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Ann Thorac Surg 2003;76:S47-S67
© 2003 The Society of Thoracic Surgeons
a Division of Cardiovascular-Thoracic Surgery, Children's Memorial Hospital, Northwestern University, Feinberg School of Medicine, Chicago, Illinois, USA
b Department of Surgery, Institute of Human Values in Health Care, Medical University of South Carolina, Charleston, South Carolina, USA
* Address reprint requests to Dr Mavroudis, 2300 Children's Plaza, M/C 22, Chicago, IL 60614, USA
e-mail: cmavroudis{at}childrensmemorial.org
Presented at the 50th Anniversary of the Southern Thoracic Surgical Association, Bonita Springs, FL, Nov 14, 2003.
Abstract
BACKGROUND: Members of the Southern Thoracic Surgical Association (STSA) have presented important pediatric cardiothoracic surgery papers at the annual meetings over the last 50 years. In order to determine the influence of these presentations on the practice of surgery, a review was undertaken. Early papers were characterized by emerging advances in open-heart surgery, anatomic congenital heart studies, and electrophysiologic discoveries that extended life with pacemakers. Later years were characterized by innovative myocardial preservation methods, improved cardiopulmonary bypass techniques, expanded homograft availability, emphasis on accurate repairs, intraoperative transesophageal echocardiography, and cardiopulmonary transplantation.
METHODS: All but one of the scientific programs of the annual meetings (that of 1964) were located. The programs were reviewed and 180 presentations were identified on topics in congenital heart disease, pediatric thoracic disease, and pediatric thoracic wall abnormalities. Of those 180 oral presentations, 155 manuscripts (86%) were eventually published or in press and available for critical review and analysis. Manuscripts were grouped by diagnosis or therapeutic intervention. We determined a "cumulative citation frequency" (CCF), which measures the number of times an article is cited in the bibliography of related papers in the universe of participating journals. The selected manuscripts were compared with the historic landmark contributions and the existing trends at the time, and the number of articles both by individual authors and from institutions were tallied.
RESULTS: Grouping by authors and institutions showed that 100 of 155 pediatric cardiothoracic manuscripts (65%) originated from 13 institutions. The CCF for the 20 leading articles ranged from 26 to 93.
CONCLUSIONS: This historical STSA 50-year record of pediatric cardiothoracic advances was accomplished in a milieu of collegial respect and camaraderie. Our annual meetings over the years have provided a venue for thoracic surgeons to share their ideas, innovations, and scientific inquiry. These contributions have significantly affected the practice of pediatric cardiothoracic surgery. The STSA has worked for 50 years and we trust that it will work for another 50 years and beyond.
As we celebrate the 50th Anniversary Meeting of the Southern Thoracic Surgical Association (STSA) a reflective review of the scientific contributions made by the membership at the long line of meetings is in order. The STSA members have made sentinel contributions to all aspects of cardiothoracic surgery, often in presentations at the STSA annual meetings. The purposes of this analysis are to review the pediatric cardiothoracic surgical papers that were presented to the STSA and highlight the presentations relative to their impact on the practice of pediatric cardiothoracic surgery. The 50-year period from 1954 to 2003 spans the development of open-heart surgery and witnessed the introduction of cross-circulation techniques [1], the heart-lung machine [2], the intensive care unit [3], myocardial preservation [4], and myocardial revascularization [5]. In addition myriad prosthetic materials made valve replacement or repair, aneurysm repair, defect closure, and arrhythmia treatment everyday realities [6]. Many of these advances and follow-up studies were reported at the STSA meetings as well as various other thoracic surgery meetings including the venerable American Association for Thoracic Surgery (the only thoracic surgical association older than STSA), the Society of Thoracic Surgeons, the American Heart Association, the American College of Cardiology, and the Western Thoracic Surgical Association (formerly the Samson Thoracic Surgical Association). One of the early established tenets of the STSA was to provide a venue for the thoracic surgeons in the southern part of the United States, especially its younger members, to present their work to their peers. As a result many important and avant-garde contributions were made that forged new developments in the field of pediatric cardiothoracic surgery.
Unfortunately some of the early programs and presentations were never published and are now lost to both critical review and speculation. However most of the programs and papers published in The Annals of Thoracic Surgery, our official organ since 1964, have survived, allowing critical review of the impact of these papers. Because advances over the last 50 years have been incremental we chose to analyze the STSA contributions by disease entity and to compare the manuscripts to practice standards at the time of publication. Early papers were characterized by emerging technologies that made possible open-heart surgery, anatomic studies that made possible repair of congenital heart defects, and electrophysiologic advances that extended life with pacemakers. Later years were characterized by innovative myocardial preservation methods, improved cardiopulmonary bypass techniques, expanded homograft availability, emphasis on accurate repairs, intraoperative transesophageal echocardiography, and cardiopulmonary transplantation.
To put this historical 50-year record into perspective, during this time 10 different Presidents have served the United States of America. Americans fought and died in numerous wars. The civil rights movement took root. Women's rights were advanced. Neil Armstrong landed on the moon. The Berlin Wall fell. In San Antonio, at the 10th Annual STSA Meeting during the afternoon session of November 22, 1963, it was announced to the membership that President Kennedy had been shot and killed in Dallas, Texas, just 200 miles to the north.
Material and methods
We were able to locate all but one of the scientific programs of the annual meetings; that of 1964 could not be found. We reviewed all available programs and of 1,381 presentations we identified 180 (13%) on topics in congenital heart disease, pediatric thoracic disease (lung and trachea), and pediatric thoracic wall abnormalities (pectus excavatum)Fig.1. Of those 180 oral presentations, 155 manuscripts (86%) [7–86 [87–161] were eventually published or in press and available for critical review and analysis (Fig 1).
