Surgery for Atrial Septal Defect

Surgery for atrial septal defect: from the first experiments to clinical practice Vladimir V. Alexi-Meskishvili and Igor E. Konstantinov Ann Thorac Surg 2003;76:322-327

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OUR SURGICAL HERITAGE

Surgery for Atrial Septal Defect: From the First Experiments to Clinical Practice Vladimir V. Alexi-Meskishvili, MD, PhD, and Igor E. Konstantinov, MD Deutsches Herzzentrum Berlin, Department of Cardiothoracic and Vascular Surgery, Berlin, Germany, and Division of Cardiovascular Surgery, Hospital for Sick Children, Toronto, Canada

“A more detailed history of surgery will be of use to you only later, when you have gained more insight into the merit and fallacy of certain systems, methods, and operations.” Theodor Billroth [1]

T

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he year 2002 marked the 50th anniversary of the world’s first successful open heart operation— closure of the atrial septal defect (ASD) by F. John Lewis. After initial success with extracardiac operations, such as the correction of patent ductus arteriosus, coarctation of the aorta, and the Blalock-Taussig shunt, cardiac surgeons began to explore intracardiac operations. The ASD surgery, because of its relative simplicity, became a logical starting point. It is of interest that before 1934 among 62 recorded autopsy cases of ASD, only one had been diagnosed correctly during life [2]. In 1941, the world experience with ASD was limited to clinical diagnosis in a few patients [3]. No effective means were available to help these patients. Many surgeons, who also contributed greatly to the development of other fields of cardiac surgery, attempted ingenious operations on the atrial septum.

The First Experimental Atrial Septal Defect Creation The first experimental effort to produce ASD appears to have been that of Ivan P. Dmitrieff of Moscow. He published the results of his study in 1925 in Russian and the following year in German [4]. In his experiments, Dmitrieff pushed two atrial appendages together against the septum with his fingers to detect an exact position of the fossa ovalis and then slid an instrument through the left atrial wall underneath the finger to perforate the atrial septum. In 1948, Blalock and Hanlon described their method for ASD creation [5]. The operation, later referred to as a Blalock-Hanlon operation, was not only the first successful clinical intervention on the atrial septum, but also remained the only palliation for children with transposition of great vessels for many years. A number of methods for producing and repairing ASD were proposed and studied between 1939 and 1953. Attempts to develop an effective closed technique for the permanent correction of ASDs involved a great deal of ingenuity, animal experimentation, and clinical trials [6]. Address reprint requests to Dr Alexi-Meskishvili, German Heart Institute Berlin, Augustenburger Platz 1, 13353 Berlin, Germany; e-mail: [email protected].

© 2003 by The Society of Thoracic Surgeons Published by Elsevier Inc

The First Experimental Atrial Septal Defect Closure Experimentally, the closure of ASD was first attempted in 1939 in Columbia Presbyterian Hospital. Discussing the presentation of Gordon Murray read before the American Surgical Association in Quebec on May 29, 1948 [7]. Arthur Blakemore said: “In 1939 we were rather interested in seeing how well simple inversion of the auricular appendage worked when pushed through the opening and, after getting in there, packing in some fascia lata to make it a self-retaining ball on the other side of the septum.” There was no mentioning of which appendage was inverted [8]. In 1947 Roy Cohn from San Francisco described the method of ASD closure by invaginating and suturing of a portion of the right atrial wall to the septum. The invaginated portion was cut off and the remaining atrial wall repaired (Fig 1). He operated on 8 dogs, but only 5 survived [9]. This method was never attempted clinically.

The First Clinical Atrial Septal Defect Closure In 1948 Gordon Murray from Toronto reported the first recorded clinical operation by externally suturing an ASD in a 12-year-old child [7]. He passed two sutures through the atrial septum and tied them together posteriorly. When the right atrium was diminished to less than one-half, he permanently tied the sutures (Fig 2). Subsequent catheterization revealed that the defect was only partially closed. Paul Santy and his colleagues from Lyon, France, performed a successful clinical operation on August 28, 1949, using the technique of intussusception of the right auricular appendage through the defect into the left auricular appendage [10]. In 1949 Forrest D. Dodrill from Detroit described his experimental double-clamp technique, which permitted compression of the right and left atrial walls against the septum so that the right lateral wall could be sutured to the rim of the septal defect [11] (Fig 3). This method was never used clinically because of the technical difficulties in applying the clamp to both atrial appendages for approximation. Henry Swan working in Denver in the early 1950s suggested the method of simultaneous invagination of the two auricular appendages after a threaded curved probe had been passed from one auricular tip to another through the defect. Plastic buttons threaded on the Ann Thorac Surg 2003;76:322–7 • 0003-4975/03/$30.00 PII S0003-4975(03)00508-3


