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Ann Thorac Surg 2006;81:1443-1449
© 2006 The Society of Thoracic Surgeons

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Original article: Cardiovascular

Ventricular Volumes in Ebstein's Anomaly: X-Ray Multislice Computed Tomography Before and After Repair

^a Department of Cardiovascular Surgery, Georges Pompidou European Hospital, Paris, France
^b Department of Radiology, Georges Pompidou European Hospital, Paris, France
^c Department of Pediatric Cardiology, Necker Hospital, Paris, France
^d Department of Physiology, Le Kremlin-Bicêtre Hospital, Le Kremlin-Bicêtre Cedex, France

Accepted for publication October 4, 2005.

^_* Address correspondence to Dr Chauvaud, Department of Cardiovascular Surgery, Georges Pompidou European Hospital, 20 rue Leblanc, 75015 Paris, France (Email: sylvain.chauvaud{at}egp.ap-hop-paris.fr).

	Abstract

Top Abstract Introduction Patients and Methods Results Comment Southern Thoracic Surgical... Acknowledgments References

 
BACKGROUND: This study was performed to measure right and left ventricular volumes in Ebstein's anomaly before and after operation.

METHODS: Twenty-six consecutive patients were operated on with tricuspid valve repair and plication of the atrialized right ventricle, associated with a bidirectional cavopulmonary shunt in 15 patients. Right ventricular (RV) and left ventricular end-diastolic and end-systolic volume indexes were measured by x-ray computerized tomography before and after surgical repair. Left ventricular stroke index and ejection fraction were calculated.

RESULTS: Before surgery, the mean stroke index of the atrialized RV was 36 ± 33 mL/m², with severe reduction in 9 patients and aneurysmal aspect in 2 patients. After surgery, the atrialized RV was no longer identifiable. Both RV end-diastolic volume index and stroke index of the remaining effective RV were reduced. Bidirectional cavopulmonary shunt was a determinant factor of decrease in the effective RV end-diastolic volume index after repair. Conversely, left ventricular ejection fraction and stroke index increased significantly after surgery.

CONCLUSIONS: The atrialized RV with dyskinesia seems a good indication for a plication. RV end-diastolic volume index of the effective RV decreased after surgery. In severe cases, bidirectional cavopulmonary shunt was useful by decreasing RV end-diastolic volume, thus preventing further RV dilation. In all cases left ventricular ejection fraction and stroke volume index increased after surgery.

	Introduction

Top Abstract Introduction Patients and Methods Results Comment Southern Thoracic Surgical... Acknowledgments References

 
Ebstein's anomaly is a congenital tricuspid valve malformation defined as a downward displacement of the septal leaflet. It is well established that the right (RV) [1] and the left (LV) [2] ventricular functions are impaired in some cases. The spectrum of pathologic features is wide [3, 4]. The impact of surgery on ventricular functions is not well investigated.

The present study of preoperative and postoperative RV and LV volumes (1) assesses the preoperative motion of the atrialized RV, (2) measures RV and LV volumes, and (3) detects the patients at risk of a postoperative RV failure.

	Patients and Methods

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Patients
Twenty-six consecutive patients with Ebstein's anomaly were studied. Mean age was 30 ± 16 year (13 males, 13 females; Table 1). All patients were symptomatic, with exertional dyspnea (100%), cyanosis (47%), and rhythm disturbances (35%). The degree of tricuspid valve incompetence, estimated (from 1+ to 4+) by preoperative echocardiography was as follows: 2+/4, 3 patients; 3+/4, 14 patients; 4+/4, 9 patients. According to Carpentier's classification of Ebstein's anomaly [5], 8 patients had type B and 18 patients type C (Fig 1). Briefly, in type B, the anterior leaflet is normally developed, the septal leaflet is moderately displaced, and the atrialized chamber is mildly developed. In type C, the anterior leaflet is restricted, the atrialized RV is large, and the tricuspid valve incompetence is massive.

View larger version (33K): [in this window] [in a new window]   Fig 1. Artist's view of the right ventricle. In type B (top), the septal leaflet is moderately displaced. The atrialized right ventricle (aRV) is extended no more than one third of the height of the ventricular septum. In type C (bottom), the anterior leaflet (AL) is restricted and the septal leaflet is displaced with distal attachement in the apex. The atrialized right ventricle is large. The atrialized right ventricle is limited by the atrioventricular junction and the attachment of the septal leaflet, corresponding to various parts of the inlet right ventricle. The effective right ventricle (eRV) is between the anterior leaflet and the pulmonary valves, including the trabecular and infundibular parts of the right ventricle.

