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J Thorac Cardiovasc Surg 2000;119:880-889
© 2000 The American Association for Thoracic Surgery
Surgery For Congenital Heart Disease |
From the Section of Pediatric Cardiology,a the Division of Thoracic and Cardiovascular Surgery,b and the Section of Biostatistics,c Mayo Clinic and Mayo Foundation, Rochester, Minn.
Address for reprints: David J. Driscoll, MD, Mayo Clinic, 200 First St SW, Rochester, MN 55905 (E-mail: driscoll.david @mayo.edu ).
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Abstract |
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 Top Abstract IntroductionPatients and methods Results Discussion Conclusions References |
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Introduction |
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TopAbstractIntroductionPatients and methods Results Discussion Conclusions References |
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1 and since then there have been reports of several reasonably large operative series. 1-16 The purpose of this study was to describe the long-term outcome of the largest reported series of patients who underwent surgical repair of partial ASD at one institution. We determined the incidences of survival, reoperation, and occurrence of significant left atrioventricular valve regurgitation, stenosis, left ventricular outflow tract obstruction, and arrhythmia. |
Patients and methods |
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TopAbstractIntroductionPatients and methods Results Discussion Conclusions References |
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2,5,8,10 The current study, however, includes a considerably longer follow-up period and larger number of patients. This study was approved by the Mayo Foundation Institutional Review Board.
The Mayo medical record of each patient was reviewed, and data were collected regarding the anatomic details of the cardiac defect and details of the operation, especially whether the left atrioventricular valve cleft was closed. Passive follow-up was done with the Mayo medical records to ascertain vital status and outcome events of interest (residual or recurrent ASD, left atrioventricular valve stenosis, and left atrioventricular valve regurgitation, left ventricular outflow tract obstruction, bacterial endocarditis, cardiac reoperation, and arrhythmias). Active follow-up was done with a questionnaire sent to each of the patients not known to be dead and for whom current information regarding the outcome events of interest was not available in the Mayo medical record as of June 1, 1995. 9 For the few patients lost to follow-up, a nongovernmental index that tracks death information on a national basis (www.ancestry.com ) was searched to ascertain vital status.
The presence and severity of left atrioventricular valve regurgitation, stenosis, arrhythmias, and left ventricular outflow obstruction were assessed from clinical evaluations, echocardiographic data, cardiac catheterization data, and operative data contained in the Mayo medical record or obtained from the medical questionnaire sent to the patient or the patient’s physician.
Left atrioventricular valve regurgitation was considered to be present if the patient underwent operation for regurgitation or if there was clinical, angiographic, or Doppler echocardiographic evidence for left atrioventricular valve regurgitation. The severity of the regurgitation was based on the severity assigned by the attending physician and recorded in the medical record. The severity assigned was associated with the first recorded observation of regurgitation subsequent to the primary operation.
Left atrioventricular valve stenosis was considered to be present if the patient underwent operation or reoperation for stenosis or if there was a left atrial—left ventricular end-diastolic pressure gradient of more than 3 mm Hg by cardiac catheterization or Doppler echocardiography.
Left ventricular outflow tract obstruction was deemed to be present if there was 5 mm Hg or more pressure gradient across the left ventricular outflow tract measured at cardiac catheterization (peak-to-peak gradient) or Doppler echocardiography (mean gradient) or if operation for relief of obstruction was performed.
A clinically significant arrhythmia was defined as one for which an antiarrhythmia drug (excluding digoxin) treatment or a pacemaker was required.
The date of onset of the event of interest was based on the date of documentation of that event most proximal to the date of the original operation.
