Background The optimal substitute for severely diseased aorticvalves in children and young adults is unknown. The use of amechanical prosthesis requires permanent treatment of the patientwith anticoagulants and is associated with thromboembolic andhemorrhagic complications. Aortic-valve allografts and porcinebioprostheses, which do not necessitate anticoagulant therapy,may deteriorate and have limited durability.
Methods We therefore evaluated the use of the autologous pulmonaryvalve (i.e., the patient's own pulmonary valve) and the adjacentpulmonary artery as a replacement for the aortic valve and aorticsinuses in 33 patients. Five of the patients were from 8 to16 years of age, and 28 were from 20 to 47 years of age. Thepulmonary valve and the main pulmonary artery were used to replacethe diseased aortic valve and the adjacent aorta. The coronaryarteries were detached from the aorta and implanted into thepulmonary artery. The pulmonary valve and artery were replacedwith a cryopreserved pulmonary allograft.
Results There were no deaths during follow-up of up to 48 months(mean, 21 months). There were no episodes of infective endocarditis,and no reoperations on the aortic root were necessary. Also,there was no evidence on echocardiography of progressive dilatationof the autografts. With color-flow Doppler imaging, 22 patientswere found to have only trivial regurgitation or none, 9 patientsto have mild regurgitation, and no patients to have moderateor severe regurgitation across the autograft at the most recentfollow-up visit. The mean peak velocity of flow across the autograftwas 1.3 m per second (upper limit of normal, 1.8), indicatingthe absence of stenosis. One patient required reoperation forstenosis of the pulmonary allograft.
Conclusions Although the pulmonary-autograft procedure is morecomplex than simple aortic-valve replacement, it has been safelyapplied in selected patients, including young adults. Intermediatefollow-up indicates satisfactory function of the autografts,with no dilatation or progressive valvular regurgitation. Pulmonary-rootautografts may thus be the best available substitute for diseasedaortic valves in children and young adults.
The optimal substitute for a diseased aortic valve in childrenand young adults has not been identified. Although several mechanicalvalves have satisfactory hemodynamic characteristics, all necessitatelifelong treatment of the patient with anticoagulants. Thromboembolism,hemorrhage, and prosthetic-valve endocarditis remain importantcomplications1,2. Porcine bioprostheses, which do not necessitatelong-term anticoagulation, deteriorate rapidly in young patientsand have limited durability3. Aortic-valve allografts have excellenthydraulic function, necessitate no anticoagulant therapy, andare associated with a low incidence of thromboembolic complications.However, they also have limited durability in younger patients4,5,6.Because aortic-valve allografts are also used for other conditions(such as endocarditis, aneurysms that involve the aortic root,and reconstruction of the right ventricular outflow tract),they are not readily available.
In 1967, Ross7 described the use of the autologous pulmonaryvalve to replace the aortic valve. The valve was implanted withinthe aortic root, and the pulmonary valve was replaced with eitheran aortic or a pulmonary allograft. The viability of pulmonary-valveautografts in the aortic position has been documented, and theyare more resistant to deterioration than aortic allografts6,8.The availability of commercially prepared, cryopreserved aortic-valveand pulmonary-valve allografts in recent years and the developmentof safe techniques for the replacement of the aortic root havebroadened the indications for the replacement of the aorticroot with either aortic allografts or pulmonary autografts toinclude patients with isolated aortic-valve disease8,9,10,11.
Replacement of the aortic root with a pulmonary-root autograftin patients with aortic-valve disease is an alternative to replacementof the aortic valve with a mechanical or bioprosthetic valveor an allograft. This approach should result in optimal hemodynamicfunction, since the pulmonary valve is approximately the samesize as the aortic valve and is implanted with the adjacentsinuses of Valsalva. Also, the use of a pulmonary autograftshould, in principle, eliminate thromboembolic complicationswithout the need for anticoagulant therapy. In addition, theseviable valves have the potential for growth,6,12 a particularadvantage in children and young adults with aortic-valve disease.In this report we describe our experience in replacing the aorticroot with a pulmonary autograft in 33 patients with aortic-valvedisease and the results of serial echocardiographic studiesassessing the function of these autografts and of the pulmonaryallografts that were implanted in the right ventricular outflowtract.
