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Ann Thorac Surg 2007;83:188-192
© 2007 The Society of Thoracic Surgeons

Original Articles: Cardiovascular

Pulmonary Arterial Reconstruction for Pulmonary Coarctation in Early Infancy

Takeshi Shinkawa, MDa, Masaaki Yamagishi, MD, PhDa,*, Keisuke Shuntoh, MDa, Takako Miyazaki, MDa, Takahiro Hisaoka, MDa, Hitoshi Yaku, MD, PhDb

a Department of Pediatric Cardiovascular Surgery, Children’s Research Hospital
b Division of Cardiovascular Surgery, Kyoto Prefectural University of Medicine, Kyoto, Japan

Accepted for publication August 16, 2006.

* Address correspondence to Dr Yamagishi, Department of Pediatric Cardiovascular Surgery, Children’s Research Hospital, Kyoto Prefectural University of Medicine, Kawaramachi, Hirokoji, Kamigyo-ku, Kyoto 602-8566, Japan (Email: myama{at}koto.kpu-m.ac.jp).


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   Abstract
Top
 Abstract
Introduction
 Patients and Methods
 Results
 Comment
 References
 
BACKGROUND: Pulmonary atresia with pulmonary coarctation may complicate diminished and unbalanced pulmonary development. The aim of this study is to assess the outcome of pulmonary arterial reconstruction with cardiopulmonary bypass in early infancy for sufficient and balanced pulmonary development.

METHODS: We performed a retrospective review of 15 patients with pulmonary coarctation younger than 4 months of age who underwent pulmonary arterial reconstruction between 2001 and 2005. The mean age and weight were 42.2 days and 3.62 kg, respectively. The patient population included 5 biventricular repair candidates and 10 Fontan candidates. To evaluate the pulmonary arterial development, the preoperative and postoperative pulmonary arterial index and minimum diameter of the pulmonary artery were compared.

RESULTS: No early or in-hospital deaths occurred, and there was no nonconfluent pulmonary artery development or segmental mal-development after a mean follow-up period of 14.9 months. Immediate pulmonary flow regulation was required in 2 patients because of excessive pulmonary flow. The mean pulmonary arterial index increased significantly from 103 mm2/m2 to 343 mm2/m2, and the mean minimum diameter of the pulmonary artery increased significantly from 2.02 mm to 4.45 mm. Four biventricular repair candidates completed definitive repair, and 2 required surgical reintervention in the pulmonary artery. Six Fontan candidates completed the Glenn procedure, and 1 completed the Fontan procedure. Three required surgical reintervention in the pulmonary artery. Two late deaths occurred after the Glenn procedure because of ventricular dysfunction and respiratory infection.

CONCLUSIONS: Pulmonary arterial reconstruction in early infancy provides sufficient and balanced pulmonary arterial development for pulmonary atresia with pulmonary coarctation.

Pulmonary atresia with morphologic problems in the pulmonary artery (PA), such as pulmonary arterial stenosis at the ductal attachment (pulmonary coarctation), may complicate diminished pulmonary blood flow or unbalanced pulmonary perfusion [1]. They will combine with future PA mal-development and will adversely affect postoperative prognosis and reduce the patient’s quality of life [2–5]. We hypothesized that the early surgical intervention of the morphologic problems in the PA could prevent these prospective problems, and we performed pulmonary arterial reconstruction and augmentation with cardiopulmonary bypass (CPB) in early infancy as the initial surgical intervention to obtain sufficient and balanced pulmonary perfusion and development. In this study, we assessed the outcome and the usefulness of pulmonary arterial reconstruction for pulmonary coarctation with CPB in early infancy.


   Patients and Methods
Top
 Abstract
 Introduction
 Patients and Methods
Results
 Comment
 References
 
Patients
From April 2001 to December 2005, 15 consecutive infants younger than 4 months of age, with pulmonary atresia and pulmonary coarctation, underwent pulmonary arterial reconstruction and augmentation with CPB at the time of their first surgical intervention at the Children’s Research Hospital, Kyoto Prefectural University of Medicine. The clinical characteristics and anatomic features are summarized in Table 1. Mean age and weight at the time of the operation were 42.2 ± 10.8 days (range, 30 to 62 days) and 3.62 ± 0.6 kg (range, 2.6 to 4.4 kg). Heterotaxia was found in 4 patients.


