| HOME | HELP | FEEDBACK | SUBSCRIPTIONS | ARCHIVE | SEARCH | TABLE OF CONTENTS |
|
|
|
|||||||
|
|||||||||
Ann Thorac Surg 2004;78:1839-1842
© 2004 The Society of Thoracic Surgeons
a Department of Pediatric Cardiac Surgery, National Heart Hospital, Sofia, Bulgaria
b Department of Cardiac Surgery, Grabengasse, Germany
c Department of Pediatric Cardiology, University Heidelberg, Grabengasse, Germany
Accepted for publication July 21, 2003.
* Address reprint requests to Dr Loukanov, National Heart Hospital, Pediatric Cardiac Surgery, ‘Mladost-2’ bl.223/9 ap. 156, Sofia 1799, Bulgaria.
tsloukanov{at}abv.bg
 |
Abstract |
|---|
 Top Abstract IntroductionCommentReferences |
|---|
|
Introduction |
|---|
|
TopAbstractIntroductionCommentReferences |
|---|
The term, pulmonary vascular sling, was introduced in 1958 by Contro and coworkers [1] and describes a congenital anomaly in which the left pulmonary artery (LPA) originates anomalously from the right pulmonary artery. In the typical vascular sling, the anomalous LPA arises from the posterior aspect of the right pulmonary artery, passes over the right main stem bronchus near its origin, and turns to the left coursing between the trachea and esophagus to reach the left pulmonary hilus. Complete tracheal rings of various extensions and intrinsic tracheal stenosis accompany this anomaly in more than 50% of the cases [2]. One of the most common misperceptions about this congenital anomaly, according to Jonas [3] is that tracheal stenosis occurs secondary to compression by the LPA. In fact, these two anomalies are very often associated.
A male infant, 4 months of age, weighing 6000 g was referred to our hospital with respiratory distress. A previous operation at another institution was performed at 2 months of age for pulmonary artery sling syndrome. Previous surgical repair consisted of dividing the arterial ligament and antetracheal reimplantation of the anomalous LPA into the main pulmonary artery (MPA) under cardiopulmonary bypass. The tracheal pathology was not recognized and no reconstruction of the tracheobronchial segment was undertaken.
Actual angiography with concomitant tracheobronchography revealed a marked stenosis at the origin of the reimplanted LPA, tracheal diverticulitus, tracheal stricture of the compressed airway segment from the LPA, "funnel trachea" with complete cartilaginous rings and the right upper lobe bronchus arising independently as an bronchus suis, and patent foramen ovale (Fig 1).
|
The patient was unable to be weaned from the respirator. Surgical reintervention was undertaken at our institution at 5 months of age.
In this child, we elected to perform the complete repair in one stage. The repair was performed through a midline re-sternotomy with the use of cardiopulmonary bypass.
The aortic arch and pulmonary arteries were dissected out and extensively mobilized. The LPA was found to be implanted into the MPA relatively far distally and in a rather posterior location. The LPA formed together with the right pulmonary artery was a tissue bridge exerting a residual, significant compression at the distal part of the trachea near the tracheal bifurcation anteriorly and to the left. The left and right pulmonary arteries were extensively mobilized out to the hilus. Both pleural cavities were opened and dissected free of adhesions. The pulmonary ligaments were transected. The intrathoracal part of the trachea, including the carina and the proximal segments of the main bronchus were dissected free from rather dense adhesions. The right upper lobe bronchus was found to arise as bronchus suis at 2 cm proximally from the carina. Proximally and adjacent to the origin site of the bronchus suis, a diverticulum of peanut size was observed, which was also diagnosed by bronchoscopy. The tracheal wall from the anterior part of the carina and the part of the trachea adjacent to the original site of the pulmonary sling was unstable and showed signs of tracheomalacia (Fig 2).
|
The right atrium was opened under circulatory arrest. No intracardiac pathology was detected upon inspection, except a 3 mm to 4 mm open foramen ovale. Direct closure of the defect was performed. Cardiopulmonary bypass was reinstituted after removal of air.
The trachea was carefully mobilized with bilateral hilar release and transected just proximally to the carinal region. The inspection of the lumen revealed the picture of the "funnel trachea" with complete cartilaginous rings. The internal diameter of the tracheal lumen was 2.5 mm to 3 mm. The mucosal surface of the trachea showed extensive fibrotic changes. The proximal limits of the dysplastic tracheal segment were evaluated under bronchoscopic control. They were defined from the diverticulitus level proximally to a few millimeters before the carinal level distally (Fig 2 inlet). The last pre-carinal cartilage was with normal morphology and consistency. The resection of the dysplastic tracheal segment was performed in proximal direction in a segment by segment technique in order to exactly determine the proximal resection line. The length of the resected trachea was finally 28%. The dysplastic bronchus suis was included in the resected specimen. The tracheal ends were anastomosed end-to-end with interrupted 4-0 polydioxanone sutures after additional mobilization (Fig 3). The anastomotic site was free of tension. Palpatory examination of the tracheal wall at the bifurcation level revealed stable consistency. The bronchoscopic control showed no residual obstruction of the reconstructed carinal region, and there were no disturbances from the sutures inside. The bronchus suis was evaluated to be dysplastic at long distance so that no reimplantation seemed reasonable.
|
The postoperative period was uneventful. The patient was extubated on postoperative day 10 under bronchoscopic monitoring. Previous endoscopy, performed on postoperative day 3 showed tracheal lumen free of any obstruction. Both main bronchi were of normal lumen sizes.
