Physiological rationale for a bidirectional cavopulmonary shunt

J

THoRAc CARDIOVASC SURG

90:391-398, 1985

Physiological rationale for a bidirectional cavopulmonary shunt A versatile complement to the Fontan principle The original Fontan procedure included a classic superior vena cava-to-right pulmonary artery (Glenn) shunt Subsequent experience demonstrated that this anastomosis was not essential and was an unnecessary commitment of the larger right pulmonary circulation to the smaller blood volume of the superior vena caval return. With application of the Fontan principle to more complex cardiac malformations, there has been a reconsiderationof possible benefitsof a cavopulmonary shunt in selected patients.A modified shunt from the divided end of the superior venacava to the side of the undivided right pulmonary artery uti6zed in 21 patients is described. This shunt is designed to allow bidirectional pulmonary arterial distributionof both superior venacavalinflow andright atrial outflowafter completion of the Fontan procedure. Twelve patients had the bidirectional shunt performed prior to a Fontan operation; five of these had a subsequent atriopulmonary connection and seven await operation. Eight patients had construction of this shunt at the time of their Fontan procedure. One patient had a bidirectional shunt constructed foUowing atriopulmonary anastomosis to help relieve right atrial outflow obstruction. Two patients with univentricular heart undergoing simultaneous Fontan procedure and a bidirectional shunt died while in the hospital. The remaining 19 patients have been foUowed up for 2 monthsto 9 years with one late suddendeath at 9 years. There have been no bidirectionalcavopulmonary shunt failures, stenoses, kinks, or recognized pulmonary arteriovenous malformations. Postoperatively, eight patients had assessment of pulmonary distribution of shunt blood flow by angiography. Seven of these patients were also evaluated by radionuclide angiography. Superior vena caval blood flow via the bidirectional cavopulmonary shunt tended to be greater to the right lung,but bilateral pulmonaryflowwas documented in all but one patient After Fontan operation, six of seven patients tested also demonstrated bilateraldistributionof atriopulmonary flow. We concluded from our experiencethat this modified shunt (1) provides exceUent relief of cyanosis, (2) allows bidirectional pulmonary distribution of both superior vena caval return and also the right atrial blood flow after atriopulmonary connection, and (3) may be done before, with, or after a Footan procedure and is compatible with all currently recommended modifications. Perioperative hemodynamic adjustments to the Fontan procedure may be improved by reducing atrial volume, and this may also be of potential benefit in the long-term adaptation to Fontan physiology by minimizing atrial distention.

Richard A. Hopkins, M.D., Brenda E. Armstrong, M.D., Gerald A. Serwer, M.D., Richard J. Peterson, M.D., and H. Newland Oldham, Jr., M.D., Durham. N. C.

Diversion of blood from the superior vena cava (SYC) to the right pulmonary artery (RPA) was

From the Departments of Surgery and Pediatrics, Duke University Medical Center, Durham, N. C. Received for publication June 26, 1984. Accepted for publication Dec. 10, 1984. Address for reprints: H. Newland Oldham, Jr., M.D., Department of Surgery, P.O. Box 3043, Duke University Medical Center, Durham, N. C. 27710.

performed experimentally by Carlon, Mondini, and de Marchi! in 1951, and additional investigators confirmed the physiological feasibility of this technique." 3 This procedure was followed by clinical use of the shunt by Glenn" and by Bakulev and Kolesnikov' to increase pulmonary blood flow as palliation for cyanotic congenital cardiac lesions. Experimental models of complete bypass of the right ventricle usually included the SYCRPA anastomosis as did Fontan's first patient undergoing successful correction of tricuspid atresia.':" Subsequent modifications of the Fontan procedure, however, 391

392 Hopkins et al.

Fig. 1. A bidirectional shunt with anastomosis of the divided SVC to the undivided RPA artery.