Continuous variables are summarized as a median with corresponding 25th (Q1) and 75th (Q3) percentiles, and comparisons between patient groups were based on the Wilcoxon rank sum test. Discrete variables are summarized as a percentage of the total, and comparisons between patient groups were based on the 
2 test. Incident outcomes of primary interest consisted of all cause deaths, first reoperation, left atrioventricular valve regurgitation and/or stenosis, left ventricular outflow tract obstruction, and arrhythmias. Survival-free events were estimated with the Kaplan-Meier method. Expected survival was estimated with the life tables for the United States white population and observed survival included in hospital deaths. Associations between time to first end point with clinical and surgical findings were investigated within the Cox proportional hazards framework, adjusting for decade of surgery. Independent variables included age at operation, sex, decade of operation, suture closure of cleft, parachute valve malformation, double-orifice valve, and presence of complete or partial cleft. Time of surgery was indicated as 1950s to 1960s, 1970s, and 1980s to 1990s and was used as an adjusting factor to control for the potential changes in referral patterns in the time of this study. Determinants of long-term survival were investigated in subjects with at least 30 days of follow-up and excluded deaths occurring less than 30 days after operation. Events occurring within 30 days of operation are summarized as a percentage of the total cohort. When all-cause deaths were considered, subjects with left atrioventricular valve regurgitation or stenosis and left ventricular outflow tract obstruction as end points were censored at the time of any prior reoperation or last available follow-up date. Censoring was imposed at reoperation because of the potential of altering the risk of the development of subsequent events, which could not be accounted for at baseline.
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Results |
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TopAbstractIntroductionPatients and methods Results Discussion Conclusions References |
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Follow-up information was obtained for 211 of the 248 patients who were sent questionnaires. Of the 37 patients without follow-up, 13 patients were ascertained to be deceased and 24 patients were lost to follow-up. For these 24 patients, we confirmed that 3 patients had died. The remaining 21 patients were censored for survival analysis at the date they were last known to be alive. Of the 248 patients, 9 patients did not respond to the survey, but their vital status was ascertained.
A complete left atrioventricular valve cleft was present in 247 patients; a partial cleft was present in 74 patients, and 4 patients had no cleft. A double-orifice valve was present in 23 patients, and a parachute valve was present in 6 patients. Combination of a double-orifice valve and complete cleft of the valve occurred in 9 patients. Four patients had a complete cleft that was associated with a parachute valve. Ten patients had a double-orifice valve and partial cleft. Some patients had more than one left atrioventricular valve abnormality and were counted more than once.
Surgical procedure
Eight patients (2%) underwent suture closure of the ASD, and 326 patients (98%) underwent patch closure. As part of the initial operation, 283 patients (85%) underwent suture closure of a left atrioventricular valve cleft, 17 patients (5%) underwent left atrioventricular valve annuloplasty, 5 patients (1.5%) underwent atrioventricular valve replacement, and 6 patients (1.8%) underwent left ventricular outflow tract operation for relief of obstruction. The postoperative hospital stay ranged from 5 to 46 days (median, 9 days; Q1, 8 days; Q3, 11 days ). The median duration of follow-up for these 334 patients was 19 years (Q1, 10 years; Q3, 29 years).
Survival
There were 41 deaths observed. The estimated survival in the 334 patients was 97% 30 days after operation, 94% at 5 years, 93% at 10 years, 87% at 20 years, 85% at 30 years, and 76% at 40 years, compared with 100%, 99%, 98%, 97%, 94%, and 92% in an age- and sex-matched control population, respectively (P < .001; Fig 1). Ten of these deaths occurred less than 30 days after the primary operation. All of these early deaths occurred before 1980.
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The reported cause of death was determined for 33 of the 41 deaths. There were 24 reported cardiovascular-related deaths. Additionally, there were 3 deaths from drowning, 3 deaths from motor vehicle accidents, 8 deaths from unknown cause, and 3 noncardiac deaths.
There was only 1 death among the 18 patients with Down syndrome. Hence death for patients with or without Down syndrome was similar.
