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Ann Thorac Surg 2005;79:1358-1365
© 2005 The Society of Thoracic Surgeons

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Original articles: Cardiovascular

Operative Results and Outcomes in Children With Shone's Anomaly

^a Section of Cardiothoracic Surgery, James Whitcomb Riley Hospital for Children and Indiana University School of Medicine, Indianapolis, Indiana
^b Section of Pediatric Cardiology, James Whitcomb Riley Hospital for Children and Indiana University School of Medicine, Indianapolis, Indiana

Accepted for publication September 7, 2004.

^_* Address reprint requests to Dr Brown, Section of Cardiothoracic Surgery, Indiana University School of Medicine, 545 Barnhill Dr, EH 215, Indianapolis, IN 46202-5123 (E-mail: jobrown{at}iupui.edu).

Presented at the Poster Session of the Fortieth Annual Meeting of The Society of Thoracic Surgeons, San Antonio, TX, Jan 26–28, 2004.

	Abstract

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BACKGROUND: The outcome of children with multilevel left heart obstructions with mitral valve involvement (Shone's anomaly) is generally poor. This study reviews our results and outcomes in patients with staged repair for left ventricular outflow tract obstruction (LVOTO) and mitral valve reconstruction.

METHODS: A total of 27 children underwent staged repair for Shone's anomaly between 1978 and 2003. The mean age at the first operation was 9.0 months (range, 2 days to 3 years). Mitral stenosis was present in all; with supravalvular mitral ring (n = 11), mitral valve abnormalities including parachute mitral valve, fused chordae, single papillary muscle (n = 10), and "typical" (Ruckman & Van Praagh) congenital mitral stenosis (n = 14). The LVOT obstruction features included subaortic stenosis (n = 16), valvar aortic stenosis (n = 7), bicuspid aortic valve (n = 24), and coarctation (n = 18). All 27 patients underwent 94 surgical procedures, including 22 mitral operations and 48 LVOT operations.

RESULTS: There were no operative deaths at the first operation. Mean follow-up was 4.7 ± 4.4 years (range, 6 months to 15 years). There were three late deaths (11%). All late deaths were secondary to severe mitral valve disease. Overall 15-year actuarial survival was 89%. All surviving patients are in New York Heart Association functional class I or II.

CONCLUSIONS: Patients with Shone's anomaly are seen with a wide spectrum of major anatomic and hemodynamic abnormalities. A favorable outcome is possible for most. Operative mortality is adversely affected by the severity of mitral valve disease, the degree of left ventricular hypoplasia, and the need for multiple operative procedures.

	Introduction

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In 1963 Shone and colleagues [1] described a syndrome consisting of multilevel obstruction of all left heart structures. The original features of Shone's complex include a parachute mitral valve, supraannular mitral ring, subaortic stenosis, and coarctation of the aorta. In practice, the management of patients born with multiple left heart obstructive or hypoplastic lesions is complex because of a broad spectrum anatomy, ranging from mild lesions that may not require any intervention to severely obstructive or hypoplastic left heart anatomy that necessitates single-ventricle (Norwood/Fontan) palliation. These patients are considered to be a high risk subgroup [2–5]. Limited data are currently available concerning patients with Shone's syndrome and therefore we reviewed our experience in 27 children with emphasis on operative results and outcome in this anomaly.