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  		Table 1. Details of Patients Who Underwent Pulmonary Arterial Reconstruction in Early Infancy
 
Five of the 15 patients were biventricular repair candidates, and 10 were Fontan candidates. No patients had severe atrioventricular valve insufficiency at the time of operation. The particular pulmonary arterial morphology was diagnosed by cardiac catheter examination in 3 patients, helical computed tomography (CT) with three-dimensional (3D) reconstruction in 9 [6], and both in 3 (Fig 1). The patients who had pulmonary arterial stenosis at a ductal attachment of less than 2.9 mm by cardiac catheter examination or helical CT with 3D reconstruction were diagnosed with pulmonary coarctation and were indicated to the surgical intervention. No patients underwent catheter intervention before the surgical intervention. Patients were maintained by the continuous infusion of prostaglandin E1.


Figure 1
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  		Fig 1. Helical computed tomograms with three-dimensional reconstruction of the patient with a single ventricle, pulmonary atresia, and pulmonary coarctation. (A) Preoperative posterior view shows patent ductus arteriosus (PDA) and the left pulmonary coarctation. (B) Postoperative posterior (P) view shows the widely augmented central pulmonary artery and the Blalock-Taussig shunt.

 
Methods
With approval by the Ethics Committee of Kyoto Prefectural University of Medicine and individual consents for the study from all patients’ parents, the hospital records of all 15 patients were retrospectively reviewed, and follow-up data were obtained through clinic notes and contact with the patients and their families.

To determine pulmonary arterial development, pulmonary arterial size was measured in the frontal view on angiograms and then by helical CT. The diameters of right and left PAs were measured at just proximal to the first branch, and the pulmonary arterial index (PAI) was calculated with the following equation:


Formula

The preoperative and postoperative PAI and the minimum diameter of the PA were compared.

Surgical Technique
In all patients, CPB was established through a median sternotomy. The patients underwent complete resection of ductal tissue and incision of the stenotic central PA. To avoid distal pulmonary arterial stenosis, the pulmonary arterial incision was limited within the central PA. The PA was reconstructed and augmented with a fresh autologous pericardial patch. A modified Blalock-Taussig shunt was established between the brachiocephalic artery and the augmented PA (most in the pericardial patch) with an expanded polytetrafluoroethylene graft (Fig 2). The size of the Blalock-Taussig shunt was 4.0 mm in 14 patients and 3.5 mm in 1 patient. At the weaning from CPB, the pulmonary blood flow regulation was performed by partial clipping of the Blalock-Taussig shunt, if necessary. No patients had a concomitant procedure.


Figure 2
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  		Fig 2. Operative scheme of a patient with pulmonary coarctation. (A) Resection of ductal tissue and the incision of stenotic central pulmonary artery (PDA = patent ductus arteriosus). (B) Central pulmonary arterial augmentation with a fresh pericardial patch. (C) Placement of a Blalock-Taussig shunt (BT) on the pericardial patch.

 
Statistical Analysis
The Student t test was used to compare the continuous variables using Stat Mate III software (ATMS Co, Tokyo, Japan), and p < 0.05 was considered significant. Data are expressed as means ± standard deviation and as ranges.


   Results
Top
 Abstract
 Introduction
 Patients and Methods
 Results
Comment
 References
 
Postoperative Course
The follow-up period was 14.9 ± 11.5 months (range, 3 to 42 months). There were no early or hospital deaths in the patient population, and no nonconfluent pulmonary artery or segmental pulmonary mal-development occurred in any patient.

Among the 5 biventricular candidates, 3 achieved definitive repair. One patient underwent a palliative right ventricular outflow tract reconstruction procedure owing to left pulmonary arterial stenosis, which was unsolved by the catheter intervention, followed by definitive repair, and 1 patient was waiting for definitive repair. Two patients required surgical reintervention to the PA, including concomitant reintervention with definitive repair. Among the 10 Fontan candidates, 6 underwent the Glenn procedure, 4 were waiting for the Glenn procedure, 1 underwent the Fontan procedure, 3 were waiting for the Fontan procedure, and 2 died after the Glenn procedure.

