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Eur J Cardiothorac Surg 2004;26:754-761
© 2004 Elsevier Science NL

The left atrioventricular valve in partial atrioventricular septal defect: management strategy and surgical outcome

^a Department of Paediatric Cardiology, Royal Brompton Hospital, Sydney Street, London SW3 6NP, UK
^b Department of Cardiac Surgery, Royal Brompton Hospital, Sydney Street, London SW3 6NP, UK

Received 22 December 2003; received in revised form 26 April 2004; accepted 14 June 2004.

^* Corresponding author. Address: 318 Edgware Road, London W2 1DY, UK, Tel.: +44-20-7724-5267
e-mail: amira_alhay{at}hotmail.com

	Abstract

Top Abstract 1. Introduction 2. Materials and methods 3. Discussion 4. Conclusion References

 
Objective: To test the hypothesis that in patients with a partial atrioventricular septal defect (PAVSD) and a competent left atrioventricular valve (LAVV), sutures should be placed across the line of apposition of the superior and inferior bridging leaflets, septal commissure (SC), to prevent the development of regurgitation. Outcome of surgery and risk factors for the need for LAVV reoperation of patients with mild or no LAVV regurgitation (LAVVR) were evaluated. Background: Controversy over management of the LAVV in PAVSD. Method: One hundred and forty seven children with PAVSD underwent surgical repair at the Royal Brompton Hospital between January 1983 and December 1999. Of this group, 21 (16.7%) had LAVVR of sufficient severity to require surgical intervention and were therefore excluded from analysis. The median age and weight at repair of those with mild or no LAVVR was 4.1 years and 15.4 kg. One hundred and eight had normal chromosomes, 13 Down syndrome and five other syndromes. The interatrial communication was closed using a pericardial patch in 62.7% and with synthetic material in the remainder. Intraoperative testing of LAVV competence was undertaken using saline injection into the left ventricle. In 80.9%, sutures were placed across the line of apposition of the left sided superior and inferior bridging leaflets partially to close the SC (sometimes incorrectly named the mitral valve cleft). Result: The overall hospital mortality was 3.2% (95% confidence interval (CI) 1, 8.4%), which did not differ statistically in the last 20 years. No specific risk factors for early death were identified. Eleven patients (8.7%, 95% CI 4.7, 15.4%) required reoperation, 10 for LAVV repair and 1 resection of subaortic stenosis. Univariate analysis of risk factors for LAVV reoperation were low weight, relatively small size LAVV, the presence of a small preoperative interventricular interchordal communication and duration of ventilation. Ten (9.8%) of 102 patients in whom SC was sutured required LAVV reoperation but none for 24 in whom the commissure was left alone. Conclusion: The hypothesis that in the absence of preoperative LAVVR it is necessary to place sutures in the SC has not been proven. We consider that in addition to preoperative cross sectional echocardiographic assessment of LAVVR intraoperative evaluation of LAVV function allows discrimination between those valves where sutures to the septal commissure are necessary and those where the valve can be left undisturbed.

Key Words: Ostium primum atrial septal defect • Partial atrioventricular septal defect • Left atrioventricular valve • Septal commissure

Abbreviations: AVV, atrioventricular valve • CI, confidence interval • DOLAVV, double orifice left atrioventricular valve • LAVV, left atrioventricular valve • LAVV, regurgitation LAVVR • PAVSD, partial atrioventricular septal defect • SC, septal commissure

	1. Introduction

Top Abstract 1. Introduction 2. Materials and methods 3. Discussion 4. Conclusion References

 
In an ostium primum atrial septal defect, or partial atrioventricular septal defect (PAVSD), left and right atrioventricular valves (AVV) guard a common atrioventricular junction. The superior and inferior bridging leaflets are joined by a connecting tongue of tissue that separate left and right AVV orifices [1]. Between the left superior and inferior bridging leaflets is a line of apposition known as the septal commissure (SC), sometimes incorrectly named the mitral valve cleft. Carpentier [2] described this as a space between the leaflets that functions as a commissure rather than a cleft even if it is not supported by a papillary muscle. The trileaflet left atrioventricular valve (LAVV) may be regurgitant and less frequently stenotic, dysplastic, hypoplastic or have an accessory (double) orifice (DOLAVV). Early or late reoperation for significant residual LAVV regurgitation (LAVVR) continues to be a major cause of morbidity. The purpose of this study was to determine whether placing sutures in the SC in patients with no to mild LAVVR would obviate the need for a further bypass procedure to repair the LAVV. We have also evaluated outcome and risk factors for early death and LAVV reoperation.