did not typically include the cavopulmonary shunt.":" The realization that it was not an essential component of the Fontan procedure and concerns about its role as a palliative shunt resulted in a dramatic decrease in the use of the Glenn shunt. 16-24 Creation of a bidirectional shunt, omitting ligation of the proximal RPA, was first investigated experimentally by Haller and associates" in 1966 and used clinically by Azzolina, Eufrate, and Pensa in 1972. Isolated reports of the use of such a shunt were followed by the recent description by Glenn of Abrams' larger experience. 26-29 This report discusses the physiological rationale for the bidirectional modification of the Glenn shunt and its use since 1976 as a complement to the Fontan principle for 21 patients with tricuspid atresia or univentricular heart. Methods Patients. Twenty-one patients underwent creation of a bidirectional shunt because of increasing cyanosis. The clinical courses, serial hemodynamic data, angiograms, cardiac catheterization data, and radionuclide angiocardiograms (RNAs) were reviewed to assess shunt function, bidirectionality of shunt flow, and hemodynamic benefit. Twelve patients had tricuspid atresia, 11 of whom had

The Journal of Thoracic and Cardiovascular Surgery

normally related great vessels. Nine patients had a univentricular heart, and two of these also had pulmonary atresia. Sixteen patients had undergone a variety of previous systemic arterial-to-pulmonary arterial shunts between 1 and 30 months of age, including three bilateral shunts, one Waterston shunt, and one central shunt. Twelve patients had construction of the bidirectional SVC-RPA shunt as a staging operation at an average age of 3V2 years (range 1 to 8 years), and five of these had a subsequent Fontan operation after an interval of 1 to 5 years. Eight patients received a shunt at the same operation as the Fontan procedure between 20 months and 15 years of age. One patient had a shunt constructed 4 months after an atriopulmonary connection because of restrictive right atrial outflow. Surgical technique. An anterolateral thoracotomy was used in the 12 patients who received a staging shunt. SVC pressures rose to 25 to 50 mm Hg with crossclamping, and in five patients a temporary heparinbonded shunt was inserted between the SVC and the right atrium. This technique effectively decompressed the SVC but also made exposure difficult. In three patients the head of the table was elevated while the SVC was briefly clamped without decompression, and a continuous absorbable suture was used to facilitate performance of a rapid anastomosis. Cardiopulmonary bypass was used in the remaining four patients having a staging shunt because of inadequate oxygenation after test occlusion of the RPA. A median sternotomy was utilized in the eight patients undergoing simultaneous shunt construction and Fontan procedure. The SVC cannula for cardiopulmonary bypass was inserted at the base of the innominate vein. Interrupted sutures were used for the anastomosis in 10 patients, and in 11 patients continuous monofilament absorbable suture material was used. Evaluation of pulmonary blood flow. To quantify relative distribution of blood flow to right and left pulmonary vascular beds, first-pass RNA studies were performed on seven patients following bidirectional Glenn procedures. Three patients had serial RNA studies performed during a 3Vz year period. Four patients underwent a modified Fontan procedure prior to their RNA study. RNA studies were performed by bolus injection of technetium 99m pertechnetate (0.03 mCijkg to a maximum of 10 mCi). A multicrystal gamma camera was used and data were recorded over the anterior precordium and both lung fields at 25 msec intervals for a 1 minute period." Separate regions of interest were zoned over the right and left lung images, and timeactivity curves were then generated for right lung, left