One of the patients who died after the Glenn procedure was diagnosed as having single ventricle and pulmonary atresia and had right ventricular dysfunction with an ejection fraction of 0.40 at birth. The right ventricular dysfunction progressed after the initial pulmonary arterial reconstruction and after the Glenn procedure, without additional pulmonary flow, performed at age 1 year. He died 1 year after the Glenn procedure owing to progressive ventricular dysfunction. The other patient was diagnosed with polysplenia, double outlet right ventricle, atrioventricular discordance, and pulmonary atresia. At 10 months of age, she had a Glenn procedure, without additional pulmonary flow, and her condition was quite good, without atrioventricular valve regurgitation. However, a severe respiratory infection suddenly developed 4 months after the Glenn procedure and she died in another hospital.

Two patients with heterotaxia required fine regulation of the pulmonary blood flow soon after the operation because of congestive heart failure owing to excessive pulmonary flow: One underwent a 4.0-mm Blalock-Taussig shunt banding 7 days after the operation, and the other underwent conversion of the Blalock-Taussig shunt size from 4.0 mm to 3.5 mm 14 days after the operation.

Anatomic contiguousness between the brachiocephalic artery and the PA may cause postoperative excessive pulmonary flow. Three of 10 patients required surgical reintervention to the PA concomitant with the Glenn procedure (Fig 3).


Figure 3
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  		Fig 3. Scheme of postoperative course. (A) Postoperative course of the biventricular repair candidates. (B) Postoperative course of the Fontan candidates. (BTs = Blalock-Taussig shunt; BVR = biventricular repair; PA = pulmonary artery; pRVOTR = palliative right ventricular outflow tract reconstruction.)

 
Postoperative Pulmonary Arterial Growth and Balance
Eleven of 15 patients underwent postoperative cardiac catheter examination 7.3 ± 3.7 months (range, 5 to 18 months) after the operation. These 11 patients included 2 who required postoperative regulation of the pulmonary blood flow.

The results of preoperative and postoperative examination were compared. PAI increased significantly from 103 ± 38.1 mm2/m2 (range, 50 to 162 mm2/m2) to 343 ± 156 mm2/m2 (range, 216 to 691 mm2/m2) after the operation. Among biventricular repair candidates, PAI increased significantly from 115 ± 42.0 mm2/m2 (range, 76 to 162 mm2/m2) to 296 ± 62.1 mm2/m2 (range, 216 to 362 mm2/m2). Among Fontan candidates, PAI increased significantly from 96.0 ± 37.2 mm2/m2 (range, 50 to 144 mm2/m2) to 371 ± 190 mm2/m2 (range, 217 to 691 mm2/m2) (Fig 4A).


Figure 4
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  		Fig 4. (A) Changes in pulmonary arterial index (thick black line, all patients; thin dark gray line, biventricular repair candidates; thin light gray line, Fontan candidate). (B) Changes in minimum diameter of the pulmonary artery (thick black big line, all patients; thin gray lines, each patient).

 
The minimum pulmonary arterial diameter increased significantly from 2.02 ± 0.35 mm (range, 1.6 to 2.6 mm) to 4.45 ± 1.91 mm (range, 1.7 to 7.7 mm), respectively. The minimum pulmonary arterial diameter of the all patients, except 1, increased (Fig 4B). Postoperative mean pulmonary arterial pressure was 16.2 ± 2.74 mm Hg (range, 13 to 21 mm Hg), respectively.

Eight of 15 patients underwent postoperative lung perfusion scintigraphy 3.3 ± 2.3 months (range, 1 to 8) after the operation. The postoperative mean pulmonary perfusion balance determined by scintigram was 36:64 (range, 28:72 to 43:57).


   Comment
Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
References
 
Pulmonary atresia with morphologic problems in the PA may combine with subsequent problems, such as nonconfluent PA, unilateral hypoplasia, and segmental pulmonary hypertension [1], and result in deterioration of postoperative prognosis and patient’s quality of life [2–5]. To improve these prospective ill effects, it is necessary to obtain adequate and balanced pulmonary blood flow. It is necessary to resolve the morphologic problems in early life, especially in Fontan candidates, and pulmonary arterial reconstruction and augmentation in early infancy may provide a solution to these problems [7, 8]. The catheter intervention to the pulmonary arterial stenosis can be the choice of treatment, but we think that the catheter intervention for the pulmonary coarctation in early infancy may have higher risk [9].

In our experience, the increase of PAI would indicate the pulmonary arterial growth after the reconstruction, because only the central PA was augmented with the pericardial patch. Not all patients have a nonconfluent PA and unilateral PA mal-development, and reconstruction in early infancy should provide sufficient and balanced pulmonary arterial development. Even if only a small number of patients could complete definitive repair or the Fontan procedure by now, more patients are expected to complete the repair in the future because they achieved sufficient and balanced pulmonary arterial development. However, surgical reinterventions concomitant with the next-stage operation or as independent operations were required in some patients. These morphologic problems in the PA seemed not to be easily resolved; thus, it may be better to select a staged strategy and to perform pulmonary arterial reconstruction and augmentation in every staged operation.