	2. Materials and methods

Top Abstract 1. Introduction 2. Materials and methods 3. Discussion 4. Conclusion References

 
Between January 1983 and the end of 2003, 147 consecutive patients with a PAVSD underwent surgical repair at the Royal Brompton Hospital. The diagnosis was established by physical examination and cross sectional echocardiography. All patients had preoperative and early postoperative echocardiographic studies while follow-up echocardiography was undertaken biannually or when clinically indicated. Follow-up information on all patients was obtained up to 30 November 2003 with a median period of 150 months (range 46, 249). The interatrial communication was labeled as large when it measured more than 40% of the atrial septum in an optimized subcostal view. LAVVR was graded on the basis of the presence of an apical systolic murmur combined with colour flow mapping on cross sectional echocardiography (Table 1). In all cases assignment to a particular grade was undertaken prospectively by the echocardiographer prior to surgery and retrospectively by a second observer blinded to details of surgical technique. In 96% of cases, there was agreement on the grade of LAVVR but in 4% tapes were not available for retrospective analysis. Patients with moderate or severe 21 (14.3%) LAVVR were considered separately as this degree of LAVV incompetence invariably required valve repair (or rarely replacement). Our particular concern, however, has been to concentrate on those cases with minimal or no LAVV incompetence where a choice could be made as to whether the SC could be left alone. Cases with isomerism of right or left atrial appendages were excluded, as were cases where a biventricular repair was not considered feasible. Patients who required patch closure of an interventricular interchordal communication, intermediate type AVSD, were also excluded from the study.

2.1. Operative technique
PAVSD repair was carried out using a standard cardiopulmonary bypass technique with moderate or profound hypothermia (mean 22.3 °C, ranges 15–32 °C). Deep hypothermic circulatory arrest was used in six patients who weighed less than 3 kg, or were under 3 months. All had a single period of aortic cross-clamping (mean=45 min, range 27–91) and cold cardioplegia solution. The heart was arrested with cold crystalloid cardioplegia in 110 (87.3%) and blood cardioplegia in 16 (12%). Ice slush was applied to the surface of all hearts. The cardioplegic solution was instilled into the aortic root as per protocol and recovered from the left atrium as the coronary sinus had been left on the left side of the atrial septum. Blood cardioplegia was preferred in older children (large size).

The median cardiopulmonary bypass time was 50 min (range 13–90). In 77 (61.1%) patients, the ostium primum atrial septal defect was closed with a patch of autologous pericardium (two were fenestrated as the left ventricular inflow was considered restrictive), two were bovine pericardium and in 47 (37.3%) synthetic materials were used (46 Dacron and 1 Gortex). Patch was secured to the ventricular crest by a continuous 4-0 Ehthibond in 113 (89.4%) and by interrupted 4-0 Ethibond pledgets in 13 (10.6%). The interatrial communication was patched leaving the coronary sinus draining to the left atrium in 119 cases (94.4%). In three patients with left SVC to coronary sinus, the coronary sinus drained to the right atrium. In one patient with left SVC to left atrium diagnosed intraoperatively a full patch of bovine pericardium was used to reroute the left SVC into the right atrium.

