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a Department of Cardiovascular Surgery, German Heart Center Munich at the Technical University, Munich, Germany
b Department of Cardiac Surgery, University Clinic Schleswig-Holstein, Campus Luebeck, Luebeck, Germany
c Department of Cardiothoracic Surgery, Erasmus University Medical Center, Rotterdam, the Netherlands
d Sana Herzchirurgische Klinik, Stuttgart, Germany
e German Heart Center Berlin, Berlin, Germany
f Department of Mathematics, School of Science and Technology, University of Sussex, Brighton, United Kingdom
Accepted for publication April 22, 2009.
* Address correspondence to Dr Hörer, Department of Cardiovascular Surgery, German Heart Center Munich at the Technical University, Lazarettstrasse 36, Munich, D-80636, Germany (Email: hoerer{at}dhm.mhn.de).
Presented at the Forty-fifth Annual Meeting of The Society of Thoracic Surgeons, San Francisco, CA, Jan 26–28, 2009.
| Abstract |
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Methods: Neoaortic echo dimensions from 48 children (<16 years) undergoing Ross operation who had follow-up echocardiograms before age 20 were analyzed (mean follow-up, 5.1 ± 3.3 years).
Results: The mean age at the time of the Ross operation was 10.0 ± 4.3 years. Mean z values of the neoaortic annulus (1.5 ± 0.4), sinus (2.5 ± 0.4), and sinotubular junction (2.6 ± 0.9) when the autograft was implanted were significantly larger compared with normal values (p < 0.001, all). The mean z values significantly increased with follow-up at the level of the sinus (0.5 ± 0.1/year, p < 0.001) and the sinotubular junction (0.7 ± 0.2, p < 0.001), but not at the level of the annulus (0.1 ± 0.1, p = 0.59). AR increased with follow-up time (0.07 ± 0.02 grade/year, p < 0.001). AR increased with sinotubular junction diameter (p = 0.028), but there was not significant evidence of an association with annulus diameter (p = 0.25) or sinus diameter (p = 0.40).
Conclusions: Children undergoing Ross operation have larger neoaortic root dimensions than healthy children. Growth of the annulus matches somatic growth. The diameters of the sinus and the sinotubular junction increase significantly relative to somatic growth. The latter may explain the development of AR.
| Introduction |
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In children, the increase of neoaortic root dimensions is desirable; therefore, many surgeons prefer the pulmonary autograft to other prostheses because of its alleged growth potential [3, 6]. However, pathologic neoaortic root dilatation in children may also occur out of proportion to somatic growth. In contrast to the studies of adult patients, data on neoaortic dilatation in children are controversial. Growth in proportion to somatic growth as well as pathologic root dilatation has been reported [7–9]. So far, longitudinal parametric analyses of aortic root dimensions in exclusively pediatric patient populations are lacking. In addition, the effect of neoaortic root dimensions on aortic regurgitation is unclear [4, 7, 10]. Therefore, we analyzed longitudinal echocardiographic data of a pediatric Ross operated population to describe the changes of annulus, sinus, and sinotubular junction dimensions with time and to identify the relationship between aortic root dimensions and aortic regurgitation.
| Patients and Methods |
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Study Population and Operative Data
Data from the Dutch-German Ross Registry database were analyzed. The study included all patients who were aged younger than 16 years old at the time of the Ross operation and who had follow-up echocardiograms. Excluded were echocardiographic data from patients aged older than 20 years at the time of the examination. The study population included patient data from three Departments of Cardiac Surgery in Germany, and one Department of Cardiac Surgery in the Netherlands.
The Ross operations were performed between October 1988 and October 2006. The follow-up data from each center were entered into the database, and subsequently, a common systematic, prospective registry was started in January 2002. The responsible surgeon at each center determined the surgical technique. Root replacement was performed in 44 patients, and the subcoronary technique was applied in 4. In 9 patients the autograft implantation technique was modified for size matching purpose. Patients' characteristics and operative data are listed in Tables 1, 2, and 3.
Details of the operative techniques have been described elsewhere [11–13].
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Echocardiographic Data Acquisition and Measurements
Autograft dimensions were measured as described by Roman and colleagues [14] at three different levels: annulus at the level of the autograft leaflet hinges, sinus of Valsalva at the largest anteroposterior diameter, and sinotubular junction (supraaortic ridge level) at the distal rim of the sinuses of Valsalva. Z values of autograft dimensions were calculated according to the regression equations published by Daubeney and colleagues [15] from the body surface area (BSA) and the echocardiographically derived measurement. Aortic regurgitation (AR) was assessed by multiple techniques with parasternal long-axis and apical 5-chamber views.
Pulsed wave Doppler and color flow Doppler imaging were used for mapping the left ventricular outflow tract, including determination of the ratio of jet height to left ventricular outflow tract height. Continuous Doppler imaging was applied to measure the deceleration slope and pressure half-time of the autograft regurgitation jet. AR was graded with the use of standard criteria in most examinations [16].