During the operation, it is important to remove the ductal tissue completely and to use a relatively large autologous pericardial patch, because ductal tissues may result in postoperative restenosis of the reconstructed PA. The autologous pericardium is the best material for pulmonary arterial patches because of its growth potential, anti-thrombogenicity, and tolerance to infection [10]. The homograft is also a useful material for the pulmonary arterial patches, but it lacks growth potential and has the possibility of degeneration and calcification in future.

Increased risk during the operation and difficulty in controlling pulmonary blood flow concomitant with postoperative changes in pulmonary resistance are problems in pulmonary arterial reconstruction with CPB in early infancy [11]. In our series, 2 patients required pulmonary flow regulation soon after the operation due to excessive pulmonary flow concomitant with a postoperative reduction of pulmonary resistance. Thus, it seems important to determine the optimum oxygen saturation at the time of weaning from bypass, to evaluate pulmonary blood flow correctly, and to avoid causing any sudden changes in pulmonary resistance during postoperative care [12].


   References
Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
 References
 

  1. Luhmer I, Ziemer G. Coarctation of the pulmonary artery in neonatesPrevalence, diagnosis, and surgical treatment. J Thorac Cardiovasc Surg 1993;106:889-894.[Abstract]
  2. Stamm C, Friehs I, Zurakowski D, et al. Outcome after reconstruction of discontinuous pulmonary arteries J Thorac Cardiovasc Surg 2002;123:246-257.[Abstract/Free Full Text]
  3. Agnoletti G, Boudjemline Y, Bonnet D, Sidi D, Vouhe P. Surgical reconstruction of occluded pulmonary arteries in patients with congenital heart diseases: effects on pulmonary artery growth Circulation 2004;109:2314-2318.[Abstract/Free Full Text]
  4. Trivedi KR, Karamlou T, Yoo S-J, et al. Outcomes in 45 children with ductal origin of the distal pulmonary artery Ann Thorac Surg 2006;81:950-957.[Abstract/Free Full Text]
  5. Waldman JD, Karp RB, Gittenberger-deGroot AC, Agarwala B, Glagov S. Spontaneous acquisition of discontinuous pulmonary arteries Ann Thorac Surg 1996;62:161-168.[Abstract/Free Full Text]
  6. Shiraishi I, Yamamoto Y, Ozawa S, et al. Application of helical computed tomographic angiography with differential color imaging three-dimensional reconstruction in the diagnosis of complicated congenital heart diseases J Thorac Cardiovasc Surg 2003;125:36-39.[Free Full Text]
  7. Ishibashi N, Koide M, Uchita S, Seguchi M. When should pulmonary artery angioplasty be performed for Fontan candidates with pulmonary coarctation? Two cases of pulmonary artery angioplasty with the Blalock-Taussig shunt on pump in neonates Jpn J Thorac Cardiovasc Surg 2004;52:185-188.[Medline]
  8. Kim HK, Kim WH, Kim SC, Lim C, Lee CH, Kim SJ. Surgical strategy for pulmonary coarctation in the univentricular heart Eur J Cardiothorac Surg 2006;29:100-104.[Abstract/Free Full Text]
  9. Alwi M, Choo KK, Latiff HA, Kandavello G, Samion H, Mulyadi, MD. Initial results and medium-term follow-up of stent implantation of patent ductus arteriosus in duct-dependent pulmonary circulation J Am Coll Cardiol 2004;44:438-445.[Abstract/Free Full Text]
  10. Hibino N, Shin’oka T, Kurosawa H. Long-term histologic findings in pulmonary arteries reconstructed with autologous pericardium N Engl J Med 2003;348:865-867.[Free Full Text]
  11. Shen I, Giacomuzzi C, Ungerleider RM. Current strategies for optimizing the use of cardiopulmonary bypass in neonates and infants Ann Thorac Surg 2003;75:729-734.
  12. Nakano T, Kado H, Shiokawa Y, et al. The low resistance strategy for the perioperative management of the Norwood procedure Ann Thorac Surg 2004;77:908-912.[Abstract/Free Full Text]



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