We have reported previously our strategy with regard to management of the trileaflet LAVV [1]. Early in that series, the approach was simply to close the commissure between the bridging leaflets, although we questioned the logic in preventing regurgitation should none be present. In the present series, at operation we judge the leaflets, as they would lie during ventricular end diastole by filling the left ventricular cavity with saline solution injected rapidly by a syringe. When the cavity fills, the left ventricular component of the bridging leaflets bulge and tend to come together closes the SC the so-called cleft. The extent of any regurgitation is dependent on the accuracy of the apposition. When regurgitation is present its site is easily determined. As a consequence in 102 (81%) of 126 with minimal or no LAVVR as judged by cross sectional echocardiography, because of the intraoperative findings, sutures were placed in the SC using buttressed or pericardial pledget stitches. Following placement of sutures in the SC, the LAVV was retested to exclude any further regurgitation. Should there be residual regurgitation additional sutures would be placed providing in the surgeon's judgment this did not result in a stenotic valve. The LAVV was left as trileaflet in the remaining 24 patients. In 102 patients, sutures were placed in the SC so as to prevent regurgitation but without significantly narrowing the effective valve orifice. In 65 (63.7%) three or more sutures were required and in 37 (36.3%) one or two sutures. One patient also required an annuloplasty because of repeat testing, the LAVV was incompetent both between the superior and inferior bridging leaflets and centrally. In this patient, the SC was poorly defined and the area of coaptation of the bridging leaflets was not readily determined. The superior and inferior bridging leaflets were sutured together using four interrupted 5.0 prolene sutures. Retesting the LAVV, there was still central incompetence, which appeared to be due to annular dilatation. An annuloplasty stitch was used at the commissure between the mural and inferior bridging leaflets. The valve was tested and appeared competent as was confirmed by postoperative echocardiography. Eighteen patients had a small preoperative interventricular interchordal communication, 50% undergoing direct closure with one or two stitches. The others were not considered to require intervention.

Out of 12 patients with additional valvar pulmonary stenosis, five underwent an open valvotomy during operation. Two children underwent repair of coarctation of the aorta prior to repair of PAVSD. Two other patients found to have mild subaortic stenosis did not require intervention at the time of PAVSD repair nor during follow-up.

Following repair of PAVSD the median time to extubation was 1 day (range 0–18) and for discharge from hospital 6 days (3–66). Hospital death was defined as death within 30 days of repair of PAVSD. Reoperation was defined as a second operation that required cardiopulmonary bypass.

2.2. Statistical methods
Univariate analyses were used to assess whether there was a relationship between surgical outcome (hospital death and/or reoperation) and possible predictors. These predictors were demographic (age at operation, weight, sex, presence of Down or other syndromes), cardiac (size of interatrial communication, the degree of LAVVR, LAVV size, presence of a double orifice LAVV, dysplastic LAVV, presence of a small preoperative interventricular interchordal communication), and surgical variables (year at operation, type of patch used to close the interatrial communication, whether or not the SC was sutured, cardiopulmonary bypass time, aortic cross-clamp time and time of ventilation). Fisher's exact test was used for some categorical data with zero value of the cells that impede the use of other tests. Logistic regression was used when the outcome variables were binary (e.g. dead or alive) with an attempt to relate the probability of need for surgical outcome to possible predictor variables, e.g. age or LAVVR. Initially each variable was examined individually to see how strongly it was related to the outcome. Odds ratio with 95% confidence interval (CI) was recorded. A multivariate logistic regression analysis was then performed to find the best multivariate model for outcome. Selecting the significant variables from the multivariate model was carried out in a stepwise regression. A cut-off P=0.1 was used for removing variables and P=0.05 for adding variables. The results quoted were for the odds ratio and its 95% CI. Kaplan–Meier actuarial survival curves were calculated for freedom from reoperation using standard techniques [3]. Cox's regression test was used to assess whether there was a significant difference in survival from reoperation between statistically significant variables. P-value <0.05 was considered to be significant. All statistical analyses were conducted using the software package of social science (SPSS, version 9) and STATA, version 6 with no non-default option.