Ventricular Evaluation
Evaluation of RV and LV volumes was performed with electron beam computerized tomography (Imatron C 100 and Siemens Evolution; Siemens AG D-80333 Munich, Germany). A morphologic study was carried through with 3-mm contiguous axial slices, electrocardiographically triggered after contrast injection (350 mg/mL iodine). The cineangiographic study was performed at 12 levels with 12 to 20 images per level in a short-axis view using two injections of contrast with a mean total volume of 170 mL in adult patients. Measurements were performed independently by two examinators (A.H. and E.M.). Postprocessing was done by manual drawing of the ventricular endocardial contours (Fig 2). End-diastolic (EDV) and end-systolic (ESV) volumes of each ventricle were recorded and indexed to the body surface area. Right ventricular and LV stroke indexes were calculated. The LV mass was also measured. Before surgery the volumes of both atrialized RV and effective RV (eRV) were measured. The atrialized RV was defined as the RV cavity located between the normal atrioventricular junction and the plane of insertion of the abnormal leaflet tissue in the RV cavity, whereas the eRV was defined as the RV cavity situated between the plane of the leaflets and the plane of the pulmonary cusps. After surgery the atrialized RV was excluded and the RV volume was reduced to the volume of the eRV (Figs 2, 3). The LV wall thickness was obtained by outlining the myocardium in multiple slices, and the LV mass (in grams) was calculated by multiplying the myocardial volume (in milliliters) by 1.05 (myocardial specific gravity). The LV mass was then indexed to the body surface area. Postoperative measurements were performed 7 to 10 days after repair.

View larger version (141K): [in this window] [in a new window]   Fig 2. Type C with massive tricuspid valve insufficiency. Patient 19 is a 38-year-old man with cyanosis, functional class IV, and cardiothoracic ratio (chest radiograph) of 0.80. Surgery included valve repair, a prosthetic ring, and a bidirectional cavopulmonary shunt. Electron-beam computed tomography section along the short-axis of the heart at the level of atrioventricular valves was performed. The black arrow (B) is the prosthetic ring. Contours of the right ventricle were manually drawn during end diastole (left) and end systole (right). (A) Preoperative conditions. Stroke volume index of the atrialized right ventricle (aRV) is –23 mL/m2; ejection fraction of the left ventricle (LV) is 0.52. (B) Postoperative conditions. Ejection fraction of the left ventricle is 0.74. White arrow indicates pericardial effusion. Eight years after repair the patient is leading a normal life, with 2+/4 residual tricuspid valve insufficiency. (eRV = effective right ventricle; RA = right atrium.)

View larger version (136K): [in this window] [in a new window]   Fig 3. Patient 13 is a 20-year-old woman with functional class IV rhythm dysturbances. Surgery included valve repair, a prosthetic ring, and no bidirectional cavopulmonary shunt. Contours of the right ventricle were manually drawn during end diastole (left) and end systole (right). (A) Preoperative conditions. Stroke volume index of the atrialized right ventricle is 28 mL/m2; indexed end-systolic volume is 118 mL/m2; indexed end-diastolic volume is 186 mL/m2; ejection fraction of the left ventricle is 0.53. (RA = right atrium.) (B) Postoperative conditions. End-systolic volume index is 79 mL/m2; end-diastolic volume index is 118 mL/m2; ejection fraction of the left ventricle is 0.69. Six years after repair the patient is leading a normal life, with permanent sinus rhythm and no tricuspid valve insufficiency.

Statistical Analysis
All values are given as mean ± standard deviation. The postoperative values were compared with the preoperative values using two-tailed Student's t test for paired samples, each patient serving as his or her own control. A p value of less than 0.05 was considered significant.

	Results

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After repair, patients were in New York Heart Association functional class I or II. Tricuspid valve regurgitation at echocardiography was 0 in 7 patients, 1+/4 in 14 patients, 2+/4 in 4 patients, and 3+/4 in 1 patient (Table 1).

Atrialized Right Ventricle
Before surgery, mean atrialized RV EDV index was 98 ± 49 mL/m², and mean atrialized RV ESV index was 62 ± 36 mL/m², thus resulting in a 36 ± 33 mL/m² mean atrialized RV stroke index. Nine patients had a limited atrialized RV stroke index (25 mL/m²); among them, 2 had an aneurysmal aspect of the atrialized chamber with dyskinetic wall motion. There was no correlation between limited atrialized RV stroke index and age or functional type. The atrialized RV was plicated in all cases as part of the initial technique [5]. Its postoperative volume was therefore no longer measurable.