SURGICAL HERITAGE ALEXI-MESKISHVILI AND KONSTANTINOV HISTORY OF ATRIAL SEPTAL DEFECT SURGERY

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Fig 1. Roy Cohn’s method of the atrial septal defect (ASD) closure by invagination and suturing of a portion of the right atrial wall to the septum. (1) Introduction of the suture from the outside of the right auricular wall, which attaches a portion of the auricular wall to the septum. (2) Introduction of the steel wire into the auricular muscle. The wire surrounds the portion of the muscle sutured to the ASD. (3) Relation of the steel wire to the portion of the auricular wall attached to the ASD. (4) Completely introduced steel wire and the beginning of the introduction of the running silk suture. (5) Detached portion of the auricular wall closes the ASD. (6) Closure of the right atrial wall. (Reprinted from Cohn R, Am Heart J; 1947;33:453–7 [9], with permission.)

transatrial sutures were approximated by tightly tying the sutures (Fig 4) [12]. In 1953 Charles P. Bailey working in Philadelphia reported 6 patients operated on using Swan’s method with 2 operative deaths and incomplete closure of the ASD in the surviving patients [13]. In 1951 K. Alvin Merendino and coauthors working at the University of Washington in Seattle described a method of plugging an ASD by the use of a pericardial bag or a tampon of autogenous fat, which was fashioned by suture in the general shape of mushroom (Fig 5). With this method the ASD was closed completely in 5 of 6 animals [14]. In the same year, Hufnagel and Gillespie also described an experimental method of applying two halves of a polyethylene button respectively to the opposite sides of an artificially created septal defect, the prototype of modern umbrella device methods (Fig 6). This method was applied in 3 cases, but unfortunately all the patients died [15]. These early attempts illustrate that many “new” ideas are often based on old methods that have been forgotten. In 1952 Bailey and associates published their technique [16]. Bailey inserted his index finger through the right atrial appendage to explore manually the right atrium and septal defect. He then used his finger to help suture the right atrial wall to the rim of the defect (Fig 7). The first successful case, a 38-year-old woman, underwent

the operation on January 11, 1952. Mortality among 14 patients with ostium secundum operated on by Bailey was 14.3%, however, in those with ostium primum defect mortality was as high as 45%. Although the technique became somewhat popular [8], significant deformation of the right atrium and potential stenosis of the coronary sinus were drawbacks. Conrad L. Lam of Detroit in 1955 described an improved technique of atrioseptopexy using mobilization of the pulmonary veins to prevent their stenosis and fingerguided placement of the sutures [17]. In 1954 Tyge Sondergaard from Aarhus, Denmark, reported his circumclusion technique for ASD closure (Fig 8). Sondergaard applied his operation clinically on October 26, 1952 [18]. The technique was described as follows [19]: The operative procedure is based on the fact that the atrial septum in the peripheral part consists of the separated walls of the two atria with fat and loose connective tissue between. This cleavage is easy to dissect down to the point where the muscular fibers of the right and left atrial walls join to form the real septum. The cleavage between the right and the left atrium is dissected as far down as possible. The right index finger is inserted into the right atrium through the auricle, and the type, size, and location of the defect is determined. An ordinary blunt probe is bent in a suitable curve, the tip is placed at the upper end of


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Fig 3. Forrest D. Dodrill’s experimental method of “clamp technique” showing instrument (interrupted line for the instrument part behind the heart) applied to atria. Inset a shows sutures in place and clamp released. Inset b shows clamp reapplied and defect of the atrial septum being made. (Reprinted from Dodrill FD, J Thorac Surg; 1949;18:652– 62 [11], with permission.) MISCELLANEOUS

Semiopen Technique

Fig 2. Gordon Murray’s technique of atrial septal defect (ASD) closure. Arrows indicate application of anterior-posterior approximating mattress sutures to close the ASD with invaginated left and right auricles. (Reprinted from Bailey CP, et al, J Thorac Surg; 1953;26: 184 –219 [13], with permission.) the cleavage, and with very slight pressure the tip disappears in the tissue. Almost by its own weight and with the finger in the atrium the probe is guided in the tissue along the lover edge of the defect until the tip appears on the surface at the lover end of the cleavage, in the triangle of fat located between the right and the left atrium and coronary sinus. The tip of the probe is caught with a hemostat. An oiled silk suture no. 10 is tied to the probe, which is then pulled back, currying the suture. The two ends of the suture are tied over a piece of gelatin sponge placed in the bottom of the cleavage. As the suture is gradually pulled tight, the finger in the right atrium feels the defect becoming smaller and smaller and finally close. Then no more tension should be applied.