Methods
Between June 1989 and May 1993, 33 patients with aortic-valvedisease underwent replacement of the aortic root with a pulmonaryautograft and replacement of the pulmonary root with a pulmonaryallograft. The patients ranged in age from 8 to 47 years (mean,31.4); 5 patients were from 8 to 16 years of age, and 28 werefrom 20 to 47 years of age. Twenty-two of the 33 were male.The indications for aortic-valve replacement are shown in Table 1.A bicuspid aortic valve, present in 23 patients, was themost common indication for operation. The majority of thesevalves were heavily calcified; two of these patients also hadaneurysms of the ascending aorta, which were replaced with Dacrongrafts. Four patients had aortic regurgitation resulting fromrheumatic heart disease, three had infective endocarditis, andthree had failure of a previously inserted porcine bioprosthesisor mechanical valve. One of the 33 patients had previously undergoneballoon valvuloplasty, and 8 had undergone one or more previousoperations on the aortic valve. The patients were for the mostpart healthy and generally had good left ventricular function.The mean preoperative New York Heart Association functionalclass was 2.2 (on a 4-point scale, with 1 indicating no symptomsand 4 indicating symptoms at rest).
All the patients underwent preoperative left-heart catheterizationand coronary arteriography to assess the severity of the aortic-valvedisease and left ventricular dysfunction, to determine the sizeand location of the coronary arteries, and to determine thesize and number of proximal septal perforating branches of theanterior descending coronary artery. These branches must beprotected during the removal of the pulmonary valve and theadjacent right ventricular myocardium in order to prevent infarctionof the ventricular septum13. Any large conal branches of theright coronary artery, which may connect with the anterior descendingsystem and which may be injured during the removal of the pulmonaryroot, were also identified.
Operative Procedure
Intraoperative transesophageal two-dimensional and color-flowDoppler echocardiography was performed in most of the patientsbefore cardiopulmonary bypass began in order to determine thediameter of the annulus and the sinuses of the pulmonary rootand to assess the competence of the pulmonary valve. The sizeof the aortic annulus and the aortic sinuses was also measured.Mismatches in size of more than 2 to 3 mm between the aorticand pulmonary roots or more than trivial regurgitation in thepulmonary valve were not found in any of the patients. Patientswith dilatation of the aortic root were not considered candidatesfor this procedure.
The technique for excision of the pulmonary root and its implantationinto the aortic root is shown in Figure 114. The procedure wasperformed during a period of cardiopulmonary bypass that averaged199 minutes (range, 105 to 286). During the period of myocardialischemia, which averaged 134 minutes (range, 70 to 200), themyocardium was protected by the infusion of cold blood (4 °C)or crystalloid cardioplegic solution directly into the coronaryostia or in a retrograde fashion into the coronary sinus. Externalcooling of the heart with iced crystalloid solution or witha cooling jacket was also used. After the discontinuation ofcardiopulmonary bypass, the competence of the pulmonary-autograftvalve and of the pulmonary-allograft valve in the right ventricularoutflow tract was assessed by transesophageal echocardiography.
Figure 1. Schematic Representation of the Operative Procedure.
First, the aortic valve and the adjacent aorta are excised, leaving buttons of aortic tissue surrounding the coronary arteries (Panel A). The pulmonary valve, with a small rim of right ventricular muscle and the main pulmonary artery, is also excised. Next, the pulmonary autograft is sutured to the aortic annulus and to the distal aorta, and the coronary arteries are attached to openings in the pulmonary artery (Panel B). A pulmonary-root allograft is then sutured into the right ventricular outflow tract (Panel C).
Postoperative Evaluation and Management
Transthoracic M-mode, two-dimensional, color-flow, and Dopplerechocardiograms were obtained before discharge from the hospitaland every 6 to 12 months thereafter. The presence of regurgitationin the autograft was determined by continuous-wave, pulsed-wave,and color-flow Doppler methods. The severity of regurgitationwas measured with the method of Perry et al.,15 in which theratio of the width of the jet of regurgitation to the diameterof the left ventricular outflow tract at the level of the valveannulus is determined. This measurement has been shown to correlatewell with the severity of aortic regurgitation as indicatedby angiography15. A ratio of less than 0.2 was considered toindicate trivial regurgitation; a ratio of 0.2 to 0.39, mildregurgitation; 0.4 to 0.6, moderate regurgitation; and morethan 0.6, severe regurgitation. A numerical grade was assignedto each of these ranges (Table 2). Using the method of Romanet al.,16 we measured the three dimensions of the pulmonary-rootautograft at end-diastole: the diameter at the annulus, themaximal diameter of the sinuses of Valsalva, and the diameterat the supravalvular ridge. We also measured the peak velocityof the flow across the pulmonary-autograft and pulmonary-allograftvalves in order to detect evidence of any obstruction of bloodflow.
