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Ann Thorac Surg 2007;84:1357-1363
© 2007 The Society of Thoracic Surgeons

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Original Articles: Cardiovascular

Surgical Strategies and Outcomes in Patients With Shone Complex: A Retrospective Review

The Children’s Medical Center at the Medical College of Georgia, Augusta, Georgia

Accepted for publication May 1, 2007.

^_* Address correspondence to Dr St. Louis, Medical College of Georgia, 1120 15th St, BA-4300, Augusta, GA 30912-4040 (Email: jstlouis{at}mcg.edu).

Presented at the Fifty-third Annual Meeting of the Southern Thoracic Surgical Association, Tucson, AZ, Nov 8–11, 2006.

	Abstract

Top Abstract Introduction Patients and Methods Operative Procedures Results Comment Discussion References

 
Background: Multilevel obstruction of left-sided heart structures was originally characterized by Shone and colleagues. The formulation of an appropriate operative strategy remains problematic. Surgical outcomes have generally been poor. This review examines operative strategies and mid-term outcomes.

Methods: A retrospective review was done of 28 patients who presented with the diagnosis of Shone complex at a median age of 14 days, with 46% presenting within the first week of life. At presentation, 5 patients were in cardiovascular collapse, and 10 were at New York Heart Association functional class III or IV. All patients had a structurally abnormal mitral complex. A mean transvalvular pressure gradient of 8 mm or greater existed in 15 patients, 6 had a supraannular mitral ring, and 26 had a coarctation of the aorta. Twelve mitral valve operations were done, with an average time from presentation to operative intervention of 15 months.

Results: Two patients were lost to follow-up. Of 26 remaining patients, 2 died after a second operative intervention. The average follow-up of 24 patients was 6.3 years (range, 1 to 16 years). Overall survival was 93%. All surviving patients are in class I or II congestive heart failure.

Conclusions: Long-term survival of patients diagnosed with Shone complex is excellent. Operative strategies for this complex group should be individualized. Mitral interventions may generally be deferred.

	Introduction

Top Abstract Introduction Patients and Methods Operative Procedures Results Comment Discussion References

 
Multilevel obstruction of left heart structures was initially described by Shone and colleagues [1]. Their original description included four defects of the systemic circulation: a supraannular mitral ring, subaortic stenosis, a parachute mitral valve deformity, and a coarctation of the aorta. In this series, only 2 patients presented with all four defects. It is this variability in presentation and severity of individual lesions that has made formulation of appropriate management strategies problematic. Outcomes from early aggressive surgical repair have historically been poor [2, 3]. Recent studies have shown improved operative survival in patients treated with an initial conservative approach to the mitral valve deformities [4]. Our group identified 28 patients that presented with diagnosis of Shone’s complex, with emphasis on presentation, surgical strategy, and mid-term outcomes.

	Patients and Methods

Top Abstract Introduction Patients and Methods Operative Procedures Results Comment Discussion References

 
We retrospectively reviewed 28 patients (20 boys, 8 girls) who presented with the diagnosis of Shone complex between 1988 and 2004. Our Institutional Review Board approved this study with a waiver of informed consent. Admission notes, echocardiography reports, operative notes, discharge summaries, and clinic notes were reviewed. The median age at initial presentation was 14 days (range, 1 day to 5 years), with 46% of patients presenting within 1 week of birth. The average weight at presentation was 5.1 ± 4.8 kg (range, 2.7 to 17 kg), and 63% weighed less than 3.5 kg. Severe hemodynamic instability was present in 5 patients, and 10 were in New York Heart Association (NYHA) functional class III or IV heart failure.

The review excluded patients who were not suitable for a two-ventricle repair and those diagnosed with hypertrophic cardiomyopathy, double outlet right ventricle, a partial atrioventricular canal defect, or isolated mitral valve disease.

