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Ann Thorac Surg 2005;79:1656-1660
© 2005 The Society of Thoracic Surgeons

Original articles: Cardiovascular

Late Left Pulmonary Artery Stenosis After the Norwood Procedure is Prevented by a Modification in Shunt Construction

David P. Bichell, MDa,*, John J. Lamberti, MDd, Glenn J. Pelletier, MDa, Cynthia Hoecker, MDc, Mark W. Cocalis, MDb, Frank F. Ing, MDb, Richard A. Jensen, MDb

a Division of Cardiovascular Surgery, Children's Hospital San Diego, San Diego, California
b Division of Cardiology, Children's Hospital San Diego, San Diego, California
c Division of Emergency Medicine, Children's Hospital San Diego, San Diego, California
d Division of Cardiac Surgery, Oakland Children's Hospital, Oakland, California

Accepted for publication November 10, 2004.

* Address reprint requests to Dr Bichell, Division of Cardiovascular Surgery, Children's Hospital, San Diego, 3030 Children's Way, Suite 202, San Diego, CA 92123 (E-mail: dbichell{at}chsd.org).


   Abstract
Top
 Abstract
Introduction
 Patients and Methods
 Results
 Comment
 References
 
BACKGROUND: Late left pulmonary artery (LPA) stenosis occurs commonly after the Norwood procedure, and complicates subsequent stages. Compression by the neoaorta and ductal stump may favor flow into the right pulmonary artery, resulting in LPA hypoplasia. We hypothesize that an early compromise of LPA flow contributes to late LPA stenosis, and have modified our shunt to compensate.

METHODS: We reviewed 34 consecutive neonates undergoing the Norwood procedure between 1999 and 2002, and morphometric data from angiograms obtained before the bidirectional cavopulmonary anastomosis (BDCPA). The Norwood technique included an autologous arch reconstruction with or without augmentation, and a polytetrafluoroethylene Blalock-Taussig shunt (BTS). Starting February 2001, the distal shunt was modified from an end-to-side construction to an oblique anastomosis directed into the retroaortic LPA.

RESULTS: Norwood survival was 82%. LPA stenosis required plasty in 10 of 13 (77%) premodification survivors, and in 2 of 9 (22%) postmodification (p = 0.027). Bypass time was 151 ± 65 minutes with LPA plasty versus 95 ± 50 minutes without. Mortality (15% vs 0%), hospital stay (25 ± 35 vs 9 ± 6 days), and incidence of subsequent interventions were correspondingly higher with LPA stenosis. Ten of 13 patients (77%) with a BTS insertion point outside the central region of the pulmonary artery required LPA plasty, versus 2 of 9 (22%) with an insertion nearer to the center (p = 0.027).

CONCLUSIONS: An oblique distal BTS anastomosis directed leftward onto the retroaortic pulmonary artery at the time of the Norwood procedure may prevent late LPA stenosis and its attendant morbidity.


   Introduction
Top
 Abstract
 Introduction
Patients and Methods
 Results
 Comment
 References
 
Left pulmonary artery stenosis and hypoplasia often complicates the second stage palliation after a Norwood procedure [1, 2]. Even as Norwood stage I survival continues to improve, late mortality remains significant, often lacking consistent explanation [3]. Contributing to late morbidity and mortality is pulmonary artery stenosis and hypoplasia that can advance in severity with time. Mechanisms contributing to pulmonary artery compromise after the Norwood procedure are multiple, and include narrowing at the ductal remnant site, extrinsic compression by the neoaorta or the ductal stump, and distortion by the modified Blalock-Taussig shunt (BTS) insertion. Particularly at risk for these influences is the retroaortic segment of the common pulmonary artery and the left pulmonary artery (LPA). Compromised early flow to the LPA may contribute to LPA hypoplasia over time, which can increase morbidity of the subsequent bidirectional cavopulmonary anastomosis (BDCPA) and Fontan procedures. Flow dynamics models of BDCPA and Fontan pathways point to the importance of pulmonary artery geometry as a determinant of pulmonary flow efficiency, early and late outcome [4–6]. Clinical studies show that abnormal pulmonary artery architecture is an independent risk factor predicting poor Fontan outcome [7–9]. Infants undergoing the Norwood stage I procedure are predisposed to a variety of risks affecting the success of the subsequent stages along the Fontan pathway, notably including pulmonary artery distortion and hypoplasia requiring a late pulmonary artery reconstruction in greater than 50% of cases [10, 11]. We adopted a modified strategy of shunt construction at the time of the Norwood procedure aimed at compensating for the factors that diminish flow to the LPA, and present data comparing pre and postmodification outcomes.