2.3. Result
Of 126 patients with Grade 0 to II LAVVR, four (3.2%, 95% CI 1, 8.4%) died within 30 days of operation as did one of 21 patients with Grades III to IV incompetence. The cause of death was low output state in one patient with known immune deficiency syndrome, inability to wean from cardiopulmonary bypass in two and a severe pulmonary hypertensive crisis with septicemia in one patient. Univariate analysis of risk factors for hospital death showed none of the listed variables to be risk factors. Following surgery, three (2%) had a pericardial effusion that required drainage, two developed pulmonary hypertensive crises and one developed a hemiparesis that resolved during follow-up.

Eleven of 126 patients with Grade 0-II required reoperation (8.7%, CI 4.7, 15.4%), 10 for LAVVR, and one for subaortic stenosis (with bicuspid aortic valve and mild arch hypoplasia). Of the 17 patients with moderate regurgitation Grade III 8 required LAVV reoperation as did one of four patients from the severe group (Grade IV). Reoperation was undertaken at a median time of 2 months (range 0.3–68.9) after the initial operation. Twenty year actuarial freedom from reoperation was 91.6% (Fig. 1) . Table 2 presents the details of patients who had reoperation after the initial repair. Intraoperative findings during LAVV reoperation in patient number 9 showed that the left SC was securely closed, however, there was severe annulus dilatation. A De Vaga Annuloplasty was carried out using two circumferential 4/0 Ethibond sutures. The LAVV was competent on testing. LAVV reoperation was undertaken in patient 10 and 11 to restore torn out stitches between the superior and inferior bridging leaflets. Both LAVV rings were markedly dilated; therefore, an annuloplasty was performed around the commissure between the bridging and mural leaflets. When the valves were tested they appeared to be completely competent. The other causes of postoperative LAVV dysfunction were dehiscence of sutures placed across the SC in four patients and failure of coaptation of LAVV leaflets with marked annular dilatation in three. These three cases were treated with LAVV replacement (size 21 mm HP St Jude medical prosthesis), as in addition to annular dilatation two had prolapsing and redundant bridging leaflets with elongation of the chordae and one had absence of the mural leaflet so that repair would result in LAVV stenosis.

View larger version (14K): [in this window] [in a new window]   Fig. 1. Actuarial freedom from reoperation after repair of PAVSD.

View larger version (15K): [in this window] [in a new window]   Fig. 2. Estimated curves for freedom from LAVV reoperation in relation to the presence or absence of small interventricular interchordal communication.

	3. Discussion

Top Abstract 1. Introduction 2. Materials and methods 3. Discussion 4. Conclusion References

 
The overall survival of patients undergoing repair of a PAVSD has improved considerably over the last three decades [1,4]. Our experience confirms that a PAVSD can be repaired with a low operative (3%) and no late mortality. These results are similar to other studies (0% [5,6], 1.6% [7], 6% [8], 7.9% [9], 10.7% [10], and 13% [11]). Correction of a PAVSD may be challenging because of difficulties in repairing the LAVV. Morphological features of the valve that contribute to this include the position of the commissures between the three leaflets of the LAVV, the abnormal position of the two papillary muscles in the left ventricle and there may be a deficiency of leaflet tissue especially in the left component of the inferior bridging leaflet [12].

Various surgical techniques have been advocated to improve valve function but despite this, postoperative LAVVR remain a clinically important issue. Particularly when the valve is initially non-regurgitant the decision whether or not to close the SC of the LAVV remains controversial. In 1965, Rastelli et al. [13] reported that in the absence of significant LAVVR, the need for suturing the line of apposition between left superior and inferior bridging leaflets is debatable. They commented that it is advantageous simply to close the interatrial communication when preoperative LAVV incompetence is absent. Similarly in 1978, Carpentier [2] advocated a trifoliate technique. Stewart et al. [14] also in 1987, left the commissure undisturbed in the absence of regurgitation. Conversely, King et al. [15] reported that their practice is to close the SC regardless of the degree of preoperative LAVV incompetence as is also advocated by El-Najdawi et al. recently [16]. The latter group, however, did not analyse separately these with minimal or no LAVVR preoperatively. Barnett et al. [17] reporting their experience with repair of PAVSD in adults found that LAVVR may eventually progress many years after repair whether or not the SC was sutured initially even if preoperative cardiac catheterisation showed no or mild regurgitation.