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September, 1985

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lung, and combined regions. Postoperative arteriograms were obtained in eight patients; three had simultaneous Fontan procedures and five had staged procedures. Results Clinical. All 12 patients undergoing a staging shunt survived and had improved oxygen saturation. Five of these had a subsequent atriopulmonary anastomosis performed, with no deaths, after an average interval of 3 years. The remaining seven have had excellent palliation and are awaiting elective Fontan operation. Five of eight patients having simultaneous procedures did well and were followed up to 9 years. Two patients with univentricular heart died perioperatively, one of sepsis and renal failure on the thirty-second postoperative day. The second death occurred in a 20-month-old child with intractable arrhythmias on the first postoperative day. One patient with univentricular heart died suddenly 9 years after operation. One patient received a shunt 4 months after a Fontan repair. This 3-year-old girl with small pulmonary arteries had undergone a direct atriopulmonary anastomosis for tricuspid atresia. Her postoperative course was characterized by fluid retention, and repeat catheterization demonstrated elevated right atrial pressure. At reoperation, the anastomosis between the right atrium and RPA was enlarged, and construction of a bidirectional Glenn shunt resolved the problem of fluid retention. The patients have been followed up for 2 months to 9 years (mean = 37 ± 36 months). SVC syndrome did not occur, and early postoperative SVC pressure was 17.3 ± 4.5 mm Hg ± standard deviation). There were no shunt failures, stenoses, kinks, pulmonary vascular obstructive problems, or recognized pulmonary arteriovenous malformations, and all patients had improved oxygen saturation. Complications occurred in three patients. Two patients experienced a transient neurological deficit on the second postoperative day. These were believed to be embolic, and both cleared completely prior to the patients' discharge from the hospital. Temporary shunt decompression of the SVC had been employed in one patient and cardiopulmonary bypass in the other. One patient had a persistent right pleural effusion postoperatively, which cleared after diuretic treatment. This same patient had proximal RPA stenosis because of a previous pulmonary artery band 'and was the only patient without demonstrable bidirectional shunt flow. Flow studies. After the shunt was created, seven of eight patients tested had bidirectional SVC to pulmonary artery blood flow. This was demonstrated qualitatively by angiography in seven of eight patients and

Fig. 2. Angiogram with SVC injection demonstrating bilateral pulmonary blood flow, via the shunt, prior to a completion Fontan procedure for tricuspid atresia,

quantitatively by RNA studies in six of seven patients tested (Figs. 1 and 2). In the single patient with an RPA stenosis, there was no left pulmonary artery blood flow from an SVC injection. Following atriopulmonary connection, right atrial flow to the pulmonary arteries was difficult to quantitate by RNA in three patients because of prolonged emptying time of the atrium, but in two patients the inferior vena cava clearly contributed a large volume of blood to RPA blood flow. Six patients had bilateral pulmonary arterial flow from the right atrium documented by angiography (Figs. 3 through 5). One patient had conduit malfunction 7 years after a simultaneous Fontan procedure and bidirectional shunt. RNA studies demonstrated slowed atrial emptying, and the primary source of pulmonary blood flow was derived from the shunt. The conduit containing a destroyed valve and thick peel was removed at reoperation, and a nonvalved reconstruction was performed. Right atrial emptying was restored, and RNAs demonstrated bilateral flow of IVC blood with 60% to the right lung and 40% to the left lung (Fig. 6). Discussion Status of the standard cavopulmonary shunt. The original cavopulmonary, or Glenn, shunt has been used to palliate a variety of congenital heart lesions with excellent relief of cyanosis. Glenn was able to report a collected series of 537 cases by 1966,5-31 The results were considered good, with mortality rates less than 10% for children. In the series of 33 patients with tricuspid atresia or univentricular heart reported by Glenn and associates," there were no deaths. Advantages of this systemic vein-pulmonary artery shunt

3 94 Hopkins et al.

The Journal of Thoracic and Cardiovascular Surgery

Fig. 3. A bidirectional shunt combined with a direct atriopulmonary anastomosis.