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Results
The Annals of Thoracic Surgery was launched in January 1965 as the official organ of the Society of Thoracic Surgeons and the STSA. Because the STSA, which was inaugurated in 1954, predated the founding of The Annals of Thoracic Surgery, 12 manuscripts were published in other journals such as Surgery (3 papers), Journal of Thoracic and Cardiovascular Surgery (2), Journal of Surgical Research (2), Annals of Surgery (1), Surgery, Gynecology, and Obstetrics (1), Surgical Forum (1), Circulation (1), and North Carolina Medical Journal (1). The remainder of the articles were mostly published (139/143) in The Annals of Thoracic Surgery. The groupings of the 155 STSA pediatric cardiothoracic surgery articles are noted in Tables 1 through 4. Table 5 shows the institutions that contributed the majority of pediatric cardiothoracic surgery articles over the 50-year study period. Table 6 ranks authors by number of manuscripts published during the study period. Table 7 shows the CCF for the 20 leading articles among all categories since 1965 for which the Science Citation Index [162], upon which the CCF is based, was available for our use. Additionally, as noted in Tables 1 through 4, as of 2002, 7 of 39 recipients (18%) of the President's Award for the best scientific paper of the yearly meeting were awarded this coveted prize for pediatric cardiothoracic contributions.
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In its 50 years the STSA has witnessed remarkable advances in pediatric cardiothoracic surgery, many of which were contributed to by STSA members who used the platform of the annual STSA scientific meetings to present their findings to a larger audience. These contributions largely reflected the advances made during successive eras of pediatric cardiothoracic surgery: the beginnings of pediatric cardiothoracic surgery, notably closed-heart surgery for repair of simple defects and palliation of more complex anomalies, the intermediate era of early open-heart surgery, often performed with preliminary palliation, and our current era of initial early (often neonatal) one-stage open-heart repair or staged repair of lesions previously considered inoperable.
Closed heart surgery
Some of the early STSA contributions in congenital heart disease centered on closed heart techniques, which included institutional results with ligation of patent ductus arteriosus, systemic-to-pulmonary artery shunts, pulmonary artery band, and coarctation of the aorta (Table 1).
Landmark contributions involving patent ductus arteriosus ligation include the first report of successful ligation by Gross [163] in 1939; this practice was upheld by STSA authors Bosher [7] (1955) and Wilcox [21] (1967). An early report of ligation in the premature neonate [164] in 1973 was reaffirmed by STSA authors Mavroudis [60, 71] (1983, 1985), Novick [116] (1995), and Hines [128] (1998). The first report of thoracoscopic ligation by Laborde [165] in 1993 was reconfirmed and underscored by STSA authors Hines [154] (2003) and Jacobs [155] (2003). These STSA authors were instrumental in adding important technical advances: neonatal unit ligation using local anesthesia and thoracoscopy.
Success with systemic-to-pulmonary artery shunts evolved over a period of time starting with the classic Blalock-Taussig shunt [166] in 1945, the Potts shunt [167] in 1946, the Glenn shunt [168] in 1958, and the first reported series of modified Blalock-Taussig shunts (polytetrafluoroethylene [PTFE] systemic-to-pulmonary artery shunts) [169] in 1981. The STSA authors contributed many articles to support these landmark studies. Vasko [24] (1968), Martin [28] (1970), Daicoff [31, 39] (1971, 1974), Tyson [49] (1978), and Midgley [44] (1977) confirmed the excellent previous results and demonstrated methods for intraoperative evaluation, the development of subclavian steal in some subsets of patients, and the ability to perform shunts in very small patients. Daicoff received the President's Award for best scientific paper of the 1970 STSA meeting for his contribution. Mills [67] (1985) reported on a new method of using the azygos vein in augmenting the shunt, and Donahoo [53] (1980) and Pennington [56] (1981) reported on the long-term outcomes using the modified Blalock-Taussig shunt and the Glenn shunt respectively. The most recent contributions were by Bradley [148] (2002) and Reinhartz [156] (2003). Bradley showed the efficacy of dividing the main pulmonary artery after shunt creation to avoid pulmonary overcirculation and Reinhartz performed a systemic-to-pulmonary artery shunt in utero (lamb model) to create pulmonary hypertension.
The first repairs of coarctation of the aorta are credited to Crafoord [170] in 1944 and Gross [171] in 1945. During subsequent years, these operations were modified and applied to younger and younger patients. Lynxwiler [172] reported a successful end-to-end anastomosis in an infant in 1951, Vossschulte [173] reported the first prosthetic onlay graft in 1961, and Waldhausen [174] introduced the subclavian patch aortoplasty in 1966. Newer techniques using PTFE [175] and extended end-to-end anastomosis [176] also took their place in the surgeon's armamentarium. The STSA authors contributed to the development, refinement, and evaluation of coarctation and interruption of the aorta over the years. Adam [18] (1966) reported on their experience in infancy, Haller [22] (1968) on long-term results, and Weldon [38] (1973) on results with recoarctation, a significant problem at the time. Sampath [59] (1982), Sade [63] (1984), Palatianos [68] (1985), Pennington [69] (1985), and Sweeney [70] (1985) made important contributions and observations regarding ascending aortic aneurysms complicating coarctation repair, growth of the aorta after PTFE aortoplasty, changing trends in the treatment of coarctation, using an intraluminal shunt in infants, and review of reoperative techniques for recoarctation of the aorta respectively. In particular it was Sade and associates who were the early observers of aortic growth after PTFE aortoplasty (CCF 22), a very important issue at the time because of concerns about growth as well as aneurysm formation after such repair, especially in infancy. In subsequent studies Kron [90] (1990), Merrill [108] (1994), and Zehr [117] (1995) reported on the incidence of recoarctation and the appropriate reintervention method, operative risk factors, and durability of the coarctation repair, and a remarkable 30-year experience involving coarctation repair. These latter contributions highlight the ongoing problems of coarctation repair and the concerted efforts that STSA surgeons have made to improve the overall results. Hammon's contribution [73] (1986) to the staged repair of interrupted aortic arch and ventricular septal defect is a classic article that emphasized that staged repair ought to be done after only 3 months instead of the longer delay that many surgeons were using at the time. The standard contemporary operation for this malformation is a one-stage transmediastinal approach [177]; however at the time Hammon's paper was instrumental in showing that an excellent outcome can be achieved in these difficult patients. He was presented the President's Award for best scientific paper at the 1985 STSA meeting and in the following years this paper achieved a CCF of 21 (Table 1).