The anatomy at presentation is summarized in Table 1. All patients had a structurally abnormal mitral valve complex; of these, 10 (36%) had the diagnosis of typical congenital mitral valve stenosis (TCMS) [5], and 18 (64%) had a parachute mitral valve deformity [6]. A mean pressure gradient across the mitral valve of 8 mm Hg or greater was considered significant [4] and existed in 15 (54%) patients at a range of 8 to 15 mm Hg. A supraannular mitral ring was present in 6 patients (22%).

The most common associated cardiac lesion was an atrial septal defect, which occurred in 11 patients (39%), and 10 (36%) had a ventricular septal defect. Other cardiac lesions included patent ductus arteriosus (n = 5), aberrant right subclavian artery (n = 2), and a left superior vena cava (n = 2).

	Operative Procedures

Top Abstract Introduction Patients and Methods Operative Procedures Results Comment Discussion References

 
An operative intervention was done in 27 of the 28 patients at the initial presentation. The operative procedures are summarized in Table 2. In 21 patients (78%), isolated repair of a coarctation was done with an extended end-to-end anastomosis (n = 12, 57%), a subclavian flap aortoplasty (n = 8, 38%), or a carotid turndown procedure (n = 1, 5%) Three patients with severe proximal transverse arch hypoplasia underwent patch augmentation combined with excision of a coarctation through a median sternotomy. One of these two patients underwent an emergency balloon aortic valvuloplasty before arch augmentation. One patient underwent an emergency open aortic valve commissurotomy for critical aortic stenosis.

A second operation has been performed on 17 (63%) of 27 patients to date, for a total of 34 procedures (Table 2). The mean age at this second operation was 1.3 ± 1.48 years (range, 1 month to 6 years). A mitral valve operation was done in 8 patients, with an average time from presentation to operative intervention of 1.26 ± 0.92 years (range, 3 months to 3 years; Table 3). Of these 8 patients, 4 underwent an initial mitral valve replacement. The average time from presentation to valve replacement was 1.9 ± 0.86 years (range, 11 months to 3 years). The average weight at the time of replacement was 9.0 ± 3.4 kg. These 4 patients had undergone an initial repair of a coarctation, at which time the average mean transvalvular pressure gradient across the mitral valve was 5.5 ± 2.1 mm Hg (range, 3 to 8 mm Hg).

Four patients underwent a mitral repair without a prior mitral intervention. Three patients had undergone an initial repair of a coarctation. The average time from presentation to valve repair was 7.5 ± 4.6 months (range, 4 to 14 months). The average weight at the time of repair was 6.14 ± 2.4 kg. All had a parachute deformity of the mitral valve, with an average mean transvalvular pressure gradient of 9.75 ± 3.5 mm Hg (range, 8 to 15 mm Hg). The average mean transvalvular pressure gradient in these 3 patients at the time of repair was 12.5 ± 2.1 mm Hg (range, 10 to 15 mm Hg). All underwent splitting of a single papillary muscle and separation of fused chordae tendineae. One patient required replacement of the mitral valve with a mechanical prosthesis in the immediate postoperative period. The most recent mean transvalvular pressure gradient in the remaining three patients is 2.2 ± 2.0 mm Hg (range, 0 to 4 mm Hg). One patient with moderate mitral regurgitation is currently being followed up. Four patients underwent excision of a supramitral ring. The average age at the time of resection was 3.9 ± 5.4 years (range, 4 months to 12 years).

Five patients underwent closure of a ventricular septal defect. A subaortic membrane was excised in 3 patients. In 2 patients, a left ventricular apex–to–descending aortic conduit was placed for severe tunnel stenosis of the left ventricular outflow tract. The patient who had undergone emergency placement of a left ventricular apex–to–descending aortic conduit underwent a successful Ross procedure and takedown of an left ventricular–apex–aortic conduit. Two patients underwent repair of a recoarctation with a patch aortoplasty through a left thoracotomy. One recurrence occurred after a homograft augmentation of the transverse arch and the other occurred after a radical extended end-to-end reconstruction.