Table 2. Echocardiographic Assessment of Regurgitation in the Autograft.
The patients were evaluated at 6-to-12-month intervals aftersurgery for symptoms and signs of valve regurgitation and forother cardiac abnormalities. No patient was lost to follow-up.No anticoagulant or antiplatelet drugs were administered toany of the patients.
Statistical Analysis
The dimensions of the valve autograft were expressed as means±SD. Repeated-measures analysis of variance was usedto compare these measurements and the numerical values for theseverity of regurgitation in the autograft at each echocardiographicstudy for each patient. A two-tailed P value of <0.05 wasconsidered to indicate statistical significance.
Results
Mortality and Morbidity
There were no deaths in the hospital. One patient required earlyreoperation for bleeding. Twenty-two patients (67 percent) didnot require transfusion of homologous blood products duringthe perioperative period. Fifteen of these patients had predepositedone or two units of autologous blood, which were transfusedduring or after the operation. In the 11 patients who requiredhomologous blood, a mean of 2.7 units of red cells was transfused.There was no electrocardiographic or echocardiographic evidenceof perioperative myocardial infarction in any patient. Sevenpatients had a pattern indicative of right bundle-branch blockafter the operation. In one patient, an eight-year-old childwho had previously undergone balloon angioplasty of the aorticvalve, complete heart block developed and implantation of apermanent pacemaker was required. One patient had partial homonymoushemianopia. This was probably the result of intraoperative embolizationof a fragment from the severely diseased aortic valve. The medianpostoperative stay in the hospital was 7 days (range, 4 to 32).
There have been no deaths during the follow-up period, whichat this writing extends to 48 months (mean duration of follow-up,21 months), nor have there been any episodes of thromboembolismor infective endocarditis. No reoperations have been requiredon the valve autografts. One 15-year-old patient required reoperationfor circumferential stenosis of the pulmonary allograft in theright ventricular outflow tract above the level of the pulmonaryvalve 16 months after the initial operation. The obstructionwas successfully relieved with a pericardial patch. An aortic-rootangiogram obtained immediately before the reoperation showeda normal-sized aortic root and no regurgitation in the autograft.The mean New York Heart Association class at the most recentfollow-up was 1.0 for the 33 patients.
Echocardiographic Studies
Pulmonary-Autograft Function
The numerical grades for the severity of aortic-autograft regurgitationin 31 patients, as determined by echocardiography, are shownin Figure 2. At the initial postoperative study, performed inthe first month after the operation, 29 patients had trivialregurgitation or none, and 4 patients had mild regurgitation.At the most recent follow-up study, 22 patients had only trivialor no regurgitation, 9 patients had mild regurgitation, andno patients had moderate or severe regurgitation across theautograft. Of the 16 patients who were examined 19 to 24 monthspostoperatively, 11 had trivial regurgitation or none, and 5had mild regurgitation. For this subgroup, the mean regurgitationscore was 0.34 ±0.30 at the initial postoperative examinationand 0.52 ±0.44 at the later study (P = 0.3). For thegroup as a whole, there was no appreciable increase in the severityof regurgitation with time.
Figure 2. Serial Echocardiographic Assessments of the Severity of Regurgitation in the Pulmonary Autograft in 31 Patients.
The numerical grades were assigned according to the severity of regurgitation, as follows: 0, none; 0.5, trivial; 1.0 to 1.5, mild; 2.0, moderate; and 3.0, severe.
Continuous-wave Doppler echocardiography across the pulmonaryautograft at the initial postoperative study showed mildly increasedflow velocity (from 2.0 to 2.2 m per second) in six patients(18 percent of the group). The upper limit of normal is 1.8m per second. At the most recent examination, all values forvelocity were within the normal range (mean, 1.3 m per second;range, 1.0 to 1.8). The early postoperative increase in velocityprobably reflected a hyperdynamic state rather than valvularobstruction.