Because this was a multicenter study, the final decision of AR grading was left to the decision of the responsible echocardiographer's preference and experience, and AR severity was reported on a scale of grade 0 to 4. Trace (trivial) aortic insufficiency, defined as a very tiny regurgitation jet in early diastole near the detection limit, was included in the analysis as grade 0.5.
We analyzed 129 measurements of the neoaortic root dimensions from 48 patients, and 403 measurements of the neoaortic regurgitation from 135 patients. Mean duration of the echocardiographic follow-up was 5.1 ± 3.3 years (range, 0.2 to 15 years) for the measurements of the dimensions, and 4.4 ± 2.4 years (range, 0.2 to 15 years) for the measurements of the neoaortic regurgitation.
Statistical Analysis
Frequencies are given as absolute numbers and percentages. Continuous data are expressed as means and standard deviation. Statistical analysis of clinical variables and initial fitting was performed using SPSS 16.0 software (SPSS Inc, Chicago, IL). The echocardiographic data of two or more echocardiographic observations per patient were analyzed by using a hierarchical multilevel linear model (MLWin 2.0, Centre for Multilevel Modeling, London, UK) that provides a linear regression line with an intercept and slope for each individual patient. The intercept ± standard error (SE) corresponds to the notional value at the time of surgery; the slope ± SE represents the annual progression of these measurements [17].
| Results |
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AR With Neoaortic Root Diameters
The same measurements as used for the analysis of neoaortic root dimension with follow-up time and BSA were analyzed. A linear model was chosen to model z value of the neoaortic root diameters and AR grade with the term:
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Reoperations
AR at the final follow-up of the studied 48 patients was none or trivial in 18 (38%), mild in 18 (38%), moderate in 8 (17%), severe in 1 (2%), and severest in 3 (6%). The 4 patients who presented with more than moderate AR required autograft explantation at a mean of 13.0 ± 3.7 years after the Ross operation.
| Comment |
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Because aortic root dimensions and regurgitation develop with time, parametric analyses of repeat longitudinal echo data may be more appropriate to describe changes of dimensions and regurgitation. Following these prerequisites in the present parametric analysis of a pediatric population, we obtained the following results: The aortic root dimensions immediate after the Ross operation are larger than in healthy children, and the annulus grows in proportion to the somatic growth of the child. In contrast, the sinus and the sinotubular junction dilate with time. AR develops slowly but significantly and is associated with a dilated sinotubular junction.
The initial dimensions of the neoaortic root immediately after the autograft implantation are determined by the size of the pulmonary autograft. However, previous studies reported different root dimensions immediately after the operation. Elkins and colleagues [8] found a mean z value of the annulus of –0.5, and thus almost normal dimensions. In contrast, Tantengco and colleagues [18] observed a mean z value of the annulus of 1.4. Our data support this observation, with a mean z value of the annulus of 1.5. This is not surprising, because the pulmonary autograft is usually larger than the aortic valve. In fact, the initial diameters of the neoaortic root were slightly larger compared with normal values obtained from the pulmonary root of healthy individuals. This may be due to immediate dilatation of the pulmonary autograft when transferred into the systemic circuit [18]. Hence, the mean initial diameters of the aortic root immediately after implantation are already larger than in healthy children.
Most studies that included adolescent patients and young adults reported dilatation of the aortic root during follow-up. Tantengco and colleagues [18] noticed pathologic dilation according to z values up to the first year after the Ross procedure, but without progress thereafter. Pasquali and colleagues [10] demonstrated a significant increase in z values of the annulus, sinus, and sinotubular junction over time. Kouchoukos and colleagues [4] observed a consistent increase of the sinus and sinotubular junction dimensions over time, but not of the annulus dimensions; however, their measurements were not indexed to the patients' BSA measurements.
Inconsistent data are reported with regard to proportional or disproportional growth of the neoaortic root in children. Simon and colleagues [7] observed growth parallel to somatic growth without undue dilatation. Elkins and colleagues [8] reported 10 of 23 patients presenting with pathologic annular dilatation after root replacement. In contrast, Solowiejczyk and colleagues [9] observed a disproportionate increase in the size of the sinus but minimal change in the z scores of the annulus.
Our observations affirm this difference in growth pattern between the annulus and the sinus: The curve depicting the z values of the annulus against the follow-up time (Fig 1A) parallels the x-axis. The curves displaying the annular dimension of the Ross population and the control population do not diverge with increasing BSA (Fig 2A). Hence, the annulus grows in proportion to somatic growth. In contrast, the curves depicting the z values of the sinus and the sinotubular junction increase over time (Fig 1B and C). The curves displaying the sinus and the sinotubular junction dimension of the Ross population and the control population diverge with increasing BSA (Fig 2B and C). Hence, there is dilatation of the sinus and the sinotubular junction, but not of the annulus.