A third operation has thus far been required in 3 patients (6 procedures). One patient underwent replacement of mitral valve prosthesis (17-mm to 21-mm St. Jude mechanical prosthesis) in addition to a Konno (St. Jude aortic valve) operation and takedown of a left ventricular apex–to–descending aortic conduit. Another patient only required replacement of a mitral valve prosthesis (19-mm to 23-mm St. Jude mechanical prosthesis). The final patient underwent a mitral prosthesis replacement (19-mm to 21-mm St. Jude mechanical prosthesis) combined with an open aortic valvotomy. Their average age at the time of these three mitral prosthesis replacements was 6.7 ± 3.1 years, with an average patient weight of 19.3 ± 9.5 kg. The average mean transprosthesis pressure gradient was 18.3 ± 5.9 mm Hg.

Statistical Analysis
Univariate and multivariate analysis of the following risk factors for survival and need for an initial mitral valve intervention included initial mitral valve gradient, mitral valve pathology, age, weight, associated cardiac anomalies, hemodynamic presentation, and left ventricular outflow tract pathology. For categoric variables, contingency tables were constructed, and ² and Fisher exact tests were used to test for significance. For continuous variables, the Student t test was used to test for significance. The Kaplan-Meier product limit method was used for analysis of freedom from reoperation. Data are expressed as mean, standard deviation from the mean, and range. A value of p 0.05 was considered significant.

	Results

Top Abstract Introduction Patients and Methods Operative Procedures Results Comment Discussion References

 
Overall, two deaths (12%) occurred in 17 patients who had the second operation, for a overall in-hospital survival of 93%. The first death occurred in a 5-month-old male who had undergone repair of a coarctation as a newborn. At the time of coarctation repair, the mean transmitral pressure gradient was 3 mm Hg and the valve pathology was consistent with TCMS. The child had a bicuspid aortic valve with no gradient and a large perimembranous ventricular septal defect. The patient underwent closure of the ventricular septal defect at 5 months. The mean transmitral pressure gradient had not changed at the time of surgical repair. The procedure was complicated by Enterococcus sepsis and renal failure, and the patient later died.

The second death occurred in an 11-month-old girl who had undergone an initial repair of a coarctation with a subclavian flap aortoplasty. She presented with a transmitral pressure gradient of 8 mm Hg and pathology consistent with TCMS. The initial surgical repair was complicated by Klebsiella pneumonia. At age 11 months, the mean transmitral pressure gradient had worsened to 11.3 mm Hg. Severe diffuse subaortic fibromuscular tunnel stenosis also developed, and the patient underwent a mitral valve replacement with a 17-mm St. Jude mechanical prosthesis in addition to a left ventricular apex–to–descending aortic conduit. The patient could not be weaned from cardiopulmonary bypass and was placed on extracorporeal membrane oxygenation. Cardiac function did not improve after 10 days of support, and the patient died of biventricular failure.

Of the 21 isolated coarctation repairs during the neonatal period, 2 (9.5%) of 21 patients required an intervention for significant recoarctation. One patient had undergone an extended end-to-end reconstruction, which was complicated by the development of a gradient of 55 mm Hg by age 4 months, and underwent patch augmentation of the recoarctation. The second patient presented with a recurrent gradient of 44 mm Hg at 1 year after a subclavian flap aortoplasty and underwent a successful balloon angioplasty. A severe recoarctation developed at age 3 months in a patient who had undergone an excision of a coarctation and transverse arch reconstruction with a homograft patch. The patient underwent successful patch augmentation through a thoracotomy. Three of the four patients who presented with a discrete subaortic membrane required excision, with no recurrences.

Three patients required placement of a left ventricular apex–to–descending aortic conduit for severe subaortic tunnel stenosis; as described, 1 patient in this group died. The other 2 patients have successfully undergone definitive repair of the subaortic obstruction with either a Ross procedure or Konno operation and takedown of the conduit.

