients who underwent the procedure, 3

patients had disease recurrence. Hence, the recurrence rate was 23.08% for this

technique. Considering all the procedures, 3 out of the 12 cases who had recurrence

belong to this group, amounting to 25 % of disease recurrence in this study.

Trusler repair A total of 20 patients had undergone this procedure accounting for 40%

of the repairs in this study. Of the 20 patients who underwent the procedure, 4 had

disease recurrence. Hence, the recurrence rate was 20 % for this technique. Considering

all the procedures, 4 out of the 12 cases who had recurrence belong to this group,

amounting to 33.3% of disease recurrence in this study.

Pericardial patch repair A total of 2 patients had undergone this procedure accounting

for 4 % of the repairs in this study. Of the 2 patients who underwent the procedure,

none had disease recurrence. Hence, the recurrence rate for this technique was nil.

Considering all the procedures, none of the 12 cases who had recurrence belong to this

group, amounting to zero percent disease recurrence in this study.

Yacoub repair A total of 4 patients had undergone this procedure accounting for 8 % of

the repairs in this study. Of the 4 patients who underwent the procedure, none had

disease recurrence. Hence, the recurrence rate for this technique was nil. Considering all

the procedures, none of the 12 cases who had recurrence belong to this group,

amounting to zero percent disease recurrence in this study.

The disease recurrence was compared between the various techniques of aortic valve

repair, using Chi- squared test, the difference was not statistically significant. The ?²alue

was 4.468 and the p value was 0.346 EFFECT OF ANNULOPLASTY ON DISEASE

RECURRENCE From table 2.8, it is known that, 14 (28%) of the patients had undergone

annuloplasty along with the aortic valve repair in this study. Six out this 14 patients had

disease recurrence.

Whereas, six out of the 36 patients who had not undergone annuloplasty also had

disease recurrence. When the recurrence was compared between the two groups, using

Chi- squared test, the difference was not statistically significant. The ?² value was 3.791

and the p value was 0.052. THE EFFECT OF ADDITIONAL SURGICAL PROCEDURES ON

THE DISEASE RECURRENCE From table 2.9, it is known that, a total of 14 (28%) patients

had undergone additional surgical procedures. ICR was done in 11(22%) of the patients

of which 2 (4%) of the recurrence.

This accounted for 16.7% of recurrence in the study. SOV repair was done in 1 (2%)

patient who did not have recurrence. Mitral valve repair was done in 1(2%) who had

recurrence. This accounted for 4 % of recurrence in the study. When the recurrence was

compared between the groups, using Chi- squared test, the difference was not

statistically significant. The ?² value was 8.059 and the p value was 0.153. TREND OF THE

GRADES OF AORTIC REGURGITATION OVER THE FOLLOW UP PERIOD From table 3.1

we infer, At the time of discharge, 11 (22%) had no aortic regurgitation, 33 (66%) had

mild grade of aortic regurgitation, 6 (12%) had moderate grade of regurgitation and

none had severe grade of aortic regurgitation. The mean rank (grade of aortic

regurgitation) at the time of discharge was 2.98. At the time 1st month follow up, 9

(18%) had no aortic regurgitation, 35 (70%) had mild grade of aortic regurgitation, 6

(12%) had moderate grade of regurgitation and none had severe grade of aortic

regurgitation. The mean rank (grade of aortic regurgitation) at the time of discharge was

3.04.

At the time of 6th month follow up, 8 (16%) had no aortic regurgitation, 39 (78%) had

mild grade of aortic regurgitation, 3 (6%) had moderate grade of regurgitation and

none had severe grade of aortic regurgitation. The mean rank (grade of aortic

regurgitation) at the time of discharge was 2.89. At the time of 1st year follow up, 9

(18%) had no aortic regurgitation, 33 (66%) had mild grade of aortic regurgitation, 7

(14%) had moderate grade of regurgitation and 1 (2%) had severe grade of aortic

regurgitation. The mean rank (grade of aortic regurgitation) at the time of discharge was

3.07.