Open heart surgery (Table 2a)
Open heart repairs improved dramatically after the introduction of the heart-lung machine, the intensive care unit, and myocardial preservation techniques (Table 2A). Transposition of the great arteries is the most common cyanotic disease in infancy. The initial attempts at its surgical palliation were by Lillehei [178] and Baffes [179] who directed the inferior vena cava flow to the left atrium and the right pulmonary veins to the right atrium. These were the beginnings of the atrial baffle procedures, which were performed by Senning [180] in 1959 and Mustard [181] in 1964. Arterial switch operations were attempted by a number of authors, sometimes providing oxygenated flow to the coronary arteries and sometimes not. The era of anatomic correction began in earnest in 1975 when Jatene [182] reported successful arterial switch. During the time of this study STSA surgeons made significant contributions which documented and confirmed that these atrial baffle operations and later the arterial switch operation could be performed with acceptable results (Table 2A). Wilson [12] (1962), Haller [26] (1969), Lindesmith [41] (1975), and Weldon [62] (1983) all reported on their experience with the various atrial baffle operations and stressed issues such as timing of the operation, techniques to avoid baffle leaks, techniques to avoid atrial arrhythmias, and the Mustard operation as a palliative operation in the face of a ventricular septal defect and elevated pulmonary vascular resistance.
The initial large series of the arterial switch operation for infants with transposition of the great arteries and intact ventricular septum were by Castaneda [183] (1984), Radley-Smith [184] (1984), and Quaegebeur [185] (1986). Shortly thereafter STSA author Mavroudis [78] (1987) reported on a series of neonates with transposition of the great arteries and intact ventricular septum who had arterial switch with excellent results. This series, with a CCF of 14 (Table 2A), proved that this complex operation could be accomplished with consistent and excellent outcomes at a relatively small center. Subsequent reports by STSA authors Backer [83] (1989) and Elkins [105] (1994) showed that the arterial switch operation could be achieved with the same or better survival statistics as atrial baffle operations and that left ventricular hemodynamics are excellent after arterial switch respectively. After these successes the arterial switch operation was applied to patients with more complex variants of transposition of the great arteries. Association author Mavroudis [119] (1996) reported on his experience with arterial switch and the Kawashima operation [186] for Taussig-Bing anomaly (double outlet right ventricle with subpulmonic ventricular septal defect) with excellent results for both operations. This paper was selected for the President's Award for the best scientific paper at the 1995 STSA meeting.
Because many patients with transposition of the great arteries patients presented late for arterial switch, Mee [187] (1986) introduced the idea of staged pulmonary artery band in order to strengthen the left ventricle in preparation for arterial switch. This series of operations presented daunting risks because the patients were severely ill. Mavroudis [139] (2000) presented a series of patients in whom conversion from Mustard baffle to arterial switch could be accomplished with excellent results, thereby confirming the results achieved by Mee [188] and others [189]. In the Mavroudis paper arrhythmia surgery was concomitantly used in some of these patients for the first time, thereby demonstrating that patients could undergo arrhythmia surgery in addition to Mustard takedown and arterial switch.
Atrial septal defects (ASD) and ventricular septal defects (VSD) were among the first intracardiac defects to be corrected. Ingenious methods to close ASD without cardiopulmonary bypass [190] (1948) had limited success. Gibbon [2] (1954) performed the first successful open-heart operation, an ASD closure, using cardiopulmonary bypass in 1953. Lillehei [1] (1955) undertook cross-circulation techniques (mother or father of pediatric patient used as pump oxygenator) to repair ASD, VSD, and tetralogy of Fallot. Association authors have chronicled their results over the years with ASD and VSD closure. Early reports included Bosher [8] (1957) on ASD closure without cardiopulmonary bypass using a modified Sondergaard technique, and the Pate and Butterick [16] (1965) experimental assessment of various prosthetic patch materials for ASD closure, initially presented at the 1960 annual meeting. Hoffman [27] (1969) and Wilcox [29] (1970) documented their results with ASD closure with regard to postoperative follow-up and pulmonary vascular dynamics after surgery respectively. With regard to VSDs, STSA authors Robicsek [14] (1963) and Ibach [33] (1971) evaluated techniques (vertical versus horizontal ventriculotomy) and follow-up after VSD closure respectively The management of VSD in infancy has evolved over the years from selective use of pulmonary artery banding and prolonged observation to early closure. Manuscripts by STSA authors have paralleled these advances. Fisher [46] (1978) recommended "planned delay" of VSD closure in some patients and emphasized the high mortality rate for VSD closure in small infants. Hardin [96] (1992) and later Knott-Craig [115] (1995) demonstrated that VSD closure can be performed in small infants with improved survival but cautioned that associated anomalies such as multiple VSD can still impact negatively on the result. These findings hold true even today.
There have been seven STSA manuscripts dealing with the various forms of congenital aortic stenosis over the years ranging from 1963 to 1995. To put these contributions in perspective, closed aortic valvotomy was first reported by Marquis and Logan [191] (1955). The first report of accurate valvotomy during cardiopulmonary bypass was by Spencer [192] (1958). Association reviews of the approach to the spectrum of congenital aortic stenosis began with Pate's early report [13] (1963), which emphasized the benefits of the open-heart technique and discussed options available for myocardial protection during surgery for relief of valvar or subvalvar aortic stenosis. Twenty years later Mavroudis [64] (1984) presented his results with surgery for valvar, subvalvar, and supravalvar aortic stenosis, emphasizing the utility of intraoperative pressure gradient measurements to accurately assess results of surgery and provide options for immediate reexploration and further intervention. Johnson [66] (1985) in a review of 22 years of experience with surgical relief of the various forms of congenital aortic stenosis focused on the incidence of reoperations so often required in this patient population.