Four patients underwent a valve replacement as their initial intervention on the mitral valve. One patient in this group died and 2 others required replacement of the mechanical prosthesis. Inadequate size with an increasing gradient was the reason for replacement in both cases. One patient was 10 years old, with a 5.7-year interval between mitral operations. The other patient was 4 years old, with a 2.5-year between operative interventions. The remaining patient is doing well, without a significant gradient across the prosthesis.

Three of 4 patients with stenotic parachute deformity successfully underwent a mitral valve repair. All are doing well, without a significant transvalvular gradient. Moderate mitral regurgitation developed in 1 patient, who is presently being monitored. The single failure occurred in a patient who presented with a significant gradient across a parachute deformity of the mitral valve. The operative repair of this lesion successfully addressed the stenosis but caused severe mitral regurgitation that required replacement in the early postoperative period. The child is currently doing well, having undergone a recent replacement of the mitral prosthesis.

Overall freedom from a repeat mitral valve intervention estimated by the Kaplan-Meier method was 87% at 1 year, 70% at 5 years, and 47 % at 10 years after the initial mitral intervention (Fig 1). When analyzed by replacement or repair, the freedom from a mitral valve reintervention at was 31.25% and 75%, respectively.

View larger version (12K): [in this window] [in a new window]   Fig 1. Kaplan-Meier estimate of freedom from reoperation for mitral repair (diamonds) and mitral replacement (squares).

Two (7%) of 28 patients were lost to follow-up, which averaged 5.8 ± 4.7 years (range, 5 months to 16 years). All surviving patient are in NYHA functional class I or II. There are 3 surviving mitral valve replacements (one patient lost to follow-up), with an average patient age of 11.7 years. One of the 4 patients that underwent a mitral valve repair has been lost to follow-up, although this patient required a subsequent mitral valve replacement.

	Comment

Top Abstract Introduction Patients and Methods Operative Procedures Results Comment Discussion References

 
This report represents a single institution’s experience with multilevel congenital defects of the systemic circulation. Shone and colleagues [1] described a constellation of defects involving multilevel obstruction the systemic circulation. Only 2 patients included in their original description had all four anatomic anomalies. Today, the diagnosis of Shone complex has come to signify a variety of left heart pathologies [7]. The management strategy of this complex population is hampered by the variable mode of presentation and differing severity of the individual lesions.

Several recent reviews have emphasized the severity of left ventricular inflow obstruction as it relates to surgical management and operative outcomes [3, 4]. A clear and concise understanding of this pathology is crucial to formulate an appropriate operative strategy. Two predominate pathologic abnormalities of the mitral complex have been described. A parachute deformity was the most prevalent variant of mitral stenosis in our study and other recent reviews [2, 3]. This lesion consists of a single papillary muscle attached to shortened cords. These lesions are rarely amenable to percutaneous dilation, making surgical correction the only option for critical stenosis [8]. Parachute deformities have traditionally been addressed surgically by replacement of the valve, with suboptimal outcomes [9].

TCMS, the other variant, consists of underdeveloped, abnormally positioned papillary muscles with shortened, thickened chordae tendineae. This lesion occurred in a few of our patients, although others have reported that this variant was present in most of their patients [4]. No patients in this review required a mitral valve intervention during the neonatal period. This is consistent with the finding of Brown and colleagues [4], which reported only 1 patient requiring an early mitral intervention.

Eight patients underwent an operative intervention on their mitral valve. Mitral valve replacement was undertaken in half of these patients, with the other half undergoing a mitral repair. A parachute deformity was the most frequent anatomic variant. The freedom from reoperation after the initial replacement was 31% in this patient group. These valves tend to fail during a period of 5 to 10 years [10]. This is consistent with the literature, which has for years highlighted a considerable failure rate with replacement in children [10–12]. In addition, valve replacement is not particularly applicable to infants younger than 1 year old [13].