According to previous studies, progressive neoaortic regurgitation and neoaortic root dilatation occurs over time in adolescents and adults; however, the correlation between AR and aortic root dilatation is not clear. In the large cohort reported by Pasquali and colleagues [10], freedom from moderate or more AR at 6 years was only 60%, and the freedom from sinus z value exceeding 8 was less than 40% at 6 years. Nevertheless, neoaortic root dilatation was not a significant predictor for AR in univariate or multivariate analysis. In the cohort reported by Kouchoukos and colleagues [4], the autografts in 11 of 119 patients required reoperation. At last follow-up, 8 of 97 patients presented with moderate AR. A consistent and substantial increase of the sinus and sinotunular junction dimensions occurred over time. The authors identified dilatation of the sinotubular junction as an independent predictor for progression of AR.
Do these data apply to children as well? Simon and colleagues [7] observed no dilatation of the aortic root over time and no increase in AR. In contrast, Takkenberg and colleagues [3] observed a significant increase in AR of 0.08 grades per year. In the present study population, AR increased with 0.06 grades per year. We observed a significant dilatation of the sinus and the sinotubular junction over time. In particular, the z values of the sinotubular junction increased with 0.7 per year. There was significant evidence that AR increases with increasing sinotubular junction diameters. Although the average effect was not strong, the mean z value of the sinotubular junction of patients exhibiting moderate or more AR during follow-up was 7.1 ± 3.3. In these patients, the mean sinotubular junction diameter was 38 mm with a maximum of 50 mm. This is worrisome, because the patients were younger than 20 years at the latest follow-up examination. Four patients required autograft explantation at a mean time of 13.0 ± 3.7 years after the Ross operation for severe AR. The mean z value of the sinotubular junction before autograft explantation was 8.8 ± 2.6. Hence, dilatation of the sinotubular junction that is out of proportion may be the cause for AR in children after the Ross operation.
This study has some limitations. The study design was a retrospective follow-up study covering a long period of patient inclusion. Changes in preoperative, operative, and postoperative management may have affected the outcome variables in a way not covered by our analysis.
The comparability of the echocardiographic findings at the time of final follow-up is limited because these data were obtained by various pediatric cardiologists in different outpatient clinics.
The graphs depicting the neoaortic root dimension against BSA do not consider different follow-up times after the Ross operation. Because there is dilation of the sinus and the sinotubular junction with time, the predicted dimensions for a grown patient who underwent a neonatal Ross operation may be underestimated by the graphs. The predicted dimensions for a grown patient who underwent the Ross procedure as an adolescent may also be overestimated.
Finally, the analysis included 4 patients in whom subcoronary technique was used, 5 in whom various techniques of annular enlargement were used, and 2 who underwent replacement of the ascending aorta. These modifications of the Ross operation may have affected AR grade and root dimension. However, the small number of patients precluded a meaningful subanalysis.
In conclusion, dilatation of the sinotubular junction and the sinus occurs after the Ross operation in children. The degree of aortic root enlargement is bothersome because it results in progressive AR and reoperations. Serial echocardiographic follow-up examinations are warranted and should include careful assessment of neoaortic root dimensions and monitoring of AR.
| Discussion |
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DR HÖRER: Four patients with the use of the subcoronary technique were included. These modifications of the Ross operation may have an impact on aortic regurgitation grade and root dimension. However, due to the small number of patients, a meaningful subanalysis was not possible.
DR FRANK PIGULA (Boston, MA): And could you just clarify, were there any efforts made to fix either the annulus or the sinotubular junction in these patients?
DR HÖRER: There were no additional procedures at the annulus.
DR PIGULA: So there weren't Teflon [DuPont, Wilmington, DE] strips or anything?
DR HÖRER: There were no Teflon strips. There were some enlargement procedures. Five patients underwent enlargement of the left ventricular outflow tract, but there were no enforcements of the annulus.
DR MARSHALL JACOBS (Newtown Square, PA): That was a very elegant and informative analysis. Some previous analyses have related the acquisition of increasing aortic valve insufficiency to the primary lesion or indication for surgery; AI [aortic insufficiency]; for example, isolated AI. Did you stratify the analysis to see if there was a relationship between primary type of aortic valve abnormality and rate of dilatation of the sinotubular junction or acquisition of insufficiency?
DR HÖRER: These are only 48 patients, so there was no significant impact of potential influencing factors like aortic regurgitation or bicuspid valve on outcomes. However, when we looked at the overall cohort of 180 patients, and checked the potential influencing factors for endpoints such as autograft reoperation, we also found a significant influence of predominant aortic regurgitation for autograft reoperation.
DR PIGULA: I just have one other question. Has this changed your practice in your institutions? Do you now consider doing some of the modifications, such as using a tube graft around the autograft, to support the sinuses or the sinotubular junction or any other modifications to avoid this?
DR HÖRER: When the Ross operation is applied to small children, the growth potential of the autograft is desired. So there is no sense in placing a tube graft around. But in those children with an already dilated aorta with the size of a grown-up patient, we place Teflon strips around the distal autograft anastomosis or we replace the ascending aorta with an adult-sized tube graft.
| Acknowledgments |
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| Footnotes |
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| References |
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