Effective Right Ventricle
This part of the ventricle is situated between the insertion of the tricuspid leaflets and the pulmonary annulus. In the study population taken as a whole, both eRV EDV index and eRV stroke index significantly decreased (p < 0.05), whereas eRV ESV index slightly increased after surgery (Table 2), thus resulting in a reduction in eRV ejection fraction from 0.56 ± 0.11 to 0.41 ± 0.12. Noteworthy, the reduction in eRV EDV index after surgery was significant in the group with BCPS (p = 0.05), but not in the group without BCPS (Table 3).

View larger version (15K): [in this window] [in a new window]   Fig 4. Left ventricular ejection fraction before and after surgery.

	Comment

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Atrialized Right Ventricle
The atrialized RV results from an absence of delamination of the leaflets from the myocardium during organogenesis [17]. It has been suggested that the atrialized chamber has an abnormal contractility [1, 18]. In most of the previous studies, assessment of the atrialized RV remained qualitative. In the present study we measured the preoperative volumes of the atrialized RV, in an attempt to estimate its implication in the overall preoperative RV function.

There are major difficulties in interpreting the wall motion of the atrialized RV. Systolic movement of the atrialized RV wall results from many factors, such as the tricuspid valve incompetence, the "contractility" of the muscular wall, and the end-diastolic pressure of the RV. When the muscular wall is either motionless or expanding during ventricular systole, it can be assumed that the contractility is severely depressed. Thickening of the muscular wall during ventricular systole could be a more reliable measurement. However, technical difficulties of measurement will appear in the presence of paperlike thickness of the RV wall.

There is a controversy regarding the benefict of plication of the atrialized RV as a part of the surgical repair. Some authors develop conservative techniques without plication [19–21]. In contrast, we initiated the concept of longitudinal plication, which was used in all cases of our series [5, 7]. A similar technique was used by Quagebeur and colleagues [22]. Kiziltan and associates [18] were more eclectic and for tricuspid valve replacement performed a "plication of any thin and non contractile atrialized right ventricle." In the technique described by Hancock Friesen and coworkers [23], the posterior commissure is closed and the atrialized RV is shifted on the atrial side of the repair.

In the present study, 13 patients had severe impairment in wall motion of the atrialized RV chamber, among them 2 patients who had dyskinetic wall motions. In such cases it was considered inappropriate to leave intact this part with poor systolic motion of the RV, especially in the two cases with aneurysm. Furthermore, there is no evidence from the litterature that the motion of the impaired atrialized RV will recover after surgery.

We performed a plication in 11 patients with normal contractility of the atrialized RV, in accordance with the technique we have used since the beginning of our experience. From the present study our policy tends to be more selective regarding the plication. Normal motion of the atrialized RV is probably not an indication for plication. However, patients with impaired motion or dyskinesia seem to remain candidates for exclusion of the atrialized RV.

Effective Right Ventricle
End-diastolic volume of the eRV was significantly lower after operation. It is probably related to the decrease in RV preload secondary to the correction of the tricuspid valve regurgitation or to the BCPS. Confirmation of the favorable role of the BCPS was confirmed in patients with severe lesions, in which the decrease RV EDV index was significant. The benefit of this derivation was underlined by several authors [24, 25]. Salim and Coll [26] reported that the flow in the superior vena cava remained stable at 3 years of age. Therefore, the indication of BCPS seems to be reasonably extendable to adult patients. In our experience the operative survival was improved when BCPS was associated with intracardiac repair [7]. Marked cardiomegaly is a risk factor for mortality [27]. Subsequently elevated EDV is also of poor prognosis. Associated BCPS is a factor of decreasing of RV enlargement and by this mechanism can be useful.

The meaning of the indexed stroke volume is difficult to assess for many reasons. The cohort is not homogeneous in terms of age, duration of evolution, or amount of tricuspid valve regurgitation.

The decrease of the ejection fraction of the RV after surgery is not clear. Failure of myocardial protection is possible on account of the dilatation of the RV cavity. A phenomenon of stunned myocardium after repair of massive tricuspid valve insufficiency is another hypothesis. This loss of contractility had no important clinical implications. Long-term evaluation is necessaryfor assessement of RV function in this situation.