Ten (7.9%) of our patients with no or mild LAVVR required a second operation for LAVVR, three requiring valve replacement. This was similar to other studies [4,5–7,10,11,18], but was not as low as Portman et al. [8]. We found, somewhat unexpectedly that a small preoperative interventricular interchordal communication was an important risk factor for LAVV reoperation. One explanation is that the high velocity jet of blood across the restrictive interventricular interchordal communication may distort the LAVV leaflets because of a Venturi effect. Another possible explanation is that sutures used to close a relatively small hole may disturb the anatomy of the LAVV. Nakano et al. [19] found DOLAVV complicating PAVSD to be a significant predictor for late LAVVR. We did not find this although in our study of complete AVSD (Al-Hay et al.) [20] DOLAVV was an incremental risk factor for death but not for late regurgitation. In this study, there were nine patients with DOLAVV only one of whom required reoperation.

In our study, preoperative moderate to severe LAVVR was excluded because this is already known to be a major risk factor for failure of valve repair postoperatively [14,18]. Abbruzzese et al. [4] reported that in the absence of regurgitation, the LAVV can be left untouched with minimal risk for the need for reoperation. Thus, none of 25 unsutured LAVV required reoperation in our series. This is similar to an earlier series from the same institute [1]. In these patients with no or mild LAVVR on echocardiographic assessment, confirmation was performed intraoperatively by filling the left ventricle cavity with saline injected rapidly by a syringe and assessing valve competence by direct inspection. We stress the importance of this intraoperative evaluation which appears to offer optimal discrimination concerning which LAVV judged on cross sectional echocardiography to have minimal or no LAVVR can be left undisturbed. Currently LAVV competence is also assessed by transoesophageal echocardiography while weaning from cardiopulmonary bypass. If the valve is competent then we agree with others that the SC should be left untouched [2,13,14]. In the previous study [1], the number of patients where the LAVV was not sutured was small. Nonetheless, the hypothesis that in the absence of preoperative LAVVR, it is necessary to place sutures in the SC was not proven.

	4. Conclusion

Top Abstract 1. Introduction 2. Materials and methods 3. Discussion 4. Conclusion References

 
Our 21-year review confirms that a PAVSD can be operated with low hospital mortality and with a low risk of postoperative arrhythmias or heart block. The concerns about valve competence make long-term follow-up necessary in all patients. We consider that in addition to cross sectional echocardiographic assessment of LAVVR, intraoperative evaluation of LAVV function allows discrimination between those valves where sutures to the septal commisure are necessary and those where the valve can be left undisturbed. The contention that sutures should be placed in the SC even in the absence of regurgitation has not been proven. Our results suggest that if the valve is competent on intraoperative testing, it is better left alone. We also found that a small interventricular interchodal communication was associated with LAVVR whether or not it was closed with sutures.

4.1. Limitation of the study
The main limitation of our study is that of conducting a retrospective analysis over a long period. Furthermore this was not a randomized prospective investigation of the management of the LAVV but an observational study of outcomes. The study groups were unequal (102 stitched vs. 24 when the LAVV was left alone) which makes drawing definite conclusions difficult. We could, however, use the null hypothesis that our data does not support the need to intervene in the absence of valve regurgitation. We have not used direct intraoperative echocardiography or compared the use of intraoperative TOE with direct testing because it was unavailable in the early part of the study. This would, however, be of interest.

	Acknowledgments

 
We thank Miss Melissa, Wright Department of Epidemiology and Public Health, Imperial College for statistical help.

	References

Top Abstract 1. Introduction 2. Materials and methods 3. Discussion 4. Conclusion 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 Author home page(s): Christopher R. Lincoln Darryl F. Shore Elliot A. Shinebourne Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Al-Hay, A. A.A. Articles by Shinebourne, E. A. Search for Related Content PubMed PubMed Citation Articles by Al-Hay, A. A.A. Articles by Shinebourne, E. A. Related Collections Congenital - acyanotic