At the time of 2nd year follow up, 9(18%) had no aortic regurgitation, 34 (68%) had mild

grade of aortic regurgitation, 6 (12%) had moderate grade of regurgitation and 1(2%)

had severe grade of aortic regurgitation. The mean rank (grade of aortic regurgitation)

at the time of discharge was 3.02. The mean of ranks during each of the visit was

compared using Friedman test to assess any significant change in the trend of the grade

of AR during the follow up period. The p value was 0.749 and the difference was not

statistically significant DISCUSSION The earliest description of aortic cusp prolapse and

regurgitation complicating ventricular septal defects occurring in the outlet septal

region was in 1921 by Laubry and Pezzi.

Garamella and Starr et al in 1960 first descibed the surgical technique for this relatively

rare congenital heart disease. Since then, the surgical techniques had continued to

evolve. Yacoub et al in 1997 was the most recent to describe such a technique of valve

repair. With the surgical techniques being described more recently, the consensus

regarding the patient selection for each technique has been a grey area.

Similarly, though the natural course of disease has been elucidated in detail by many

authors, the factors predicting surgical success or failure have not been described,

particularly in the Asian population. Thus, this study aimed at identifying the factors that

predict a durable valve in this rare subset of patients. Hung-Chi Lue, Atsuyoshi Takao et

al had shown there is a tendency for increase in male preponderance in patients with

aortic regurgitation complicating VSD.

Similar to their observation, male patients in this study accounted for two third (66%) of

the population with girls forming the rest. Male population also accounted for 75 % of

the recurrence. But when the difference between the two groups was analysed the

difference was not statistically significant (p = 0.450).

Komai et al had concluded from their study that “the most prudent strategy for the

patient with VSD and aortic cusp prolapse is early closure of the VSD before the onset of

AR”. But in our study all patients had developed aortic regurgitation prior to the

procedure. The age of patients ranged from 1 year to 27 years. The mean age at surgery

being 8.65 ± 5.6 yrs. This is slightly higher to what Komai et al had speculated from their

study which had a median age of 4.6 years.

Hiba Gaafar Salih, Sameh R. Ismail et al in a recent study, similar to this study employing

patients with overt cusp prolapse prior to surgery had the mean age of patients 3.8 ±

2.6 years. The relatively higher age of our patients is because of a multitude of

socioeconomic factors rather than scientific factors.

The poor literacy level and low socioeconomic status of the population our institution

caters to are the major hurdles in early age at surgery. This accounts for the

considerable level of delay in diagnosis and referral, withstanding the fact of the long

waiting list of patients for surgery in our institution which is a tertiary care level hospital.

But, the comparison of mean age of patients who had disease recurrence and who did

not, had shown no statistically significant difference between the two groups. We thus

infer, aortic valve repair can be safely attempted even in the patients presenting late in

their natural course, with no extra added risk of recurrence with their younger

counterparts. Nevertheless, it is not a prudent option to delay the surgery.

This study had 6 (12%) patients with bicuspid aortic valve in contrast to the 1-2% in the

general population. The mechanism of aortic regurgitation in this group of patients was

El khoury type II caused by the cusp prolapse unrelated to the occurrence of the VSD. All

the 6 patients had a perimembranous type of VSD, 5 of which were repaired by the

transatrial approach while in the sixth case, it was repaired via the transaortic route. Two

among the 6 patients developed disease recurrence by definition, accounting for 16.7 %

of the total recurrences in the study.

In both the patients, the severity of regurgitation declined from grade 2 to grade 1 by

the end of first year and remained till the second year follow up. In contrast, patients

with tricuspid aortic valve formed 88% (44/50) of the total population with 10

recurrences accounting for 83.3% of recurrence. The comparison of recurrences between

the two groups had shown no statistically significant difference (p= 0.568).

This rules out the increased risk of recurrence after valve repair in patients with bicuspid

aortic valve in this study population. Most of the patients in this study had

perimembranous VSD accounting for 70 % of population, followed by doubly

committed VSD accounting for 22% and the rest 8% is by outlet muscular VSD.