The child was discharged to the referring center on postoperative day 17 and has reported 2 months later to be in excellent condition at home.
|
Comment |
|---|
|
TopAbstractIntroductionCommentReferences |
|---|
Willis J. Potts performed the first successful surgical repair of pulmonary artery sling in 1953 [5]. Potts divided the LPA and reanastomosed the two ends back together anterior to the trachea through a right thoracotomy.
Despite successful surgical correction of the anomaly, mortality and morbidity remain high postoperatively and appear to be related to underlying coexistent tracheobronchial abnormalities [6].
The reimplantation of the pulmonary artery should be technically perfect if it is to remain patent. Because kinking at the LPA origin can be a problem when a reimplantation is carried out, we believe that the LPA should be anastomosed at the left and anterolateral aspect of the MPA and more proximally to the pulmonary valve. Careful dissection of the entire course of the anomalous LPA before tension-free reimplantation at the MPA is considered to be one of the essential steps in minimizing the LPA patency disturbances [5, 7].
Of 68 postmortem examinations of pulmonary artery sling, 51% had associated tracheobronchial abnormalities such as: (1) hypoplasia of the distal trachea (38% with complete cartilaginous rings), (2) stenosis of the left main bronchus (5%), and (3) direct origin of the right epi-arterial bronchus from the trachea (12%) [2].
The so-called bronchus suis is an anomalous right upper bronchus branching from the trachea. The portion of the trachea distal to the origin of such a right upper bronchus is described to be frequently hypoplastic. Characteristically the vascular sling passes over the intermediate bronchus inferior to the independently arising right upper bronchus, thus, bronchus suis [2].
The constellation of tracheal stenosis, dysplastic bronchus suis, and diverticulitus of the tracheal wall necessitated in this case extended resection of the trachea combined with atypical pulmonary resection.
The mobility of the infant trachea allows one-third (25% to 30%) of the trachea to be resected comfortably [3]. Sufficient mobilization of the trachea should be performed to avoid tension. In our case, bilateral hilar release was the preferred technique.
In our opinion, tracheobronchial malformations accompanying pulmonary sling syndrome should be exactly defined by endotracheal investigation and treated surgically in such a way that any residual stenosis is avoided. We prefer the relatively high diagnostic accuracy of angiography with concomitant tracheobronchography, because both involve organ systems that can be visualized [7].
As previously shown, another possible method in cases with tracheomalacia with floppiness of the tracheal wall consists of bronchoscopic-guided external tracheobronchial suspension within a ring-reinforced polytetrafluoroethylene prosthesis [8].
We consider intraoperative endoscopic monitoring of airway constellation and subsequent decompression to be a most helpful technique in such cases.
|
References |
|---|
|
TopAbstractIntroductionCommentReferences |
|---|
This article has been cited by other articles:
|
|
 |
|
  S. Hagl, C. Sebening, W. Springer, and T. Loukanov Modified Sliding Tracheal Plasty Using the Bridging Bronchus for Repair of Long-Segment Tracheal Stenosis Ann. Thorac. Surg., March 1, 2008; 85(3): 1118 - 1120. [Abstract] [Full Text] [PDF]
|
|
|
|
 |
|
 E. Le Bret, N. Teissier, E. Belli, A. Sigal-Cinqualbre, V. Couloignier, P. Narcy, S. Demontoux, N. Gharbi, R. Roussin, T. Van Den Abbeele, et al. Slide tracheoplasty in the presence of tracheal bronchus in an infant J. Thorac. Cardiovasc. Surg., July 1, 2006; 132(1): e15 - e16. [Full Text] [PDF]
|
|
|
|
|
|
T. Loukanov, C. Sebening, W. Springer, H. Ulmer, and S. Hagl Simultaneous management of congenital tracheal stenosis and cardiac anomalies in infants J. Thorac. Cardiovasc. Surg., December 1, 2005; 130(6): 1537 - 1541. [Abstract] [Full Text] [PDF]
|
|
| ||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| HOME | HELP | FEEDBACK | SUBSCRIPTIONS | ARCHIVE | SEARCH | TABLE OF CONTENTS |
| ANN THORAC SURG | ASIAN CARDIOVASC THORAC ANN | EUR J CARDIOTHORAC SURG |
| J THORAC CARDIOVASC SURG | ICVTS | ALL CTSNet JOURNALS |