Mitral valve reconstruction, conversely, allows for growth of the annulus, does not require anticoagulation, and has shown improved surgical outcomes with advances in operative technique [14]. Because of the inherent problems with mechanical mitral valves in children, considerable attention has focused on reconstructive approaches. Reports have suggested that late outcomes of valve repair are superior to replacement in patients with isolated congenital mitral anomalies [15]. This was evident in our patients, in whom there was no mortality and a 75% freedom from reoperation at 10 years. Failure of the repair was early.

Our approach to the valve repair of parachute deformities is similar to that published by Zias and colleagues [16]. We have taken an aggressive approach to these defects, with extensive separation of fused chordae tendineae and precise splitting of the common papillary muscle. Our early technical failure was secondary to an overly aggressive splitting of the common papillary muscle. In addition, these techniques could be applied earlier in those patients in whom replacement would have been difficult. In the case of TCMS, operative repair may be successfully accomplished by splitting of the commissures with separation of the chordae. We are in agreement with others that most lesions of this nature are amenable to repair rather than replacement, chiefly because the main obstructive element in this condition is subvalvular. This approach may postpone or even eliminate the need for valve replacement [17].

Most of the patients in this series presented during the neonatal period with a critical coarctation of the aorta and minimal signs of left ventricular inflow pathology. This is consistent with other series of Shone and colleagues [18] and is of importance since the predominance of outflow tract symptoms may mask other potentially critical inflow lesions. Therefore, it is critical that an aggressive diagnostic evaluation be undertaken for patients that present with a coarctation in the setting of even trivial mitral valve pathology [19].

In conclusion, operative management of patients diagnosed with Shone complex has improved with a clearer understanding of the valvular pathology and expertise with reparative techniques. Obstructive lesions of the left outflow tract obstruction are addressed initially, with operative interventions on the inflow deformities being deferred. Reparative techniques should be applied whenever possible, with replacement being reserved for failure of repair.

	Discussion

Top Abstract Introduction Patients and Methods Operative Procedures Results Comment Discussion References

 
DR JOSEPH FORBESS (Dallas, TX): Jim, that is a wonderful series that you have presented with very admirable results, and I think it is highly likely that it is a combination of great precision both in the operating room and before you get to the operating room as far as patient selection. I would actually like to explore with you how you define the boundaries as to who goes to a two-ventricle repair and thus is included in this series. As you know, on the milder end of the spectrum of hypoplastic left heart syndrome, one sees those patients with mitral stenosis, aortic stenosis, coarctation, and then variable degrees of left ventricular hypoplasia. My first question is, how do you select the patients to take them to two ventricles versus one ventricle?

DR ST. LOUIS: That is a great question, Joe. These patients certainly can fall into the gray area between going down the one-ventricle or two-ventricle pathway, and we have made mistakes in trying to put patients on a two-ventricle pathway thinking their mitral capacity was adequate enough. What we have done and we have adopted is in these patients that fall in the gray areas using the calculator that was published by Laughlin in the Congenital Heart Surgeons’ Society to calculate which pathway is best for these patients depending on mitral valve annulus size, aortic annulus size, age, and that is how we have taken these patients.

DR FORBESS: Secondly, I wanted to ask about the timing of intervention on mitral pathology, because I think you, as most surgeons and programs, when going two-ventricle route, have not intervened on the mitral valve in the neonatal period. We follow these children along, but I admit I don’t exactly know what are the criteria for mitral valve intervention. What is your opinion of transmitral gradient, symptoms, or the right-sided pressures as trigger points? I personally have followed kids for numbers of years just looking at their right-sided pressures, suspecting that they are "safe" if their right-sided pressures are relatively low and they are relatively asymptomatic. What is your strategy with regard to the timing of mitral intervention.