   Patients and Methods
Top
 Abstract
 Introduction
 Patients and Methods
Results
 Comment
 References
 
We retrospectively examined the courses of 34 consecutive neonates undergoing the Norwood procedure at Children's Hospital, San Diego between January 1999 and May 2002. Patient chart review for the study met all content, ethical, finacial and operational criteria of the Children's Hospital and Health Center IRB (CHHC #11106). Anatomic subgroups included Shones or hypoplastic left heart syndrome in 11 patients (50%), double outlet right ventricle with hypoplastic left ventricle in 3 (14%), double inlet left ventricle in 3 (14%), unbalanced atrioventricular septal defect in 2 (9%), tricuspid atresia with transposed great arteries in 2 (9%), and transposition of the great arteries with straddling mitral valve in 1 (5%). All patients had arch hypoplasia and coarctation of the aorta requiring a full arch reconstruction. Excluded were single ventricle patients requiring Damus-Kaye Stansel anastomosis and a shunt without a distal arch reconstruction. Thirty-two of 34 Norwood procedures were performed by a single surgeon. Median age at the time of the Norwood operation was 9 days (mean 11 days). Associated cardiac lesions included: pulmonary artery sling in 1 patient, total anomalous pulmonary venous connection in 1, and partial anomalous pulmonary venous connection in 2. The Norwood technique included an autologous arch reconstruction with or without patch augmentation, similar to that described by Frasier and Mee [12], and a 3.0- or 3.5-mm polytetrafluoroethylene shunt. Four of 34 neoaortic constructions were augmented with homograft material, 17 with a pericardial patch, and 11 without any augmentative material. No RV-PA conduits were performed during the study period. The operative strategy included the construction of the proximal BTS anastomosis before the initiation of cardiopulmonary bypass, initial cannulation of the ascending aorta, main pulmonary artery or both, with a transfer of the arterial cannula to the BTS after cooling to 15 to 18°C and at the initiation of low-flow bypass for the arch reconstruction. A strategy of aortic cannulation at the initiation of cooling was adopted for the hypothetical advantage to cerebral protection that evenly distributed bilateral early brain cooling might impart. Transcranial saturations were monitored by INVOS near infrared spectroscopy (Somanetics Corp., Troy, MI) throughout the conduct of the procedure. The cardiopulmonary bypass strategy included continuous selective cerebral perfusion at 10 to 30 mL · kg–1 · min–1 during the arch reconstruction, in a manner similar to that described by Pigula and colleagues [13] The cerebral perfusion rate was titrated to maintain transcranial cerebral saturations. The rate of infusion that reliably kept cerebral saturations from falling was determined to be 10 mL · kg–1 · min–1, and was the rate most commonly employed. Empiric higher rates of infusion were avoided to minimize the risk of cerebral edema for an unproven benefit. Branch pulmonary arteries were mobilized fully and the transverse sinus opened to allow their caudad displacement away from the confined retroaortic space. The main pulmonary artery was typically transected obliquely to create a sufficient flap for a generous primary closure with live autologous tissue. The rightward-based flap corresponded to the area of the MPA adjacent to the ascending aorta, and the resultant deficit in the MPA received the ascending aortic anastomosis. The transected pulmonary artery confluence was closed primarily in 29 patients, with pericardial augmentation in 4 and with resection and end-to-end reconstruction in 1. The BTS was constructed of stretch GoreTex (W.L. Gore & Associates, Flagstaff, AZ) conduit material measuring 3.0 mm (19 patients), or 3.5 mm (15 patients) in diameter, using 7-0 polypropylene suture. Starting February 2001, the distal shunt construction was modified from an end-to-side anastomosis at the proximal right pulmonary artery, to a long, oblique anastomosis directed leftward onto the retroaortic portion of the pulmonary artery confluence (Fig 1). Angiograms obtained at 3 to 4 months of age, before the BDCPA, were analyzed with particular attention to LPA morphology.