Members of the STSA also reported on outcomes and predictors of mortality for specific anatomic subsets of aortic stenosis. Stewart [91] (1990) reviewed outcomes and reoperation rates for patients with fixed subaortic stenosis who underwent localized resection of the obstruction, and Hammon [80] (1988) in a retrospective review of patients with critical valvar aortic stenosis concluded that two factors—small left ventricular dimension and elevation of pulmonary artery pressure—might be predictors of poor outcome. These latter studies, both from Vanderbilt, achieved CCFs of 23 and 39 respectively. In another retrospective study of late outcome after various surgical procedures for relief of neonatal (30 days or younger) critical aortic valve stenosis in a large cohort of patients from both Duke University and Hospital for Sick Children, London, Gaynor [111] (1995) concluded that while late survival was excellent after initial high mortality, reintervention within 10 years was required in most patients. In a report comparing the results of single sinus and bisinus patch repair of the uncommonly encountered supravalvar aortic stenosis Braunstein [88] (1990) found both techniques were safe and resulted in good hemodynamic indicators.
Isolated congenital anomalies of the coronary artery are rare however when anomalies of coronary artery origin, course, or termination associated with complex defects such as transposition of the great arteries and tetralogy of Fallot are considered, pediatric cardiothoracic surgeons encounter these anomalies on a more frequent basis. Surgical treatment of the two major isolated congenital anomalies—coronary artery fistulas and anomalous left coronary artery arising from the pulmonary artery—has evolved over the last 60 years, from Biorck and Crafoord's [193] (1947) first report of suture closure of a coronary fistula in a patient with a preoperative diagnosis of patent ductus arteriosus and Sabiston's [194] (1960) successful ligation of an anomalous coronary artery. The STSA meeting presentations reflect this, starting with the Sanger [10] (1959) report of two cases of coronary arteriovenous fistulas, one of which was correctly diagnosed preoperatively and successfully operated on. The Sanger paper, presented within a year of the probable first report [195] (1958) of the suture repair of a correctly diagnosed fistula, emphasized a departure from the then current view that clinical diagnosis of coronary arterial fistulas was unlikely and most often made only at autopsy. Forty years later Mavroudis [124] (1997; CCF 38) presented his institution's 28-year experience with surgical approaches to coronary artery fistula with commentary on the rise of transcatheter embolization techniques as an alternative intervention.
The comprehensive paper by Blake [15] (1964; CCF 93) covering the range of coronary artery anomalies and the implications for surgical therapy centered on pathologic specimens that corresponded to Edwards' classification scheme [196]. This paper was presented to the STSA membership in San Antonio, Texas, on the afternoon of Friday, November 22, 1963. With the announcement of President Kennedy's assassination the decision was made to continue with the scientific programs but cancel all social events. Later in the decade Dalton [25] (1969) presented a case report of ligation of an adult-type anomalous left coronary artery arising from the pulmonary artery. Contemporaneous with Dalton's report, alternative surgical strategies for this anomaly were being reported by others including attempts to establish a two-coronary artery system [197] and the use of coronary artery bypass vein grafts [198]. Successful reimplantation of an anomalous left coronary artery would not be reported until 1974 [199]. At the 2001 STSA annual meeting Huddleston's [147] results with reimplantation of anomalous left coronary artery over a 15-year period were reviewed with a special focus on the long-term function of the mitral valve in this high-risk group of patients. He found that mitral valve repair was not generally necessary at the time of initial operation but should mitral regurgitation persist or recur it could be associated with significant left coronary artery obstruction. At our most recent meeting in Miami Beach in 2002 Jaggers [157] presented his results with modified intramural coronary segment unroofing for anomalous aortic origin of the left or right coronary artery, including intermediate-term follow-up and assessment with stress echocardiography studies.
Lillehei and colleagues [200] performed the first successful repair of a complete atrioventricular canal defect in 1954 using cross-circulation and suturing the atrial rim of the defect to the ventricular septum crest. Later reports described the use of patches to close the ventricular and atrial components of the defect and in 1962 Maloney [201] reported repair in two children using a single-patch technique. The STSA annual meeting in 1990 was a venue for the debate between the single- and double-patch technique advocates with Merrill [94] presenting his series of patients repaired with the single-patch technique and Bailey and Watson [92] reporting their series of patients repaired using the double-patch technique. Gurbuz and Watson [132] (1999) at the 45th Annual Meeting of the STSA described a significant subset of patients who were operated on for left ventricular outflow obstruction after repair of partial atrioventricular canal defects.
Open heart surgery (Table 2b)
Prosthetic valve replacement has significantly impacted the treatment of valvar heart disease in both adults and children (Table 2B) [202]. Early successes with prosthetic valve replacement for degenerative and congenitally malformed valves [203, 204] brought many new technologic changes both in preoperative and postoperative care as well as improved valves with more favorable hemodynamic properties. The experience with children soon followed. Young and Robinson are credited with performing the first successful prosthetic valve replacement in an infant in 1964 [205]. Soon thereafter STSA authors Taylor and Foster [20] reported their experience in 1967 with prosthetic valve replacement in four children with excellent results. Thirteen years later STSA authors Smith, Cooley, and associates [55] reported a 15-year experience with aortic valve replacement in 26 children with an astonishing 3.8% mortality for the time period. The authors emphasized the need for future replacements because of patient somatic growth. The Konno procedure [206] was introduced to allow a larger aortic valve prosthesis to be placed in patients with congenitally small aortic annuli. Association authors Fleming and associates [75] presented their results at the 1986 meeting held at White Sulfur Springs and showed that this very complex operation could be accomplished with excellent results for both the short and long term.
There has been controversy concerning the anticoagulation regimen for prosthetic heart valves in children. Because warfarin therapy management can be difficult in infants and children, Weinstein, Mavroudis, and Ebert [207] reported their preliminary experience with aspirin and dipyridamole to treat children with prosthetic heart valves in 1981. The longer-term results from this same institution were affirmed in a later publication [208]. Association authors Bradley, Sade, and Crawford [121] presented their retrospective comparative experience with warfarin anticoagulation and antiplatelet drugs for children with prosthetic heart valves and found no appreciable difference between the regimens. Despite these interesting findings there is still disagreement on how to manage these patients and the answer will no doubt have to come with a well-controlled prospective clinical trial. Because of the anticoagulation problem with replacement heart valves Ross [209] and later Elkins [210] introduced and popularized the Ross operation, which entails a pulmonary autograft to be used in the aortic position and a pulmonary homograft for the pulmonary position. Other authors found some fault with this operation because of annular dilatation [212]. Because of this controversy Elkins presented his experience on how to avoid annular dilatation at the 1995 meeting of STSA [129] and showed how annular fixation could be performed in 20 patients 7 to 47 years of age without subsequent dilatation. Elkins reported his experience with children using this operation at the 1997 Society of Thoracic Surgeons meeting [211]. Elkin's experience with this operation and his efforts over the years to educate surgeons as to the technical aspects, often at STSA meetings, has earned him the respect and gratitude of his colleagues who still use this operation effectively.