Reoperation
Reoperation was performed for 38 patients (11%; Fig 2). One patient died at reoperation, and 1 additional patient died within 30 days of reoperation. The median interval between the primary repair and the subsequent reoperation was 17.8 years (Q1, 7.6 years; Q3, 28.2 years). The estimated incidence of reoperation was 3% at 1 year after initial operation, 6% at 5 years, 8% at 10 years, 12% at 20 years, 17% at 30 years, and 22% at 40 years. At reoperation, there were 7 incidents of residual ASD closure, 5 of which were at the first reoperation and 2 were at subsequent reoperation. There were 5 incidents of closure of a left atrioventricular valve cleft, 6 incidents of atrioventricular valve annuloplasty, 14 incidents of atrioventricular valve replacement, and 8 incidents for the relief of left ventricular outflow tract obstruction. Some patients had combinations of these procedures as part of one operation. After we controlled for the decade of the operation, patients who had closure of the cleft of the left atrioventricular valve at their primary operation had a suggested lower need for reoperation (RR, 0.5; 95% CI, 0.3-1.1; P = .068).
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, B ). Patients who underwent operation and who were older than 20 years showed a lower risk of left atrioventricular valve regurgitation (RR, 0.6; 95% CI, 0.4-0.9; P = .01). Of the 31 patients who underwent reoperation after diagnosis of left atrioventricular valve regurgitation, 25 patients had some type of atrioventricular valve repair during reoperation, including atrioventricular valve replacement (11 patients), atrioventricular valvuloplasty (6 patients), atrioventricular valve annuloplasty (6 patients), and closure of atrioventricular valve cleft (4 patients). Some patients had combinations of these procedures during their first reoperation.
Left atrioventricular valve stenosis
Left atrioventricular valve stenosis was identified in 19 patients (6%) after operation. For 15 of these 19 patients, the mean transvalvular gradient was 7 mm Hg (Ql, 4 mm Hg; Q3, 10 mm Hg). A complete cleft, partial cleft, or parachute valve was present in 13, 3, and 1 patients, respectively. In 2 patients the anatomy was unclear. Three of these patients were identified after their first reoperation and were censored at reoperation. Of the remaining 16 patients in whom the diagnosis of left atrioventricular valve stenosis was made before any reoperation, the median interval from their primary operation to the diagnosis of left atrioventricular valve stenosis was 7.3 days (Q1, 3.6 days; Q3, 4.1 days). The estimated incidence of diagnosis of left atrioventricular valve stenosis was 3% 30 days after reoperation, 4% at 5 years, 5% at 10 years, 5% at 20 years, and 6% at 30 years. After we controlled for the decade of the operation, increased age at operation was associated with a greater occurrence of left atrioventricular valve stenosis. Specifically, patients more than 40 years of age who had undergone primary repair demonstrated an increased risk for the development of left atrioventricular valve stenosis (RR, 4.26; 95% CI, 1.5-12.4; P = .01).
Three patients underwent left atrioventricular valve replacement after the diagnosis of left atrioventricular valve stenosis.
Left atrioventricular valve regurgitation or stenosis
There were 228 patients who were diagnosed with either left atrioventricular valve regurgitation or left atrioventricular valve stenosis before any reoperation. The estimated incidence of left atrioventricular valve regurgitation or stenosis was 36% 30 days after the operation, 43% at 1 year, 52% at 5 years, 58% at 10 years, 67% at 20 years, 75% at 30 years, and 89% at 40 years.
Left ventricular outflow tract obstruction
Left ventricular outflow tract obstruction was identified in 36 patients (11%). In 4 of these patients the diagnosis of obstruction was made after their first reoperation, and they were censored at reoperation. Of the 32 patients with obstruction before reoperation, the median interval from primary operation to diagnosis of obstruction was 4.9 years (Q1, 0.18 years; Q3, 15.88 years). The estimated incidence for diagnosis of obstruction was 3% 30 days after operation, 6% at 5 years, 7% at 10 years, 10% at 20 years, 13% at 30 years, and 16% at 40 years. After we controlled for the decade of the operation, there were no variables associated with the occurrence of left ventricular outflow obstruction.