	Material and Methods

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Patients
Between September 1978 and August 2003, 27 consecutive children with congenital multilevel left heart obstructive lesions and mitral valve anomalies underwent operation at the James Whitcomb Riley Hospital for Children in Indianapolis. After obtaining approval from our Institutional Review Board, available medical records were reviewed, including preoperative invasive and noninvasive evaluations, and operative and inpatient reports. Outpatient and referring physician reports including echocardiographic data were utilized for follow-up. There were 14 female and 13 male patients. The mean age at the first operation was 9.0 ± 11.6 months (range, 2 days to 3 years). Seventeen patients (17 of 27, 63%) were younger than 6 months of age at the first surgical procedure. Preoperatively, the majority of patients (19 of 27, 70%) were in New York Heart Association functional class III or IV with symptoms of heart failure. Patients with significant left heart hypoplasia, who were not suitable for a two ventricular repair and who had two or more additional pathologies (straddling of tricuspid or mitral valves, asymmetrical atrioventricular septal defect, multiple ventricular septal defects, unroofed coronary sinus, double outlet right ventricle, and nonapex left ventricle), were stratified to Norwood-type single-ventricle palliation and were excluded.

Anatomical evaluation was done in all patients by echocardiography or cardiac catheterization and confirmed at operation. All patients had abnormal mitral valves, and with at least two other left heart obstructive lesions were included in this study. The level of obstruction for each patient is shown in Table 1. Not all of the left heart obstructive lesions were severe enough that they required any interventions by the time of this report.

The most prevalent left sided obstructive lesion was bicuspid aortic valve which was present in 24 of 27 patients (89%) and associated valvar aortic stenosis which was noted in 7 of these patients (7 of 27, 26%). A mildly hypoplastic transverse aortic arch (diameter > 4 mm) was present in 6 patients (6 of 27, 22%), mildly hypoplastic left ventricle in 4 patients (4 of 27, 15%), and supravalvular aortic obstruction in 3 patients (3 of 27, 11%).

Additional obstructive lesions included coarctation of the aorta in 67% of patients (18 of 27), diffuse subaortic stenosis due to a fibromuscular tunnel in 10 patients (10 of 27, 37%), and a subvalvular discrete membrane in 6 patients (6 of 27, 22%). Systolic gradients at the area of subaortic obstruction ranged from 10 to 120 mm Hg (mean, 48 ± 28) and coarctation gradients ranged from 10 to 65 mm Hg (mean, 42 ± 15). Left ventricular outflow tract obstruction was less likely to produce pulmonary hypertension in these patients. Only 3 patients presented with congestive heart failure and only one had significant pulmonary hypertension.

The most common associated cardiac anomaly was ventricular septal defect (VSD; 12 of 27, 44%). Additional cardiac anomalies included atrial septal defect (ASD; 9 of 27, 33%), patent ductus arteriosus (PDA; 9 of 27, 33%), vascular ring (1 of 27, 4%), and infundibular right ventricular outflow tract obstruction (1 of 27, 4%). Symptoms of infection were present in 13 patients (48%) and 7 of 13 presented with significant pulmonary hypertension (pulmonary to systemic flow ratio [Qp/Qs] > 1.5).

Operative Procedures
Twenty-seven patients underwent a total of 54 operative procedures to repair 94 lesions (Table 2). At the first operation in 27 patients there were 47 lesions corrected, an average of 1.7 per patient. The most common procedure was coarctation repair (n = 15) and PDA ligation (n = 9). Additional procedures performed at first operation included VSD closure (n = 4), supramitral ring resection (n = 3), fibrous subaortic resection (n = 2), balloon aortic valvotomy (n = 2), and surgical mitral valve dilatation (n = 2). One patient each had closed transventricular aortic valvotomy, open aortic valvotomy, mitral valvuloplasty, limited myotomy and myectomy, mitral valve replacement (MVR), ASD closure, division of anomalous left subclavian artery, pulmonary artery banding, and pulmonary arterioplasty.

A third operation has thus far been required in 6 patients (10 lesions repaired) at a mean age of 4.4 ± 5.3 years (range, 3 months to 14 years). Procedures included resection of fibrous subaortic stenosis (n = 3), MVR (n = 2), and balloon aortic valvotomy, open aortic valvotomy, balloon angioplasty of coarctation, mitral commissurotomy, and supravalvar single patch aortoplasty (1 each).