Still another potential approach to left ventricular outflow problems is the apicoaortic conduit, which was reported by Sarnoff [213], Cooley [214], and Brown [215] among others in 1953, 1980, and 1984 respectively. Association authors Sweeney, Cooley, and associates [74] (1986) reported on the use of apicoaortic valved conduits for the relief of complex forms of left ventricular outflow tract obstruction. At Marco Island in 2000 Khanna and associates [151] presented a comprehensive and long-term review of their 10 patients who had this procedure and concluded that apicoaortic conduits provide a safe and effective treatment alternative for selected cases.
The surgical history of tetralogy of Fallot effectively started with the Blalock-Taussig palliative shunt [166] and the first complete repair by Lillehei and Varco using cross-circulation techniques in 1954 [1]. Association authors have contributed to the development of improved techniques over the years with tetralogy, double outlet right ventricle, and double-chambered right ventricle. In 1971 Bender, Haller, Gott, and associates [30] reported on the Johns Hopkins experience with tetralogy of Fallot in 84 adults who were compared with the large cohort of children undergoing repair. It is interesting to note in this series that the authors started to use aortic homografts in the pulmonary position to avoid right ventricular dysfunction seen with transannular pulmonary outflow tract patches in adults. This idea was to resurface many years later for patients like this. One year later in 1972, STSA authors Selmonosky, Ellison, and associates [37] described their results with palliation and complete repair in tetralogy patients and concluded that early correction, when feasible, is better than staged palliative repair. Although the idea of infant and neonatal repair was far away, the idea was viable and noteworthy.
A follow-up paper from Johns Hopkins describing operative management of the Blalock-Taussig anastomosis during total repair of tetralogy of Fallot [35] in 1972 proved to be a prophetic paper as three of the collaborating authors of this manuscript, Elkins, Bender, and Haller, were destined to become Presidents of the STSA. The paper was an important one for its time since the authors discuss the technical aspects of finding and ligating the shunt during complete repair. This information no doubt saved many lives at the time because of the useful techniques that were described. In 1987 STSA authors Simpson, Sade, Crawford, and associates [79] published their results on double-chambered right ventricle in 30 patients and emphasized the importance of preoperative evaluation and intraoperative management.
Knott-Craig, Elkins, and associates [130] presented their 26-year experience with surgical management of tetralogy of Fallot and included risk analysis for mortality and late intervention in 1997 at the 44th Annual STSA Meeting in Naples. The presentation was a comprehensive analysis of their results with primary repair, staged repair, and palliative procedures, which were attended by excellent short- and long-term outcomes. The paper subsequently earned a CCF of 13 and Knott-Craig was presented the President's award for the best scientific paper of the meeting. In 1999 McDonnell, Gaynor, Spray, and associates [135] presented a very nice series of tetralogy of Fallot with absent pulmonary valve, which is a distinct class among tetralogy varieties because of the gigantic pulmonary arteries that can compress the trachea and cause significant morbidity and mortality. The early mortality of 21.4% indicates the severity of the disease entity with which these authors were confronted. They emphasized the importance of pulmonary plication and valved conduits for these difficult patients, which was suggested by Mavroudis, Ebert, and associates in 1983 [216].
Association authors Brown and associates [145] presented a comprehensive review of their experience with repair of double outlet right ventricle in 124 patients in 2001 at the 47th Annual Meeting in Marco Island. This landmark paper recapped a 20-year experience and concluded that survival was excellent for all patients undergoing intraventricular tunnel repair, arterial switch operation, and repair with a conduit or a modified Fontan procedure. They stressed that careful attention to preoperative anatomy dictates the best surgical approach and will enhance outcomes. The final paper of this series was presented by de Ruijter and associates [149] in 2001. They presented their results with tetralogy repair with special reference to postoperative right ventricular dysfunction due to pulmonary insufficiency. They used pulmonary valve implantation in a select number of patients and stressed that this option should be used before the onset of symptoms in order for this to be effective.
Surgical palliation of hypoplastic left heart syndrome started with the early attempts at staged univentricular repair first reported by Litwin [217] in 1972 and then popularized by Norwood [218] in 1981. Because the early results were poor, cardiac transplantation for hypoplastic left heart syndrome became popular [219] only to be replaced in most centers by the Norwood operation owing to improved perfusion techniques, postoperative management strategies, and increasing experience with the operation [220]. Authors of the STSA made important contributions by confirming the emerging improved results from the more experienced centers. The first contribution by an STSA author was made by Gustafson, Murray, and associates [84] in 1989 at the 35th Annual Meeting in Marco Island. Among their 12 patients, there were two early and two late deaths resulting in an overall survival of 67%, which compared favorably to the early results in the literature at the time. This paper went on to score a CCF of 7 and Gustafson earned the President's Award for the best scientific paper of the meeting. In 1995 STSA authors Kanter and associates [114] presented their experience with the Norwood operation for patients who had other forms of single ventricle at the 40th Annual Meeting in Panama City Beach, Florida. They reported a 94% (17 of 18 patients) first-stage survival in these patients. For this remarkable achievement Kanter was presented the President's Award for the best scientific paper of the meeting. Association authors Bando, Brown, and associates [118] performed a retrospective review of their experience with both cardiac transplantation and the Norwood operation for hypoplastic left heart syndrome and found that both modalities were necessary in selected cases.