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  		Fig 1. Drawing representing the insertion point of a BTS onto the pulmonary artery premodification (A), and postmodification, with an oblique anastomosis to direct pulmonary blood flow preferentially into the retroaortic pulmonary artery (B). (BTS = Blalock-Taussig shunt.)

 
At the second stage palliation, the bidirectional cavopulmonary anastomosis was constructed with a full mobilization of the superior vena cava, and a posterolateral caval spatulation to permit a long direct anastomosis onto the central pulmonary arteriotomy. Occasionally, the central most hood of the BTS was retained and incorporated into the cavopulmonary anastomosis. Patch pulmonary arterioplasties were constructed where judged necessary by preoperative angiographic appearance and by intraoperative measurement by calibrated probe.

Shunt and Pulmonary Artery Morphometry
Anterioposterior angiograms with BTS injection and opacification of the branch PAs were examined to analyze the position of the insertion of the shunt with respect to the interhilar midpoint. The interhilar midpoint was defined as the midpoint between the left and right upper lobe branch PAs, and the shunt insertion point was defined as the distance between shunt insertion and interhilar midpoint. The ratio of the distance from the interhilar midpoint to the shunt insertion point as a fraction of the total distance from the midpoint to the right hilum was determined as a measure of the degree of displacement of the shunt rightward from the center (Fig 2).



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  		Fig 2. The relative position of the shunt insertion point onto the common pulmonary artery after the Norwood stage I procedure. The interhilar midpoint (M) is defined as the midpoint between right (R) and left upper (L) pulmonary arteries. A ratio of the distance from the interhilar midpoint to the shunt insertion point (M to B), divided by the distance from the interhilar midpoint to the right hilum (M to R) expresses the degree of rightward displacement of the shunt from the midpoint as a percentage (M to B)/(M to R)x100.

 
LPA/RPA Ratio
The relative diameters of the LPA and RPA were measured from pre-BDCPA angiograms on anteroposterior view. The ratio compares the minimal LPA diameter to the minimum RPA diameter, expressed as a percent.

Data Analysis
The two-tailed Fisher's exact test was applied to test the significance of association between the incidence of LPA plasty, midpoint-to-shunt distance ratios and LPA/RPA diameter ratios. A p value of less than 0.05 was set as the level of significance. Continuous data were analyzed using unpaired Student's t test, with significance set as p less than 0.05.


   Results
Top
 Abstract
 Introduction
 Patients and Methods
 Results
Comment
 References
 
Twenty-nine of 34 patients (85%) survived beyond 30 days, and one died in-house after 30 days of complications arising from preexisting renal hypoplasia, making overall early survival 82.3%. Three interstage deaths occurred at home among the surviving 28 patients, and 1 died at an outside institution after a diagnostic pre-BDCPA catheterization but before surgery, bringing the survival beyond BDCPA to 71%. Two patients died after the BDCPA, both of whom had severe long-segment LPA stenosis. Two patients who survived beyond the BDCPA had unavailable pre-BDCPA catheterization data for study, leaving 22 patients with both surgical and catheterization data for analysis.

Shunt Size and Pulmonary Blood Flow
At 48 hours postoperatively following the Norwood procedure, neither the FiO2 (31 ± 22.2 vs 31 ± 9.7), arterial pO2 (38 ± 4.6 vs 36 ± 3.7), nor oximetric arterial saturation (75 ± 5.8 vs 74 ± 3.9) was significantly different when a 3.5 versus 3.0 mm BTS was used (p > 0.1). Similarly, the final arterial blood gas determination of the hospitalization demonstrated no significant difference in FiO2 (27 ± 12.9 vs 24 ± 5.6), arterial pO2 (42 ± 3.0 vs 41 ± 3.6), or oximetric O2 saturation (80 ± 3.5 vs 78 ± 6.3) whether a 3.5-mm or 3.0-mm shunt was constructed (p > 0.1).