Three patients have thus far required a fourth operation at 1.5, 9, and 20 years of age. These procedures included resection of supraannular mitral ring (n = 1), Ross procedure (n = 1), and redo MVR using a pulmonary autograft (Ross II, n = 1). One patient has thus far required a fifth operation, a Ross-Konno procedure at 8 years of age.

Seventeen patients (17 of 27, 63%) have had one to three operations (22 procedures) on the left ventricular inflow (Table 3). The remaining 10 patients with mitral disease have not yet required intervention during the study period because of milder degree of obstruction. Average age at the time of first mitral valve operation was 3.4 years (range, 3 months to 18 years). The reasons for MVR in 5 patients included early valve failure (n = 1) or valve morphology that was not suitable for repair (n = 4). All five initial valve replacements were performed before the age of 2.5 years using St Jude bileaflet prosthetic valves (St Jude Medical Inc, St Paul, MN), sized 19 mm (n = 2), or CarboMedics (CarboMedics, Inc, Austin, TX), sized 16 mm (n = 3). The prosthesis was inserted in supraannular position in three patients. Replacement of the initial mitral prosthesis was required in 2 patients, with a 21 mm St Jude prosthesis (n = 1) and with a pulmonary autograft MVR (Ross II, n = 1).

	Results

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Mortality and Morbidity
There were no operative deaths associated with initial operation. All three deaths (3 of 27, 11%) occurred late, after second (n = 1) or third operations (n = 2). Overall survival estimated by the Kaplan-Meier method was 96% at 5 years, 93% at 10 years, and 89% at 15 years after the initial surgery (Fig 1).

View larger version (13K): [in this window] [in a new window]   Fig 1. Kaplan-Meier estimate of survival in patients with Shone's anomaly.

The second death (patient No. 7) occurred in a child who underwent coarctation repair at 2 years of age and open aortic valvotomy with resection of supraannular mitral ring at age 11 years. He experienced a sudden unexplained death at home at age 14.

The third death (patient No. 20) occurred three months after MVR with a 16 mm CarboMedics prosthesis placed in the supraannular position, resection of fibrous subaortic membrane, open aortic valvotomy, and patch aortoplasty. This patient had coarctation repair as a newborn and balloon angioplasty one year later for recurrent coarctation. Autopsy did not reveal the cause of death, which was likely due to cardiac arrhythmia. Three other patients (Nos. 2, 4, and 10) required implantation of a permanent pacemaker for postoperative complete heart block. One of these patients (patient No. 4) was also a late death. Two late deaths were in children who had been treated for severe mitral valve disease; however, univariate and multivariate analysis identified no risk factors for late death.

Reoperation
A total of seven reoperations were performed in five patients. Three patients had one and the remaining two patients underwent two reoperations.

Surgical coarctation repair (n = 16) effectively reduced the gradient across the aortic arch from 42 ± 6 mm Hg to 9 ± 4 mm Hg (p = 0.005). However, residual gradients of 15 to 28 mm Hg developed postoperatively in 4 patients (25%). Three of these patients had severe recoarctation within 6 months to 13 years after a subclavian flap aortoplasty (3 of 9, 33%) versus an end-to-end repair (0 of 6, 0%, p = 0.002). Balloon angioplasty was successful for recoarctation in two patients and surgical patch angioplasty was performed in one patient.

There were two residual obstructions (2 of 10, 20%) after transaortic resection of discrete subaortic stenosis. One patient required septal myotomy (3 years after initial surgery) and a second patient underwent a Ross-Konno procedure (7 years after initial surgery).