DR ST. LOUIS: Certainly, there is a shift in thought as we become better and better in mitral valve repair. As you saw in the series, mitral valve replacement was done later and later, and although I didn’t show the data, those right-sided pressures you alluded to were higher and remained higher afterwards.

The repairs were done earlier. We found ourselves better able to deal with patients with mild-to-moderate elevations, again, in right-sided pressures, and I didn’t include that data because it was inconsistent in the data set. But we follow them based on, as you said, right-sided pressures, symptomatology, and just when the child starts looking to the point where he needs something done.

DR FORBESS: As my final question, I would like to ask about those right-sided pressures and the pulmonary vascular resistance of these patients long-term. I have participated in transplant meetings where teenagers who were taken the two-ventricle route, with Shone syndrome, are presented. They have lost their exercise tolerance, and they have lived their entire life with a very noncompliant left ventricle. Their pulmonary vascular resistance can be elevated to the point that it can sometimes jeopardize their candidacy for transplantation.

You have a shorter follow-up period, but I was wondering, in this cohort of grade school-age kids, what are their right-sided pressures like? Do you have any sense of that? How many are normal and how many are mildly abnormal, how many are significantly elevated?

DR ST. LOUIS: The data were inconsistent, but the trend was that for repairs, and they were only followed at this point for about 4 to 5 years, the pressures were better than the replacements, but they were followed for longer; they were 6- to 10-year follow-ups so far in some of those.

In the patients that had replacements and the echoes, I went back and reviewed, their right-sided pressures, it was estimated, were in the 40 to 50 range or so; but again, that is just a gestalt of what we saw was no consistency, where the pressures with the valve repairs, again, in younger children, were more in the 30 or so range.

DR FORBESS: So not exactly normal but not prohibitively high at that point?

DR ST. LOUIS: Right.

DR FORBESS: Well, again, I would like to congratulate you on the results that you have seen with this series. I think it is a tour de force of patient selection and precision in the operating room. Thank you.

DR ST. LOUIS: Thank you.

DR EDWARD L. BOVE (Ann Arbor, MI): I rise to comment and perhaps to get your thoughts on something that troubles me a little bit in your conclusion. If I understood you correctly, you stated that the initial gradient at presentation was not predictive of the need for later mitral valve surgery. As one would expect, a large percentage of your patients were neonates and infants. These patients often have an open ductus, ASDs, poor function, and LV outflow obstruction such that the initial gradient is often fairly meaningless. So I am not quite sure how you could come to the conclusion that the initial gradient would predict later intervention.

DR ST. LOUIS: And I agree. It is an imperfect way of determining severity. We went back to look at the echoes, looking at pressures, hoping that would be a little bit more precise. We were unable to do that, and I agree. We based the 8 mm Hg on data that had already been published on small studies of Shone patients, and again, that is the issue, that every study prior to this is pretty small. I agree with you. It is a problem.

DR ANDREW C. FIORE (St. Louis, MO): Jim, I had one question related to mitral valve repair. You performed papillary muscle splitting and resection of supraannular mitral ring. Were there any other techniques that you used?

DR ST. LOUIS: The only repairs that we have done have been on the parachute valves to this point, and those were I believe were technically demanding ones. We do what Drs Mavroudis and Backer published in the ’90s. We split that common papillary muscle. And the first one that was done before I arrived at the Children’s Medical Center in Augusta was split a little too aggressively, and the patient ended up with severe MR and had replacement within that same hospitalization. And then we take the chordae and separate them all. We don’t separate the leaflets as Dr Mavroudis does, or he has published, or excise pieces of leaflets that we do, and then we put a dilator through the annulus itself.

	References

Top Abstract Introduction Patients and Methods Operative Procedures Results Comment Discussion 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): James D. St. Louis Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by St. Louis, J. D. Articles by Wiles, H. B. Search for Related Content PubMed PubMed Citation Articles by St. Louis, J. D. Articles by Wiles, H. B. Related Collections Congenital - acyanotic