Dimensions of the Autograft
The mean values for the three diameters of the autograft (annulus,sinuses of Valsalva, and supravalvular ridge) soon after theoperation and at the most recent follow-up visit (the intervalbetween the two measurements was longer than three months for31 patients) were as follows: 2.2 ±0.4 as compared with2.2 ±0.3 cm at the annulus, 3.1 ±0.6 as comparedwith 3.3 ±0.5 cm at the sinuses of Valsalva, and 2.5±0.4 as compared with 2.6 ±0.4 cm at the supravalvularridge. None of the differences were statistically significant.For the 16 patients who were examined early after surgery and19 to 24 months thereafter, the mean diameters at the annuluswere 2.3 ±0.3 and 2.3 ±0.3 cm, those at the sinusesof Valsalva were 3.3 ±0.5 and 3.4 ±0.6 cm, andthose at the supravalvular ridge were 2.6 ±0.4 and 2.6±0.4 cm. These differences were also not statisticallysignificant.
Pulmonary-Allograft Function
At the initial postoperative study, there was no regurgitationin the pulmonary-allograft valve in the right ventricular outflowtract in 18 patients and trivial or mild regurgitation in 13patients. At the time of the most recent study, mild regurgitationhad developed in three patients who initially had no pulmonaryregurgitation, and three patients who initially had trivialregurgitation or none had no regurgitation.
At the initial study, continuous-wave Doppler echocardiographyacross the pulmonary allograft showed a mild overall elevationin systolic velocity (mean, 1.1 ±0.4 m per second; range,0.6 to 2.0). The upper limit of normal for the native pulmonaryvalve is 0.8 m per second. These velocities returned to normalin later studies in most of the patients, suggesting that theearly elevations were due to postoperative hemodynamic changes.During the follow-up period, however, seven patients were foundto have moderate pressure gradients (mean, 26 ±14 mmHg; range, 12 to 45), which were measured at a point distalto the pulmonary-allograft valve, primarily in the vicinityof the anastomosis between the arterial portion of the allograftand the native pulmonary artery. The gradients were confirmedby right-heart catheterization in two patients. In one of thepatients, who had a measured gradient of 65 mm Hg at cardiaccatheterization, the obstruction was surgically corrected.
Discussion
Although replacement of the aortic root with a pulmonary autograftin patients with isolated aortic-valve disease is a longer andmore complex procedure than simple aortic-valve replacement,we and others12,17 have documented the safety of the procedure.In our series, there were no deaths in the hospital and no seriouspostoperative complications that could be attributed to theoperative technique. With the methods of intraoperative myocardialprotection that we employed, which included cold cardioplegiaand surface cooling of the myocardium, intraoperative and postoperativemyocardial dysfunction was minimal despite a duration of myocardialischemia longer than that required for simple aortic-valve replacement.Only one third of the patients required transfusion of homologousblood products. Although there is the potential for injury tothe septal branches of the left anterior descending coronaryartery during the removal of the pulmonary root, no evidenceof septal myocardial infarction was detected in any of the patients.
The advantages of using the pulmonary root as a substitute forthe aortic root in patients with aortic-valve disease includethe use of autologous tissue with documented long-term viability,8the optimal or near-optimal alignment and function of the valveleaflets, since the sinuses of Valsalva are also transplanted,and the absence of substantial transvalvular pressure gradients,thromboembolism, and the need for anticoagulant therapy12. Thereis also evidence of growth of the autograft, which makes itan attractive option for aortic-valve replacement in infantsand children6,12.
Aortic allografts that are implanted in the aortic root havehemodynamic characteristics that are similar to those of pulmonaryautografts; they are also associated with a low incidence ofthromboembolism and endocarditis, and they do not necessitatethe use of anticoagulants or antiplatelet drugs. However, valvedegeneration and reoperation because of mechanical failure occurmore often in younger than in older patients4,5,6. In the experienceof Gerosa et al.,9 aortic or pulmonary allografts that are implantedin the right ventricular outflow tract to replace the excisedpulmonary root have a cumulative rate of freedom from valvefailure or reoperation that exceeds 80 percent at 16 years.This freedom from valve failure or reoperation is superior tothat for aortic allografts in the left ventricular outflow tract.Mild or moderate allograft stenosis or incompetence is likelyto be tolerated better in the low-pressure right ventricularoutflow tract than in the left ventricular outflow tract. Thus,the need for a second operation to remove a malfunctioning ordegenerated allograft should be substantially reduced or possiblyeliminated if the allograft is placed in the right ventricularrather than the left ventricular outflow tract.