There have been no recurrences of residual supraannular mitral rings. Two patients who required mitral valve replacement needed subsequent replacements. The first patient (patient No. 16), who required a 16 mm CarboMedics prosthesis, needed reoperation and the insertion of a 21 mm St. Jude Medical prosthesis 4.5 years later due to prosthetic mitral stenosis (24 mm gradient). The second patient (No. 21) underwent MVR at age four, with a 19 mm St. Jude Medical valve, and 16 years later MVR was performed due to mitral stenosis (13 mm gradient) using a pulmonary autograft (Ross II). This patient has done well with less than a 3 mm valve gradient, and no regurgitation. Overall freedom from reoperation estimated by the Kaplan-Meier method was 93% at 1 year, 85% at 5 years, 82% at 10 years, and 77% at 15 years after the initial surgery (Fig 2).

View larger version (16K): [in this window] [in a new window]   Fig 2. Kaplan-Meier estimate of freedom from reoperation in patients with Shone's anomaly.

The recent postoperative echocardiographic study demonstrates that moderate hemodynamic abnormalities (pulmonary hypertension, moderate mitral stenosis or regurgitation, moderate subvalvar or valvar aortic stenosis, recoarctation of aorta, moderate LV dysfunction) are present in 7 of 23 children (30%), whereas the remaining 16 patients are considered free of significant obstruction (70% of all patients). Two of 7 with moderate obstruction await reoperation for multilevel aortic stenosis, whereas the other 5 are being followed conservatively.

	Comment

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Forty years ago, Shone and colleagues [1] described the developmental complexity of parachute mitral valve, supravalvar mitral ring, subaortic stenosis, and coarctation of aorta. In his original article (8 patients), only 2 patients exhibited all four aspects of Shone's complex and could therefore be considered to have classic Shone's complex. The remaining 6 patients can be regarded as having forme fruste of Shone's anomaly, usually with the absence of the supraannular mitral ring. These anomalies tend to coexist, but the severity and predominance of each individual lesion may vary making optimal management strategy challenging. In clinical practice, the definition of Shone's anomaly has been broadened beyond the original description to include patients with additional forms of left heart pathology such as supravalvular and tunnel aortic stenosis.

Understanding the morphology of the mitral valve in Shone's anomaly is critical to determine the reconstructive surgical approach. Parachute mitral valve and supravalvular mitral ring are the most prevalent variants of mitral stenosis in this disease and are rarely amenable to balloon angioplasty [3, 8]. Supraannular fibrous ring, a characteristic feature of Shone's anomaly, rarely occurs as an isolated lesion [9, 10] and is not usually severely obstructive [6]. Resection is straightforward and although recurrence has been described in one report [11], our experience (11 of 27, 41%) and that of others [2, 3, 8] show that recurrence is rare.

The morphologic findings in the present study conform to the spectrum of congenital mitral valve abnormalities encompassed by the classic autopsy studies of Ruckman and Van Praagh [6], Rosenquist [12], Shone and colleagues [1], and Oosthoek and colleagues [13]. In our experience, the most common mitral anomaly in patients was congenital mitral stenosis, consisting of leaflet dysplasia, shortened and thickened chordae tendineae, obliteration of interchordal spaces, and underdevelopment of papillary muscles [6]. In this condition, separation or fenestration of chordae may postpone or even eliminate the need for valve replacement. Brauner and colleagues [3] found "typical" congenital mitral stenosis in 25% of their patients with Shone's anomaly (Table 6). Parachute mitral valve was present in 10 of our patients. In parachute mitral valve all chordae are usually shortened and thickened, and attached to the posterior medial papillary muscle. The anterolateral papillary muscle is often absent. Rarely, both papillary muscles are present with chordal attachments to one. Oosthoek and colleagues [13] called this malformation "parachutelike asymmetric mitral valve." They observed 29 parachutelike valves from 129 autopsy specimens in patients with Shone's syndrome and only one heart had a true parachute mitral valve (ie, one single papillary muscle receiving all chordae).