In 1954 Henry Swan and colleagues wrote: “Experience in this institute with several types of indirect techniques for the closure of ASDs led to realization that, despite good results in the experimental laboratory, indirect methods did not secure complete closure of the large defects frequently occurred clinically” [20]. This notion became generally accepted. Surgeons therefore sought for methods to close the ASD under direct vision.

Robert Edward Gross of Boston in 1953 described the atrial well semiopen technique [21]. An open-bottomed rubber well or cone was sutured to an incision in a clampexteriorized portion of the right atrial wall. When the clamp was released, the blood rose into the well. Through this pool of blood, the surgeon could place sutures under digital control for direct or patch closure of the defect (Fig 9). In 1955 John W. Kirklin at the Mayo Clinic in Rochester, Minnesota, reported 29 cases of isolated ASD operated on using mostly the Gross method without deaths [17].

Fig 4. Henry Swan’s method of invaginating the atrial appendages on plastic buttons. (A) Plastic buttons positioned on the left and right appendages. (B) Inverted appendages approximated against atrial septum to close the atrial septal defect. (Reprinted from Bailey CP, et al, J Thorac Surg; 1953;26:184 –219 [13], with permission.)




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Fig 5. Alvin Merendino’s method of nonsuture closure of atrial septal defect by means of “onlay floating graft” of pedicled pericardium. (A) The index finger of the left hand is inserted through the right auricular appendage and positioned over the auricular septal defect. The heart probe is led through the opening in the left auricular tip until it engages the left index finger against the septal defect. (B) The pericardial pedicle is pulled through the left atrial appendage so that it rests snugly against the defect. The stem of the pericardial pedicle, which projects through the right auricle, is sutured to the auricular wall. Both auricular appendages are sutured. (Reprinted from Kiriluk LB, et al, American College of Surgeons Surgical Forum; 1951;2:199 –204 [14], with permission.)

the hazard of air embolism [24]. Henry Swan used caval inflow occlusion for ASD creation and closure in four successful experiments as early as 1950. Inspired by the experiments of Bigelow, F. John Lewis together with his surgical fellow Mansur Taufic also conducted number of experiments on hypothermia. On September 2, 1952, F. John Lewis with his first assistant Richard Varco and 2 second assistants, Mansur Taufic and Walton Lillehei, operated on a 5-year-old girl with ASD under

Hypothermia and the First Successful Atrial Septal Defect Closure Under Direct Vision Although direct vision operations resulted in much more reliable ASD closure, they had to be accomplished during the short period of inflow occlusion with surface hypothermia. Hypothermia had first been used clinically for the treatment of metastatic carcinoma, by Temple Fay, a neurosurgeon, in July 1936 [22]. Wilfred G. Bigelow, working in Toronto in the late 1940s, conducted a large number of now famous experiments on hypothermia and developed a workable technique of hypothermia in human cardiac surgery [23]. In 1952 Cookson, Neptune, and Bailey reported an attempt to use hypothermia for ASD closure. Unfortunately, the patient died during surgery of ventricular fibrillation. They concluded that open operations for ASD closure were contraindicated because of

Fig 7. Charles Bailey’s method of atrioseptopexy. (A) Digital exploration of septal defect through right auricular appendage. (B) Placement of the first suture of atrioseptopexy. (C) Progressive approximation of invaginated right atrial wall to periphery of septal defect. (D) Completion of atrioseptopexy. (Reprinted from Bailey CP, et al, J Thorac Surg; 1953;26:184 –219 [13], with permission.)


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Fig 6. Antony Hufnagel’s buttons closed about septal defect. (Reprinted from Bailey CP, et al, J Thorac Surg; 1953;26:184 –219 [13], with permission.)

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developing a new operation for d-transposition of the great arteries, now bearing his name (William G. Williams, personal communication, 2002).

Cardiopulmonary Bypass Era

Fig 8. Tyge Sondergaard’s method of circumclusion. (A) Schematic drawing of the septum after suture was placed; the dotted line indicates the developed cleavage. (B) The suture is pulled tight and the defect is closed. (AS ⫽ atrial septum; D ⫽ defect; VS ⫽ ventricular septum.) (Reprinted from Sondergaard T, et al, Acta Chir Scand; 1955;109:188 –96 [19], with permission.)