Left Ventricle
The most striking effect of the operation was the improvement of the LV ejection fraction. The LV in Ebstein's anomaly is not normal [11, 28]. Abnormalities were attributed to fibrosis and histologic anomalies [1]. Direct RV to LV interactions have been described in RV overloading in Ebstein's anomaly [29], as well as in other pathologic conditions [30, 31]. The paradoxic septal motion caused by RV overloading is a common feature in severe Ebstein's anomaly. On the other hand, both hypokinetic atrialized RV chamber and voluminous tricuspid regurgitation constitute a systolic "reservoir effect" that may contribute to RV to LV interactions in series, leading to an LV unloading and thus to a reduction in LV stroke volume [28]. Otherwise, the septal leaflet of the tricuspid valve and the ventricular septum have a similar development during fetal life [32].

From the present study it seems that the impairment of the LV is mostly functional and reversible. Left ventricular ejection fraction was restored to normal value early after surgery. The repair of the tricuspid valve incompetence together with the plication of the atrialized RV chamber increases the preload of the LV as well as the LV stroke volume. Noteworthy, the low preoperative LV EDV index remains almost unchanged a few days after surgery. This can be related to the diffuse fibrotic replacement of the LV myocardial muscle, often described in Ebstein's anomaly at histologic examination. Nevertheless, the LV stroke index was increased shortly after surgery, by reduction in LV ESV index, probably owing to both reduction in RV to LV interactions and LV preload increasing. These data offer a satisfactory explanation of the improvement of the clinical condition of the surgical patients.

Limitations of the Study
This study was based on a single surgical technique. Comparison among different techniques was not performed owing to the rarity of Ebstein's anomaly. The surgical population was heterogeneous, and statistical comparison of different techniques will need very large samples of patients. Our goals were to quantify the ventricular anomalies and to clarify the indications of surgical techniques.

Quantification of RV function is not routinely performed. Normal reference values are difficult to obtain. It is the reason why we used the patient as his or her own control. The main limitation is that the postoperative evaluation was performed 1 week after repair. One cannot extrapolate on a long-term basis. Further follow-up studies are needed to confirm the initial results.

We are aware that computerized tomography exposed the patient to heavy radiation and needed intranvenous contrast injection. At the present time the improvement of cardiac magnetic resonance seems to have better performances and fewer deleterious effects. Our goal was not to study the best apparatus but to analyze the volume variations. However, we have recently started a program for RV contractility study with cardiac magnetic resonance.

The method of volume calculation by manual drawing is subjective. Two analyses by independent examiners were performed, and the results were similar. We did not start a program for validation of this technique, which has already been published [15].

Conclusions
In Ebstein's anomaly, the atrialized RV is often depressed or dyskinetic. In such a situation, plication seems indicated. The major effect of surgery on RV volume is a decrease of EDVs related to the tricuspid valve repair. The systolic function of the LV is improved after surgery, most probably by increasing of the preload associated with reduction of RV to LV interactions.

	Southern Thoracic Surgical Association: Fifty-Third Annual Meeting

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The Fifty-Third Annual Meeting of the Southern Thoracic Surgical Association (STSA) will be held November 9-11, 2006, in Tucson, Arizona.

Members wishing to participate in the Scientific Program should submit an abstract by April 7, 2006, 12:00 Midnight, Central Daylight Time. Abstracts must be submitted electronically. Instructions for the abstract submission process can be found on the STSA Web site at www.stsa.org; on the CTSNet Web site at www.ctsnet.org; or in the back of the March issue of The Annals of Thoracic Surgery.

Manuscripts accepted for the Resident Competition must be submitted to the STSA headquarters office no later than September 15, 2006. The Resident Award will be based on abstract, presentation, and manuscript.

	Acknowledgments

Top Abstract Introduction Patients and Methods Results Comment Southern Thoracic Surgical... Acknowledgments References

 
We thank Marc de Leval, MD, Professor of Cardiac Surgery, Great Ormond Street Hospital for Children, for reviewing the manuscript.

	References

Top Abstract Introduction Patients and Methods Results Comment Southern Thoracic Surgical... Acknowledgments References

 

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This Article Abstract Full Text (PDF) Alert me when this article is cited Alert me if a correction is posted Citation Map Services Email this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Add to Personal Folders Download to citation manager Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Chauvaud, S. M. Articles by Hébert, J.-L. Search for Related Content PubMed PubMed Citation Articles by Chauvaud, S. M. Articles by Hébert, J.-L. Related Collections Congenital - cyanotic