Nine patients in the series had 3.5-mm shunts and the remaining 13 had 3.0-mm shunts. Larger shunts were used preferentially earlier in the series (6 of 13 premodification patients), and less frequently postmodification (3 of 9 patients), as the oblique postmodification anastomoses with a 3.0-mm shunt was clinically observed to deliver similar pulmonary blood flow as less broadly anastomosed 3.5-mm shunt.

Five patients with 3.5-mm shunts required pulmonary arterioplasty at the time of BDCPA and 6 of the 13 with smaller shunts were among those needing plasties and no significant correlation between shunt size and the incidence of treatable LPA stenosis can be concluded (p > 0.1)

Angiographic and Clinical LPA Stenosis
In the premodification group, the LPA/RPA ratio was less than or equal to 0.5 in 7 of 13 patients (56%), whereas an LPA measuring less than or equal to 50% of RPA diameter occurred in none of the 9 postmodification patients (p = 0.017). LPA/RPA diameter correlated significantly with proceeding to LPA plasty at the time of BDCPA construction, with 100% of patients with LPA/RPA less than 50% undergoing LPA plasty, as compared with 36% of those with LPA/RPA greater than or equal to 50% (p = 0.013). Of note, 1 patient had relative RPA stenosis with an LPA/RPA ratio of 125%, and requiring RPA patch plasty at the time of BDCPA construction. The pulmonary arterioplasties performed were indicated by subjective criteria at the time of surgery and by preoperative angiographic appearance, and ranged from short patches at the ductal remnant site to long segment reconstructions of diffusely hypoplastic arteries. Quantitating the indications and extent of reconstructions is not possible, although examined retrospectively, an angiographic LPA/RPA ratio less than 50% correlated highly with the judgment to perform a patch plasty.

Shunt Position and LPA Stenosis
Ten of 13 patients (77%) with a BTS insertion point greater than 33% rightward of the interhilar midpoint required LPA plasty, whereas 2 of 9 (22%) with an insertion nearer to the midpoint required LPA plasty (p = 0.027). Correspondingly, LPA stenosis requiring intervention was present in 10 of 13 surviving patients (77%) who had undergone the Norwood procedure before the BTS modification, and in 2 of 9 following the advent of the shunt modification (22%) (p = 0.027; Fig 3). Mean bypass time at BDCPA construction was 151 ± 65 minutes when LPA plasty was required versus 95 ± 50 minutes without (p = 0.037). Bidirectional cavopulmonary anastomosis mortality was 15% versus 0% (p = 0.48), and postoperative hospital length of stay was 25 ± 34 versus 9 ± 6 days when LPA plasty was required versus not required, respectively (p = 0.17). Three patients required the placement of an additional aortopulmonary shunt at the time of BDCPA for insufficient pulmonary blood flow or to encourage development of a unilateral hypoplastic pulmonary artery. Two had LPA stenosis plastied concurrently, the other had important RPA stenosis and hypoplasia. Two patients underwent LPA stent placement, one intraoperatively and one after BDCPA construction. One patient required a BDCPA takedown within 24 hours of its construction owing to unacceptable upper compartment pressure and hypoxia. The patients requiring stent placement, BDCPA takedown and 2 of 3 adjunctive BTS recipients had their Norwood procedures before the advent of the shunt modification and had significant LPA stenosis. Both post-BDCPA deaths occurred in patients who received their Norwood palliation before the advent of the shunt modification and both had important LPA stenosis. Although length of stay and mortality comparisons between the groups did not achieve statistical significance, the trend is suggestive that fewer complications result and fewer LPA procedures are necessary with the modification, apparently owing to a lower incidence of retroaortic LPA stenosis and its sequelae.