This is in contrast to the results of previous study by Hisatomi K and Kosuga K et al,

which had documented increased incidence of doubly committed VSD in the Asian

population. Okita et al had shown increased risk of residual AR with perimembranous

VSD rather than outlet VSDs. When the recurrence of AR was compared between the

various types of VSD in this study, from table 2.4, the difference was not statistically

significant (p= 0.331).

With the echocardiographic evaluation of preoperative severity of aortic regurgitation,

34% of the study population had moderate (grade 2) regurgitation and 66% of the

population had severe (grade 3 or more) regurgitation. The incidence of recurrence was

25% and 75 % respectively, the difference between the two groups on comparison

showed no statistical significance (p= 0.450).

The severity of aortic regurgitation thus doesn‟t preclude a safe valve repair in patients

with AR complicating VSD. Trusler repair was the most frequently performed procedure

in this study with a total of 20 (40 %) cases. Four (20%) out of the 20 patients who

underwent this procedure had recurrence. Annuloplasty was done in 6 of these patients

for aortic annulus dilation (=2 SD).

ICR was done in 3 of these patients for right ventricular outflow obstruction. Also, from

table 3.2 and table 3.3 we know that, the type of VSD and the route of repair had no

bearing on the recurrence. The recurrence is hence independent of the type of VSD, the

surgical approach to VSD closure, performance of annuloplasty or an additional

procedure.

It is probably the growth of the valve per se accounting for the recurrence of the

disease. Further, the qualitative analysis of the regurgitant jet by Echocardiogram is

essential for elucidating the exact mechanism involved. But, this analysis was not done

in the study.

Similarly, the effect of all the other surgical techniques the patients had undergone was

analysed for the recurrence, including the Carpenteir technique, Yacoub technique and

pericardial patch repair. Analysis had revealed that there is no difference in terms of risk

of recurrence of one procedure over the other. Subanalysis of the effect of the

anatomical type of VSD and the surgical approach to VSD closure on the recurrence of

each of the surgical procedure, had also shown to have no statistically significant

difference (table 3.2 and table 3.3). Annuloplasty was done in 14 (28 %) patients with

dilated annulus.

This was in addition to addressing the leaflet pathology. Six out of 12 patients had

recurrence, but with no statistically significant difference on comparison. None of the

patients had stenosis during follow up. The addition of subcommissural annuloplasty

has thus found not been associated with additional risk to patients undergoing aortic

valve repair.

The trend of the grade of aortic regurgitation in the study population is as shown in

table 3.1. The mean of ranks during each visit was computed and was compared

between each visits. But the difference was not statistically significant. Though there was

minor changes in the number of patients in each of the grades, the mean of the ranks

over the entire study population had not changed significantly.

Although by the definition in this study,12 patients had developed recurrence, 11 of

those twelve patients had grade 2 (moderate) aortic regurgitation and only 1 patient

had severe (grade 3)aortic regurgitation. On analysis of the transthoracic

echocardiographic mechanism of the recurrence, all the recurrence were intravalvar

leaks.

All of them were detected on regular follow up visit and none of the patients were

symptomatic for the regurgitation during the follow up period. Excluding the one

patient who required valve replacement in the immediate postoperative period, none of

the other patients either of life in required a replacement or medical management for

their valve leak.

The quality of life in all the patients who had undergone valve repair had definitely

improved during follow up though it was not objectively analysed in the study. The

study was limited by the small sample size and its retrospective nature though it had

analysed a subset of patients with very low disease incidence. The duration of follow up

was also relatively less to determine the recurrence of the disease considering the

growth potential of the patients in this study who were predominantly in the pediatric

group. The subjective evidence of improvement in the quality of life of this population

was very evident in the study.

But an objective measurement of the quality of life of this population would have given

the real impact of valve repair in this rare subset of patients on their clinical and

socioeconomic status. Aortic valve repair in patients with aortic regurgitation

complicating ventricular septal defect was thus a safe and durable procedure

considering the short term outcomes. A prospective randomised trial would be the

solution to the unanswered questions in the study.

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