The chief uncertainty regarding the use of pulmonary-root autograftsin the aortic position is the potential for dilatation of thewall of the pulmonary artery and the development of progressivevalvular regurgitation as a result of continued exposure tosystemic pressure. Experimental studies by Gorczynski et al.18have demonstrated that the leaflets of the pulmonary valve havea tensile strength that equals or exceeds that of aortic-valveleaflets. In an in vitro model of the circulation, Weerasenaet al.19 observed a smaller drop in pressure across the pulmonicvalve at systemic pressures than at pressures and flows thatoccur in the pulmonary artery. Furthermore, an increase in pressurehad a limited effect on regurgitation in this model, and atthe highest pressures most pulmonary valves closed completely,producing a competent valve19. Inward movement of the elasticarterial wall during diastole absorbs energy and reduces dynamicloading during closure19. These conditions may not exist whenonly the pulmonary valve or an aortic-allograft valve is implantedinside the aortic root, without the sinuses of Valsalva. Furthermore,the aortic wall to which a free pulmonary-valve autograft oraortic-valve allograft is attached may be noncompliant and mayprevent the optimal functioning of the valve leaflets. Theremay also be misalignment of the valve commissures and cuspswith a free graft, which will place greater stress on the valvecomponents. These findings argue for the use of the entire pulmonaryroot rather than the pulmonary valve as a substitute for a diseasedaortic valve.
Serial echocardiographic measurements of the three dimensionsof the pulmonary-root autografts in 31 patients demonstratedno increase in the size of the autograft for up to 48 monthsafter valve replacement (mean follow-up, 21 months). To ourknowledge, this is the largest series of patients with pulmonary-rootautografts in whom sequential measurements of these dimensionsand semiquantitative estimates of the degree of aortic regurgitationhave been performed.
Sievers et al.17 observed no significant increase in the diameterof seven pulmonary-root autografts at the commissural levelin adult patients a mean of 12.5 ±7 months after surgery.Elkins et al.12 made sequential measurements of autografts in13 of their 22 patients, all of whom were children. The meanduration of follow-up for their 21 surviving patients was approximately21 months. Three patients were followed for more than 48 months,the longest follow-up being 58 months. Since growth occurredin many of these patients, it was not possible to differentiateclearly the growth of the pulmonary-root autograft from dilatation.The absence of progressive dilatation of the autograft in ourpatients was associated with an absence of progression of aorticregurgitation. Similar findings were reported by Sievers etal.17. Longer follow-up will be necessary to determine whetherlate changes will occur in the pulmonary arterial wall or inthe pulmonary leaflets and whether such changes result in dilatationof the root and valvular regurgitation in the autograft.
Although replacement of the aortic root with a pulmonary rootis a more complex operation than aortic-valve replacement inpatients with aortic-valve disease, it has been used safelyin a selected group of children and young adults with no earlymortality or graft-related morbidity. Follow-up extending foras long as four years indicated excellent function of the autografts,with no evidence of dilatation or progressive valvular regurgitation.There have been no infectious or thromboembolic complicationsand no late deaths. If longer follow-up shows no evidence ofthe progression of regurgitation in the implanted valve, a pulmonaryautograft may be the optimal substitute for a diseased aorticvalve in children and young adults.
We are indebted to Drs. Donald Ross, Ronald Elkins, and JohnW. Kirklin for their advice and encouragement; to Dr. ThomasWareing, who operated on one of the patients; and to PamelaPigg and Nikki Gratigny for assistance in the preparation ofthe manuscript.
Source Information
From the Divisions of Cardiothoracic Surgery (N.T.K., T.L.S., S.F.M., J.B.P.) and Cardiology (V.G.D.-R.), Washington University School of Medicine, St. Louis.
Address reprint requests to Dr. Kouchoukos at the Department of Surgery, Jewish Hospital at Washington University Medical Center, 216 S. Kingshighway Blvd., St. Louis, MO 63110.
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