Recent advances in operative technique and the routine use of intraoperative transesophageal echocardiography and not replacement has significantly contributed to the success of valve reconstruction as the initial treatment for congenital mitral stenosis. Reports suggest that late outcomes of valve repair are superior to replacement in patients with isolated congenital mitral anomalies [3, 14, 15]. Successful repair will permit continued annular growth and delay or eliminate the need for future valve replacement and lifelong anticoagulation. In this series and others (Table 6), early valve replacement frequently requires subsequent reoperation to implant a larger prosthesis [2, 8, 15]. Currently, we believe that many such lesions are amenable to repair rather than replacement, chiefly because the main obstructive mitral element in this condition is subvalvar. The availability of smaller mechanical prostheses, the pulmonary autograft procedure (Ross II), and the aortic homograft MVR (Ross/Yacoub) have provided effective alternatives in the event of failed repair and have made MVR a viable option even in smaller patients. In the presence of pulmonary hypertension, operation should not be delayed even in young children as elevated pulmonary artery pressures adversely affect outcome [2, 16].

The majority of patients in our series (18 of 27, 67%) presented initially in the neonatal period with coarctation of aorta as the predominant outflow obstructive lesion [5, 17]. Coarctation may mask the presence and potential hemodynamic severity of associated intracardiac lesions, and indeed the diagnosis of Shone's anomaly in several patients in our series was made when evaluating symptoms of heart failure after successful coarctation repair. In the setting of neonatal Shone's anomaly, an aggressive form of aortic coarctation has been observed with early presentation and severe symptoms which can mask the intracardiac pathology until coarctation is repaired [2, 3]. We also observed a higher reoccurrence rate after repair of coarctation in patients with Shone's syndrome, especially if subclavian flap aortoplasty was performed (3 of 9, 33%). Rosenquist [12] observed normal great vessels in 53 heart specimens with coarctation of the aorta and high incidence of obstructive mitral valve pathology ranging from mild annular stenosis to parachute mitral stenosis.

The incidences of LVOT obstruction in our series were subvalvar 49%, valvar 26%, or supravalvar 11%. Subaortic stenotic lesions were rarely present at birth, however, they can develop later and become rapidly progressive [18, 19]. Discrete membranous lesions can be effectively treated by transaortic resection, which should include ventricular septal myomectomy that has been demonstrated to prevent recurrence. The incidence of recurrent subaortic stenosis after resection was observed in 10% of our series, which is considerably lower than published in other reports [18–21]. In patients with diffuse tunnel subaortic stenosis, a different surgical approach is needed to provide long-term relief of LVOT obstruction. We and others have found that the pulmonary autograft AVR (Ross), in conjunction with the Konno procedure, is a highly effective operation for tunnel aortic stenosis and has the advantage of potential growth for the aortic root without the need for anticoagulation [22–24]. This technique has emerged as the optimal repair of tunnel LVOT obstructive lesions in children with Shone's anomaly.

Due to the problems inherent with a single-ventricle palliation, including the usual need for multiple staged procedures and concerns regarding the long-term prognosis of a systemic right ventricle, a biventricular repair is thought to be preferable when possible [5]. Decisions regarding surgical management often must be made in the newborn period. Rhodes and colleagues [25] delineated a score to identify candidates for single-ventricle or biventricular repair for patients suffering a congenital critical aortic stenosis. Our operative experience with patients having Shone's complex compares favorably to that reported by other centers [2–5].

In conclusion, the management of patients with Shone's anomaly has to be tailored to the pathologic anatomy of the presenting patient. In general, we treat first the most distal obstructive lesion, which is usually coarctation. We have demonstrated that an aggressive reconstructive approach to the mitral valve and the left ventricular outflow tract can further extend event-free survival in these children. The outcomes in such patients are related to the degree to which mitral stenosis can be relieved.

	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): John W. Brown Mark Ruzmetov Mark D. Rodefeld Mark W. Turrentine Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Brown, J. W. Articles by Turrentine, M. W. Search for Related Content PubMed PubMed Citation Articles by Brown, J. W. Articles by Turrentine, M. W. Related Collections Congenital - acyanotic