over the systemic artery-pulmonary artery shunts were recognized as (1) no increase in workload of the left side of the heart, (2) no late pulmonary vascular obstructive disease in the right lung, (3) shunting of venous blood rather than an arteriovenous admixture, and (4) decrease in the volume of blood flow through the right atrium." Despite enthusiastic reports, difficulties were recognized after the Glenn shunt: (1) development of collateral venous channels from Sye to lye, (2) pulmonary arteriovenous fistulas, (3) abnormalities in regional pulmonary perfusion, and (4) difficulty in dismantling the shunt. 16·24. 33-36 Abnormal distribution of pulmonary perfusion has also been described following the Fontan procedure and may be related to lack of normal pulsatile blood flow." Although the Glenn shunt was noted to enlarge with growth of the child, late clinical deterioration was observed. This loss of palliation, however, was primarily due to the growth and increased activity of the patients and less often from anatomic changes, pulmonary vascular disease, or malfunction of the shunt.!" 20. 24. 29, 38 Trusler and Williams" found the Glenn shunt to have the best long-term palliation for tricuspid atresia when used as the primary shunt in older children or as a secondary procedure following an initial arterial shunt. Thus, although the classic Glenn shunt was recognized

Fig. 4. Angiogram with Sye injection (top) and lye injection (bottom) 2 years after simultaneous bidirectional shunt and Fontan procedure for univentricular hearts demonstrating bilateral pulmonary distribution of both Sv'C and lye flow. RNA demonstrated 70% of sye flow distributed to the right lung.

as effective for long-term palliation, the success of the Fontan approach in treatment of tricuspid atresia and univentricular heart further decreased the need for permanent shunt palliation and led to the concept of shunt staging toward an ultimate Fontan repair. 18. 39·41 Role of the cavopulmonary shunt and the Fontan procedure. Although Fontan included a Glenn shunt in his initial patient, only four of the 100 patients reported in 1983 had the cavopulmonary anastomosis as a simultaneous part of the procedure, whereas 17 had previously constructed Glenn shunts. 10. 14 Several other reports confirmed the finding that the cavopulmonary anastomosis was unnecessary in the right ventricular exclusion approach. II. 12.29.42.43 The original concept that the cavopulmonary anastomosis decreased the right

Volume 90 Number 3 September, 1985

Bidirectional cavopulmonary shunt

Right lung flow = 52%

Left lung flow = 48%

39 5

After Fontolt Procedure November 20, 1979

4669

II

Counts Before Revision June 15, 1983

o

15

0

15

Counts

Time (secondsl

Fig. 5. RNA histogram of lung counts following lye injection after a Fontan operation with previously constructed bidirectional shunt demonstrating relatively equal pulmonary flow distribution via the atriopulmonary connection. atrial volume and therefore reduced postoperative rightsided failure has, however, been supported by several articles. Pennington and associates" reported that all patients whounderwent a standard Glenn shunt prior to the Fontan operation not only survived the procedure but also had minimal postoperative hemodynamic instability without signs of congestive right-sided pleural effusion, ascites, or renal impairment. Other series not including a cavopulmonary shunt have reported an incidence of postoperative renal insufficiency as high as 30%.44-46 Deleon and colleagues" evaluated 27 patients following the Fontan operation and reported that those with an established Glenn shunt had less postoperative fluid retention and a shorter hospital stay. He did not, however, fmd any immediate hemodynamic advantage to the simultaneous use of the Glenn shunt with the atriopulmonary operation. The postoperative hemodynamic status is related primarily to left ventricular function with less documentation of a clear relationship to right atrial function." 49 The presence of the Glenn shunt was reported to be lifesaving in two patients in whom complications led to ineffective right atrial-left pulmonary arterial bloodflow." Other reports of patient survival becauseof Glenn shunt function after thrombosis of the right atrial-pulmonary arterial conduit have been published." 50 Long-termbenefitof a cavopulmonary anastomosis in conjunction with a Fontan correction has yet to be demonstrated." The left ventriclein these patients must supply the energy for both the systemic and pulmonary circulations, and patients with tricuspid atresia and univentricular heart develop left ventricular dysfunction because of volume overload and hypoxemia.v" Any significant reduction in left ventricular work may contribute to prolonged functionalimprovement and survival. Patients having a palliative Glenn shunt have been

After Fonton Revision June 27, 1983

5

Time (seconds)