Subsequent contributions were made by STSA authors Drinkwater [146] and Ungerleider, Austin, and associates [161] in 2001 and 2003 respectively who championed the importance of perioperative extracorporeal membrane oxygenation for circulatory support of the patient undergoing the Norwood operation. In general the major trends nowadays for the surgical management of hypoplastic left heart syndrome are refinements of the Norwood operation with systemic-to-pulmonary shunt or right ventricular-to-pulmonary artery shunt, early conversion to bidirectional shunt (or hemi-Fontan operation), and ultimately the Fontan operation either by lateral tunnel or extracardiac techniques.
There have been four manuscripts concerning heart and lung transplantation in children presented at STSA meetings. Two of these were presented at the 41st Annual Meeting at Marco Island. Fullerton, Jaggers, Grover, and associates [110] reviewed their experience with 30 patients 8 months to 24 years of age who had cardiac transplantation for congenital heart disease and cardiomyopathy. Their operative mortality (3.3%) with excellent midterm results ranks as one of the best series in the literature. Bridges, Huddleston, Spray, and associates [109] reported on their results with lung transplantation in patients who had pulmonary hypertension and congenital heart disease at the same meeting. They found that the usual problems of bronchiolitis obliterans and rejection episodes were similar to another cohort of patients who underwent lung transplantation for lung disease. Both of these programs have continued to perform transplants for pediatric cardiopulmonary disease and serve as models for the nation in this regard. Association authors Kanter and associates [134] and Fenton, Pigula, and associates [158] also reported their series of cardiac transplantation in children. Kanter presented a stellar series of 95 transplants in 89 children with end-stage heart failure or complex or inoperable congenital defects. They achieved a 1- and 5-year actuarial survival of 79% and 69% respectively, which compared favorably with the national norms. Fenton described the long-term survival after pediatric cardiac transplantation and postoperative extracorporeal membrane oxygenation support in a very difficult group of patients with posttransplant graft failure and pulmonary hypertension. She and her colleagues achieved excellent survival considering the conditions and severity of disease.
The surgical treatment of total anomalous pulmonary venous return did not achieve as good results initially as did repair for the other more common cardiac anomalies. In fact even up until 1974 when STSA authors Wukasch, Cooley, and associates [42] presented their experience at the 21st Annual Meeting in Williamsburg, Virginia, the results with treatment of total anomalous pulmonary venous return in infancy were less than stellar. These authors with excellent results with other disease entities reported 37% operative mortality and 57% mortality within the first year of life. Many authors took note of this paper (CCF 43, Table 7) because the report was from one of the leading institutions and eminent surgeons of that era. This report was to stand as a yardstick for improvement that occurred over the next decade [221] using better perfusion techniques and improved myocardial preservation. Association authors Fleming and associates [50] reported on their experience with four patients who had late complications after repair of total anomalous pulmonary venous return below the diaphragm. Their analysis of these complications predates the clinical recognition of sclerosing pulmonary veinitis, which has received some attention lately because of the uniformly bad results that attend this unfortunate problem [222]. Cope, Kron, and associates [122] of the STSA reported on their series of total anomalous pulmonary venous return repair by leaving patent the vertical vein as a decompressive channel to achieve postoperative stability.
There was one STSA paper on partial pulmonary venous return by Warden, Gustafson, and associates [65] in 1984, which completely revised the repair of these difficult patients by superior vena cava transection and reimplantation into the right atrial appendage after successful baffling of the pulmonary veins into the left atrium. Numerous subsequent papers sometimes referring to this classic article (CCF 21) and sometimes not have taken on these tenets for repair with excellent long-term outcomes.
Open heart surgery (Table 2c)
The next group of papers includes the topics of Fontan pathways and arrhythmia surgery (Table 4), not so much because the subjects are related to any great extent but more so because all of the four manuscripts originate from the Medical University of South Carolina where Drs Sade, Crawford, and Bradley have shown great interest in Fontan physiology and arrhythmia ablation over the years. Association authors Lee, Crawford, and associates [85] reported on accessory pathway ablation for supraventricular tachycardia in patients 1.5 to 31.6 years of age with excellent short- and long-term results. At the time of this report there were only a handful of centers around the world performing this type of surgery. Crawford's residency training with Duke Professor of Surgery, Will Sealy, a long-time member of the STSA and President in 1966, proved to be the impetus for this landmark contribution. During this time Crawford's associates made some interesting contributions concerning Fontan pathway physiology. Association authors Douville, Sade, and associates [93] described their experience with the hemi-Fontan operation en route to orthoterminal correction in 17 patients with one death. The authors stressed the advantages of staged correction. Association authors Bradley and associates [144] studied the effects of aortopulmonary collateral arteries in the Fontan patient and concluded that increased aortopulmonary collateral flow has no impact on the outcome of the Fontan operation as long as the patients are well prepared for the procedure. This idea has created some controversy in the literature and surgeons are recognizing that a controlled study will be necessary to determine whether or not aortopulmonary collaterals are important in the Fontan circulation; and if they are, which patients are most affected. Subsequently Kumar, Bradley, and associates [159] performed a retrospective study comparing lateral tunnel and extracardiac Fontan operations with regard to arrhythmias and other risk factors. They found no appreciable differences. This controversy will be a significant discussion point in the years ahead and will have to be decided by a well-planned multi-institutional prospective study.
Extracorporeal circulation, operative techniques
Topics relating to extracorporeal circulation, modified ultrafiltration, and perioperative extracorporeal membrane oxygenation have been presented to the STSA at timely intervals (Table 3). Some of the initial studies with cardiopulmonary bypass were performed by Gibbon, which resulted in the first use of extracorporeal circulation in a human [2]. It was not very long after that that STSA authors Sealy and associates [9], Ellison and associates [11], Peirce and associates [17], and Neely and Heining [23] performed studies in humans and animals to push back the frontiers of safe execution of routine cardiopulmonary bypass. Most of the efforts were centered on hypothermia techniques, induction of cardiac arrest (this was well before the clinical era of potassium cardioplegia), and hemodilution. As new technology developed, it was obvious that surgeons would have to choose between bubble and membrane oxygenators. One of the first comprehensive studies comparing these modalities in a prospective and randomized manner was by STSA authors Sade and associates [54]. They found there were no physiologic differences between bubble and membrane oxygenators; they recommended that future changes should be based on safety, cost, and convenience, not physiology. The standard for today after years of experimentation is membrane oxygenation.