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  		Fig 3. Representative pre-BDCPA pulmonary angiograms premodification (A), showing a shunt insertion point 77% rightward of the intrahilar midpoint and significant LPA stenosis, and postmodification (B), showing a shunt insertion point 6% rightward of the interhilar midpoint, and no retroaortic LPA stenosis. (BDCPA = bidirectional cavopulmonary anastomosis; LPA = left pulmonary artery.)

 

   Comment
Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
References
 
Compromised early branch pulmonary artery blood flow may contribute to generalized hypoplasia of the pulmonary artery. It has been shown in a canine model that systemic arteries adjust their diameter up or down through proliferative growth or hypoplasia to hold shear stress constant under chronic conditions of diminished or excessive flow [14, 15]. These principles likely similarly apply to pulmonary arteries with asymmetric blood flow. Diminished flow through the partially obstructed neonatal left pulmonary artery may contribute to generalized left pulmonary artery hypoplasia and enlargement on the right, where the balance of flow may be excessive. When passive pulmonary artery flow is introduced at the time of BDCPA construction, compromised LPA flow may persist, based on the pre-BDCPA growth asymmetry, resulting in suboptimal total pulmonary blood flow and distribution.

Data from this retrospective cohort study suggest that the multiple retroaortic forces that commonly produce LPA stenosis and its attendant long-term complications can be neutralized by delivering shunt flow preferentially leftward into the retroaortic pulmonary artery. A significant reduction in angiographic and clinically relevant LPA stenosis is demonstrated coincident with the advent of the shunt modification. Significant consequent reductions in cardiopulmonary bypass time, hospital length of stay, and the need for concurrent LPA plasty at the time of BDCPA are also demonstrated. Trends suggest additional corresponding reductions in mortality and late interventions such as pulmonary artery stent placement.

Numerous forces act to compromise leftward pulmonary artery blood flow after the Norwood procedure, including ductal stump contraction, buckling or kinking of the bulbous MPA stump and ductal remnant within the confined space behind the neoaortic root, shunt and pulmonary artery distortion. Though the shunt modification in this series appears to neutralize some of the forces leading to pulmonary artery compromise, the study is limited in elucidating the interplay of mechanisms that cause the compromise.

Study patients were too few for statistical analysis, but all 3 patients who had a patch closure of the MPA had subsequent LPA stenosis requiring intervention, suggesting that a patch closure of the MPA stump is not necessarily protective of LPA compromise. A strategy of primary MPA closure with a generous rightward-based flap has been adopted to minimize the bulk of the retroaortic segment of pulmonary artery and to provide any advantage that living autologous tissue confers to pulmonary artery growth. No association can be made by this study between the method of MPA stump closure and the subsequent incidence of pulmonary artery stenosis.

There is no demonstrable difference in pulmonary blood flow delivered by a 3.0-mm versus 3.5-mm BTS by clinical criteria such as oximetric arterial saturation, FiO2 or pO2 perioperatively. Five of 9 patients with a 3.5-mm BTS were among those undergoing LPA plasty at the time of BDCPA, and 6 of 13 patients with a 3.0-mm shunt required LPA plasty, suggesting without statistical significance that a larger shunt is not protective. In this series the choice of shunt size is not statistically associated with pulmonary arterioplasty but shunt insertion point is.

Additional limitations to this study include the inability to discern whether the modified shunt flow is causally responsible for the findings or whether the shunt modification, owing to its construction over the ductal remnant site, simply lessens the contractile or compressive forces exerted on the LPA by the remnant ductal tissue. Any effect of surgeon experience and era of operation on the improvements cannot be determined by this small series. Temporally coincident with, though not demonstrably causally related, is the lower incidence of LPA stenosis with the advent of the shunt modification.

Following the Norwood procedure, multiple forces can compromise leftward pulmonary blood flow. A modification of the systemic to pulmonary artery shunt insertion to direct its flow leftward and obliquely into the retroaortic pulmonary artery may neutralize some of these forces and significantly reduced the incidence of LPA stenosis following the Norwood procedure in this retrospective cohort study. Ongoing study will be necessary to affirm a mechanism and to follow long-term outcomes of this iteration of the Norwood procedure by comparison to others presently in evolution.


   References
Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
 References
 

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