10

Fig. 6. RNA histograms of right atrial counts following lye injections. These demonstrate blood transit through the right atrium into thepulmonary circuit. Thetopcurve was obtained shortly after a simultaneous Fontan procedure with bidirectional shunt. The middle curve was obtained almost 7 years later when conduit obstruction occurred and demonstrates prolonged right atrial emptying. The lower curve shows improved atrial emptying following conduit removal and autologous revision oftheatriopulmonary anastomosis. During the time of conduit malfunction, bidirectional shunt provided most of the pulmonary blood flow. shown to have better ventricular function, perhaps related to reduced volumeoverload. 53 This concern over ventricular function has also led to a consideration of application of the Fontan operation at an earlier age.54 Postoperative exercise RNA studies of Fontan patients have demonstrated increased cardiac output with depressed ejection fraction, left ventricular dilatation, and compensatory elevation of heart rate. 55 The roleof right atrial function is not clearly defined, but it is possible that dysfunction resultingfrom volumeoverload may contribute to late right-sided failure, especially when the right atrium must handle the entire venous return.56 These findings have led to a reconsideration of the role of the Glenn procedure as a staging shunt.57 Fontan" recommended a simultaneous cavopulmonary anastomosis to unload the right atrium if right atrial distention is severe at the conclusion of his procedure. Thus, it seems that even the classic cavopulmonary shunt has some advantages, both real and theoretical, in conjunction with the Fontan procedure. The primary objection is the dedication of one third of the venous return to the larger vascular bed of the right lung. This

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396 Hopkins et al.

mismatch can be avoided by utilizing the bidirectional shunt. Bidirectional cavopulmonary shunt. The bidirectional SVC-RPA shunt was studied experimentally by Haller and associates" during the developmental stages of right heart bypass research. In 1972 Azzolina and colleagues" reported on nine patients having such an anastomosis, with a 33% mortality rate. Glenn" referenced an unpublished British series of 26 such patients by Abrams and reported no deaths or significant complications. The advantages of this shunt are (1) that it does not sacrifice the continuity between the RPA and main pulmonary artery, (2) that bilateral pulmonary blood flow can occur from the SVC, and (3) that when used with an atriopulmonary anastomosis, bilateral pulmonary flow can occur from both SVC and IVC return. This report describes the application of the bidirectional SVC-RPA shunt as a complement to the Fontan principle. The shunt is a useful palliative procedure in staging to an ultimate Fontan operation. It may be performed as a simultaneous component of a Fontan procedure and is beneficial if postoperative obstruction of the atriopulmonary connection occurs. It certainly provides excellent relief of cyanosis, as does the traditional Glenn shunt, and it is followed by good intermediate-term palliation in complex conditions that are less than ideal for a Fontan procedure. An important goal in these patients is reduction of the number of staging operations prior to a Fontan procedure. Small infants with cyanosis could first have a systemic artery-pulmonary artery shunt on the side opposite the SVC, followed later by a bidirectional cavopulmonary shunt. Older children could undergo staging with an initial bidirectional cavopulmonary shunt. This shunt does not jeopardize pulmonary vascular resistance or left ventricular function. The shunt may be created before, with, or after all currently recommended Fontan modifications. It is a simple and safe shunt that retains some anastomotic growth potential and requires no revision at a subsequent Fontan operation. It allows bidirectional pulmonary distribution of both SVC and IVC blood flow and avoids committing the larger right lung to the smaller SVC return. The shunt may also be a useful adjunct in corrective operations on selected patients at an earlier age, especially when combined with the technique of direct atriopulmonary anastomosis. If not required prior to a Fontan procedure, there is no documentation of late benefit from a simultaneous Fontan and bidirectional shunt operation for either ideal or less than ideal candidates. In this situation it may, however, minimize perioperative hemodynamic adjustments by reducing

atrial volume and could potentially improve the longterm functional results achieved by the Fontan repair. REFERENCES

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