Of the 32 patients who were diagnosed with left ventricular outflow tract obstruction subsequent to their primary operation, 10 patients underwent reoperation. Seven of these 10 patients received left ventricular outflow tract reconstruction at reoperation. Of these 7, 6 patients underwent reoperation at the Mayo Clinic, and details of the anatomy of and the relief of the obstruction are available. All 6 patients had a subaortic fibrous (5 patients) or fibromuscular (1 patient) ring. Three of the 6 patients also had septal hypertrophy that contributed to the obstruction. One patient had vegetation on the noncoronary cusp of the aortic valve and obstruction at the midmuscular level because of the anomalous location of the anterior papillary muscle of the left atrioventricular valve. All 6 underwent resection of the subaortic membrane and septal myectomy. In addition, 1 patient underwent excision of some papillary muscle tissue. One year after the reoperation, 1 patient required a modified Konno procedure for persistent/recurrent left ventricular outflow tract obstruction.
Residual or recurrent ASD
Residual or recurrent ASD was identified in 12 patients during follow-up. Seven of these patients had closure of the ASD during reoperation, and 5 patients had no surgical intervention. The median interval from primary surgery to the diagnosis of residual or recurrent ASD was 10.6 years (Q1, 2.8 years; Q2, 22.6 years).
Arrhythmia
Early postoperative arrhythmias (<30 days) were identified in 37 patients. These arrhythmias included paroxysmal supraventricular tachycardia (18 patients), atrial fibrillation (7 patients), atrial flutter (7 patients), complete atrioventricular dissociation (5 patients), and premature atrial (1 patient) and ventricular contractions (1 patient). Some patients may have experienced more than one type of arrhythmia within 30 days of the primary operation.
Arrhythmias occurring more than 30 days after operation that required treatment developed in 46 patients (14%). These arrhythmias included paroxysmal supraventricular tachycardia (12 patients), atrial fibrillation (21 patients), atrial flutter (6 patients), complete atrioventricular dissociation (4 patients), premature atrial contractions (1 patient), and ventricular contractions (2 patients). Some patients may have experienced more than one type of arrhythmia. The median interval from primary repair to the onset of arrhythmia (long-term) was 11.1 years (Q1, 6.7 years; Q3, 23.9 years; Table III). The occurrence of arrhythmias increased with increasing age at primary operation (RR, 1.05; 95% CI, 1.03-1.07; P = .0001).
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Complete atrioventricular block occurred in 9 patients (2.7%), 5 patients after operation and 4 patients late. There were no cases of complete atrioventricular block in patients who had primary repair after 1979. Permanent pacemakers were implanted in 11 patients between 1967 and 1997, whose primary repairs occurred before 1987.
Bacterial endocarditis
Bacterial endocarditis was documented in 7 patients (2%). The median interval from the primary operation to this diagnosis was 8 years (Q1, 0.26 years; Q3, 23 years; range, 0.16-27 years).
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Discussion |
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TopAbstractIntroductionPatients and methods Results Discussion Conclusions References |
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7 found only 8 patients older than 30 years of age, suggesting an important effect of this lesion on long-term survival. Somerville 11 reviewed the factors responsible for the disability and death in 27 of 122 patients with partial ASD who were not treated surgically. Arrhythmia was the most common cause of deterioration and occurred in 25% by age 30 years and in 80% by age 45 years. Arrhythmia or atrioventricular block was responsible for 11 of 14 deaths that occurred after age 30 years. Except for rare cases in which the ASD is quite small, this defect should be repaired. 5,6
Survival
Hospital or 30-day mortalities have ranged from 5.5% to 10%. In the current study, it was 3%. The variables associated with operative or 30-day deaths have been inconsistent from study to study.
Losay and colleagues 12 reported 5- and 10-year survival of 86% and 81%, respectively, for a cohort of 92 patients who underwent operation between 1955 and 1975. Burke and colleagues, 14 studying 33 patients who underwent operation between 1972 and 1994, reported a 5-year survival of 89%. These investigators did not analyze the determinants of late death.
In the current study, the long-term survival at 5, 10, 20, and 40 years were 94%, 93%, 87%, and 76%, respectively. Age less than 20 years at operation and suture approximation of the left atrioventricular valve cleft were associated with better long-term survival. It is possible that, for those patients in whom the cleft was not sutured, there were associated anatomic abnormalities that precluded suture closure. Hence for patients in whom the cleft could not be closed, increased surveillance with the expectation of increased risk of late morbidity and death seems appropriate.