Experiments to evaluate the effects of deep hypothermia and circulatory arrest have been performed by surgeons from two notable STSA programs (Duke University and Johns Hopkins University). At the 39th Annual STSA Meeting in Wesley Chapel, Florida in 1992, Gillinov, Baumgartner, and associates [101] and Mault, Ungerleider, and associates [107] presented their findings using hypothermic circulatory arrest. Gillinov studied 12 dogs that underwent profound deep hypothermia and circulatory arrest using temperatures of 18°-20° C in one group and 5°-7° C in the other group; he found that the 5°-7° C group afforded better neurologic protection. This study showed that the 18° C threshold that is used in humans might be extended to colder temperatures for maximal neurologic protection. Mault performed his studies using deep hypothermic circulatory arrest to test the idea that sequential periods of hypothermic circulatory arrest could be better tolerated than one extended period. Their findings indicate that sequential periods offer better neurologic protection, which can help the clinician in the decision to re-repair an unsatisfactory result when it was performed under deep hypothermia circulatory arrest. Two follow-up studies from these institutions, those of Tsui, Gaynor, Ungerleider, and associates [120] and Tseng, Baumgartner, and associates [141], investigated the effects of neuronal nitric oxide production in the pathogenesis of central nervous system injury during deep hypothermia and circulatory arrest.
Extracorporeal membrane oxygenation for the perioperative support of infants undergoing complex open heart repairs has been described and advanced as a reasonable rescue therapy in certain subsets of patients. Association authors Weinhaus and associates [87], Jaggers, Ungerleider, and associates [138], and Ahron, Drinkwater, and associates [142] have presented their experience with perioperative extracorporeal membrane oxygenation in 1989, 1999, and 2001 respectively. In general the authors stressed the implementation of perioperative extracorporeal membrane oxygenation early to prevent end-organ failure and noted that patients who required it after a period of separation from cardiopulmonary had a better chance of survival.
The introduction of modified ultrafiltration by Naik and associates [223] and further demonstration that the technique could improve hemodynamics by the same group [224] ushered in the era of routine modified ultrafiltration in many congenital heart centers. Association authors Koutlas, Gaynor, Spray, and associates [123] used modified ultrafiltration to improve their results with operations involving cavopulmonary connections, while STSA authors Gurbuz, Watson, and associates [127] employed modified ultrafiltration to conserve blood in small patients undergoing ASD closure. Modified ultrafiltration has been shown to improve hemodynamics, improve blood conservation, decrease interstitial body water retention, and many believe that the procedure can remove molecules, yet to be defined, that can cause myocardial depression.
A number of STSA authors reported their clinical experiences based on large numbers of combined cases, operative approaches, and intraoperative transesophageal echocardiography. In 1970 at the 17th Annual Meeting in Boca Raton, Florida, STSA authors Hallman and Cooley [32] presented their combined series of 1,050 patients younger than 1 year old who underwent cardiovascular surgery. This was a landmark paper that reviewed their results with closed and open-heart surgery in an era when techniques were evolving on a daily basis. The authors discussed the anomalies they were able to treat by one-stage and two-stage techniques and noted that the high early mortality decreased over time.
In the next decade STSA authors Richi, Sade, and associates [57] (1981) and Sade, Crawford, and associates [58] (1982) reported on their preferred exposure to the left atrium in small children by the superior approach and the use of inflow occlusion for semilunar valve stenosis in young infants and neonates respectively. Association authors DeLeon and associates [72] reviewed 158 children who underwent 164 operations through repeat median sternotomy. They attributed their excellent results to the chisel and mallet technique for resternotomy and the judicious use of electrocautery to avoid ventricular fibrillation. Association authors Jaklitsch, Bolman, Fiker, and associates [126] presented a rare series of open-lung biopsy in children. This series is from the University of Minnesota, which introduced the Heath-Edwards classification of severity of pulmonary hypertension in children. Interestingly their results showed that 98% of patients undergoing open-lung biopsy resulted in an altered therapeutic approach.
The approach to right ventricular outflow tract obstruction or insufficiency is an unresolved issue not only because of the material available for reconstruction but also because of the diagnostic dilemma of when to intercede with surgery. In 1989 STSA authors Mack and associates [86] presented a series of piglets that underwent transannular right ventricular outflow tract reconstruction with gluteraldehyde-fixed bovine pericardium. At the time this was an important study because the myriad of patches that we have today were not available. Their contribution allowed surgeons to add this technique to their armamentarium. Right ventricular outflow tract reconstruction using pulmonary valves after repairs of tetralogy, pulmonary atresia, and truncus arteriosus, among others, required constant review to determine what if any valved conduit was the best. Association authors Bielefeld, Grover, Clarke, and associates [143] and Kanter, Williams, and associates [150] catalogued their experience with various pulmonary valve replacements and reported on their experience in 2001 and 2002 respectively. Bielefeld noted that longevity for second time pulmonary valve replacement is the same as for the first time. Kanter reported that the results for pulmonary valve replacement are excellent and that porcine and homograft valves function equally well. The future holds much promise for tissue-engineered valves [225], which have the benefit of genetic homogeneity with the recipient and the possibility of somatic growth. The next decade will show progress in this field and it is likely that some of these presentations will be made at future STSA meetings.
One of the most outstanding contributions involving intraoperative diagnostic management of congenital heart disease was made by Ungerleider and associates [98] from Duke University at the 38th annual STSA meeting in Orlando, Florida. Ungerleider discussed the "learning curve" for intraoperative echocardiography during congenital heart surgery and advised surgeons on the judicious use of this relatively new modality. While new at the time, intraoperative transesophageal echocardiography was destined to become a routine intraoperative diagnostic tool that has become commonplace in the majority of operating rooms worldwide. For this contribution, Ungerleider was honored with the President's Award for best scientific paper of the meeting.