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general hypothermia and inflow occlusion. Rubberized refrigerated blankets were used for cooling (Fig 10). The body temperature of the patient was 28°C and the duration of inflow occlusion was 5 minutes 30 seconds. This operation was the world’s first successful operation on the open human heart under direct vision [25] and marked the beginning of the open-heart surgery era. Two methods of hypothermia were developed. One was a bathtub method, in which the patient was held immersed in an ice bath until the body temperature dropped to 28° to 30°C, which permitted circulation to be stopped for up to 6 minutes. The other was, a venous blood cooling method, which was developed experimentally by Ite Boerema [26] and Edmond Delorme [27] and used for the first time clinically by Russel Brock and Donald Ross [28]. Brock and Ross initially connected the cooling line to a shunt between the superficial femoral artery and saphenous vein. Because of numerous difficulties, they soon abandoned this method and developed an intrathoracic cannulation technique for cooling. In 1955, for the first time in Europe, Ernst Derra in Du¨ sseldorf, Germany, operated on the patient with the help of surface hypothermia and inflow occlusion. By 1955 he operated on 8 patients with 1 death [29]. Although hypothermia with total inflow occlusion played a crucial role in prompting the era of open-heart surgery, it was too risky and did not allow sufficient time to perform major intracardiac surgery. William T. Mustard from Toronto in the late 1950s operated on a number of patients after previous ASD closure under hypothermic inflow occlusion. In some of them, presumably with inferior sinus defects, he discovered that the inferior caval vein was wrongly diverted into the left atrium. This experience gave him the idea of

John Heysham Gibbon opened the modern era of openheart surgery. Between 1933 and 1953, working in Philadelphia, he constructed the first clinically applicable heartlung machine. The world’s first successful operation with this machine was a direct closure of an ASD in an 18-yearold patient on May 6, 1953. The duration of partial cardiopulmonary bypass (CPB) was 45 minutes; that of total CPB time was 26 minutes. A clamshell incision was used. In 1954, he reported on 4 patents with 3 deaths. In 2 patients correct diagnosis of cardiac anomalies was not established and 1 patient developed cardiac arrest before initiation of CPB [30]. Disappointed with the results, John Gibbon never again attempted to use CPB for open-heart surgery thereafter. It should be mentioned that as early as 1951, Clarence

Fig 9. Method of Robert Gross of placing the sutures in the edges of septal defect, working through atrial well. (1) Atraumatic needle carrying No. 000 Deknatel silk: position of the needle in needle holder. (2) Left index finger exploring the septal defect and identifying its margin. (3) With the right hand, a 10-inch-long needle holder carries the needle into place, grasping 3 or 4 mm of the septal edge. (4) The needle holder is given to an assistant who keeps upward traction on it. The presenting point of the needle is palpated with left index finger and grasped with right angle clamp. (5) Original needle holder is removed, right angle clamp still grasping the needle. (6) Needle recovered. (Reprinted from Gross RE, et al, Surg Obst Gynecol; 1953;96:1–23, with permission.)



6. 7. 8. 9. 10. 11. 12. 13. 14. Fig 10. F. John Lewis and Richard Varco with the rubberized blanket and cooling machine used in the world’s first successful openheart operation. (Courtesy of University of Minnesota Archives.)

15.

Dennis and coworkers working in University of Minnesota attempted to use a pump-oxygenator for ASD closure in a 6-year-old girl [31]. Unfortunately, the operation was not successful. It is of interest that in 1954 Walton C. Lillehei using the method of cross-circulation achieved better results than those who tried to use CPB during the same period of time. However, CPB technique was further refined, so that by the late 1960 almost all surgeons used CPB for ASD surgery. Operations on the atrial septum paved a road to modern open-heart surgery. Fifty years passed. Operations on the open heart became routine. These operations are performed daily all over the world. Let us pay respect to the courage and ingenuity of surgeons and patients who lived and worked during the time when closure of the ASD was a major challenge.

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18. 19. 20. 21. 22. 23. 24.

We are grateful to Anne Gale and Annette Gaussmann for technical assistance. We are indebted to Hans G. Borst, C. Rollins Hanlon, K. Alvun Merendino, Mansur Taufic, and William G. Williams for their most valuable suggestions.

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Surgery for atrial septal defect: from the first experiments to clinical practice Vladimir V. Alexi-Meskishvili and Igor E. Konstantinov Ann Thorac Surg 2003;76:322-327 Updated Information & Services

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