Reoperation
Reoperation was necessary for 11% of the patients. Others have reported reoperation rates of 8.6%, 12 7.6%, 13 6%, 14 and 22.6%. 15 These studies, however, had shorter follow-up times than the present study. Considering the spectrum of abnormalities of the left atrioventricular valve in this condition, it is not surprising that left atrioventricular valve regurgitation was the most common reason for reoperation. Although we found no predictors of reoperation, there was a suggestion that failure to suture the left atrioventricular valve cleft at the original operation was predictive of reoperation.
Left atrioventricular valve stenosis and regurgitation
Assessing the presence, absence, degree, and importance of left atrioventricular valve regurgitation in a study spanning 40 years is difficult because of the changes in technology of assessment of left atrioventricular valve regurgitation. With the advent of Doppler echocardiographic technology, it has become easier to detect left atrioventricular valve regurgitation. Hence it is impossible to know whether the presence, absence, degree, and importance of left atrioventricular valve regurgitation have changed over the years with changes in patient selection for operation, timing of operation, and operative techniques. Although we found a progressively higher occurrence of left atrioventricular valve regurgitation with an increasing length of follow-up, this simply may reflect greater sensitivity of the techniques (Doppler echocardiography) available to measure regurgitation. We did find that patients who had initial repair at more than 20 years of age had a lower incidence of postoperative left atrioventricular valve regurgitation than those whose initial repair occurred at less than 20 years of age. This probably reflects the fact that patients who undergo operation at younger ages have more severe malformation of the left atrioventricular valve than patients who reach an older age before operation. Alternatively, it may reflect a more aggressive approach to the elimination of the valve regurgitation in older patients. Others 15 have observed that the degree of preoperative left atrioventricular valve regurgitation predicts postoperative valve regurgitation.
The rarity of late occurrence of significant left atrioventricular valve stenosis is reassuring in view of past concerns that suture closure of the cleft of the left atrioventricular valve would lead to a high incidence of postoperative stenosis.
Left ventricular outflow tract obstruction
Subaortic stenosis after repair of partial ASD is an uncommon but recognized problem. The reported incidence is 3% to 7%. 15,17-25 We found an incidence of 11%. With the availability of Doppler echocardiographic techniques, left ventricular outflow tract obstruction may be diagnosed more readily than it was before Doppler echocardiographic techniques were available. The presence of left ventricular outflow tract does not always warrant reoperation. Because of the elongated left ventricular tract in patients with endocardial cushion defects, adequate relief of the obstruction may be challenging.
Arrhythmia
It is generally accepted that patients with significant left-to-right shunt from an ASD have an increasing risk of the development of atrial arrhythmias with increasing age. This was true for patients with secundum ASD. 26 In the current study, we also found that the occurrence of supraventricular arrhythmias was related to older age at operation. These data support the argument for younger rather than older age at primary repair.
Complete atrioventricular block is not purely a surgical complication. Six of the 27 patients with ostium primum defect in Somerville’s series 11 manifested clinical deterioration because of the atrioventricular block without undergoing surgical repair.
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Conclusions |
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TopAbstractIntroductionPatients and methods Results Discussion Conclusions References |
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Outcomes studies such as this rely on data acquisition that is dependent on scheduled follow-up visits. This, of course, makes it difficult to know the actual time that a specific event developed and introduces difficulty in timed event analysis.
The following conclusions can be drawn from this study: (1) although long-term survival is good, it is lower than that of the general population; (2) reoperation was necessary for 11% of the patients; (3) postoperative left atrioventricular valve regurgitation is common and is the most frequent reason for reoperation; (4) it is important to close the left atrioventricular valve cleft at primary operation; (5) left ventricular outflow tract obstruction occurred in 36 patients (11%) but required operation in only 7 patients; and (6) postoperative supraventricular arrhythmias are more common for older patients.
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References |
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TopAbstractIntroductionPatients and methods Results Discussion Conclusions References |
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