Thoracic, chest wall, and tracheal topics
The idea that the heart could be electrically paced to treat heart block was well known in the early 20th century (Table 4) [226]. However it was not until 1959 that Elmquist and Senning in Stockholm reported the first totally implantable pacemaker system using epicardial electrodes [227]. The implantable devices at the time were quite large and not suitable for children. Because of this STSA authors Albert, Glass (Past President, 1974), and associates [19] formed a pedicle graft of the right atrium and sutured it to the right ventricle in dogs in an attempt to recreate atrioventricular conduction. The experiments failed but the idea almost 40 years later is a viable option especially with the development of tissue engineering techniques. Association authors Donahoo, Haller, and associates [43], Shearin and Fleming [48], and Kratz, Crawford, Sade, and associates [97] all reported on their experiences, which have mirrored the technical advances of implantable pacemaker systems.
Tracheal stenosis in pediatric patients has many etiologic factors, which include vascular rings, complications of tracheostomy, and tracheomalacia. Sentinel contributions include the first description of a "vascular ring" by Gross [228] in 1945, the first repair of a "pulmonary artery sling" by Potts [229] in 1954, and pericardial patch tracheoplasty by Idriss [230] in 1984. Association authors Rodgers and associates [61] used endotracheal cryotherapy to treat refractory airway strictures in young patients. Clevenger and associates [89] preformed aortopexy in 12 children to treat the compression syndrome caused by the aorta on the trachea. Michna and associates [81] used cricoid split for subglottic stenosis in infancy with excellent results. Chun, Haller, and associates [95] reviewed their 22-year experience with vascular rings and concluded that early repair and excision of an associated aortic diverticulum is safe, effective and allows for normal tracheal growth. Perhaps the most avant-garde presentation concerning tracheal problems was made by Jacobs, Quintessenza, and associates [133] in 1998. They described their patients who had tracheal homografts and reviewed the worldwide experience. The authors concluded that this treatment modality will play an important role in the management of difficult airway problems.
There has been a long tradition of reporting the results of pectus excavatum repair at the STSA meetings. Between 1973 and 2003 seven reports documented the excellent results that have been achieved over the years. Most of the authors used some modification of the techniques popularized by Ravitch [231] in 1949. Association authors Davis (Past President, 1968) and Shah [40] presented their experience with the sternal turnover technique as introduced by Wada [232] in 1965 for repair of pectus excavatum in 17 patients at the 20th Annual STSA Meeting in 1973 in Louisville, Kentucky. The authors discussed their departure from the Ravitch procedure and noted the advantages and disadvantages of the sternal turnover technique. Four years later STSA authors Randolph and associates [45] reported their series of 50 patients less than 36 months of age with excellent results. Although the accompanying discussion of the paper was not published in The Annals of Thoracic Surgery at the time several STSA members remembered a spirited discussion that the age of repair (less than 36 months) was too young because of the potential high recurrence rate (personal communication, C.M.).
One year later Haller and associates [112] described their results with the modified Ravitch repair and explored the idea of internal bar fixation to optimize the results. Unlike Randolph, Haller's group recommended repair between 4 to 6 years of age. In keeping with his recommendation Haller [112] presented a series of patients from another institution who had too extensive a repair and at too young an age, which resulted in chest wall growth retardation and restrictive lung disease. Haller's recommendations have proved to be correct and were emphasized in reports by STSA authors Willekes, Backer, and Mavroudis [136], Mansour, Miller, Jones, and associates [153], and Thourani, Miller, Mansour, and associates [160] in 1999, 2002, and 2003 respectively. In particular the reports by Willekes (Chicago) and Mansour (Atlanta) represented 26- and 30-year experiences with pectus repair and other chest wall abnormalities with excellent short- and long-term results.
Some of the more challenging cases in pediatric cardiothoracic surgery are the ones that require careful management of mediastinal infections and persistent chylothorax. Association authors have contributed to the accumulated literature on these subjects in a thoughtful and comprehensive manner. Cheatham, Elkins, and associates [52] reviewed their experience with hemophilus influenzae purulent pericarditis in nine patients. The authors stressed the use of echocardiography, appropriate antimicrobial therapy, and open drainage when necessary. They achieved 100% survival in their patient population. Stiegel, Guyton, Williams, and associates [82] in 1988 and Backer, Mavroudis, and associates [104] in 1994 presented their results with vascularized muscle flaps to treat life-threatening mediastinal wounds in children. Both groups extolled the benefits of using these muscle flaps especially when exposed foreign material such as prosthetic grafts complicates the healing process. The incidence of persistent chylothorax has increased in the last decade because of the improved clinical results with the Norwood operation and staged orthoterminal correction for the various types of single ventricle anomalies. Association authors Bond and associates [100] and Wolff, Rodgers, and associates [137] reviewed their experience and offered selective use of thoracic duct ligation and pleuroperitoneal shunting in difficult cases. Association authors Graham, Tribble, Daniel, Rodgers, and associates [106] stressed the use of video-assisted thoracic surgery as a minimally invasive procedure to avoid thoracotomy. Finally Aru and associates [131] reviewed an extensive series of 546 patients who had thoracotomy for congenital cardiovascular procedures and found that in the majority of patients, tube thoracostomy could be avoided altogether. This proved to be an important paper because avoidance of chest tubes in selected cases can decrease costs and discomfort for our patients.
During the STSA 50-year existence it is noteworthy that 100 of 155 pediatric cardiothoracic manuscripts (65%) originated from 13 institutions (Table 5). The list of leading authors based on the CCF (Table 6) mirrors this experience and shows the responsible surgeons who contributed to this record. It is also interesting to note that of these 10 authors, five have been STSA Presidents, five have received the President's Award for best scientific paper, six were thought worthy of the Osler Abbott Award for erudite discussion, and three received the coveted Tiki award for the most memorable slide. This historical STSA 50-year record of pediatric cardiothoracic advances was accomplished in a milieu of collegial respect and camaraderie. Every member of the STSA has appreciated the intangible essence of our meetings. The balance of scientific contributions, the self-deprecating humor, and the family participation are the foundations of our Association. It has worked for 50 years and we trust that it will work for another 50 years and beyond.
Acknowledgments
The authors would like to thank Ms Patricia Heraty for her organizational skills and editorial expertise in the preparation of this manuscript.
Appendix. List of all journals involved in cumulative citation frequency
References
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