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Right arrow Trachea and bronchi

J Thorac Cardiovasc Surg 2002;123:145-152
© 2002 The American Association for Thoracic Surgery


General Thoracic Surgery

Management of congenital tracheal stenosis by means of slide tracheoplasty or resection and reconstruction, with long-term follow-up of growth after slide tracheoplasty

Hermes C. Grillo, MD, Cameron D. Wright, MD, Gus J. Vlahakes, MD, Thomas E. MacGillivray, MD

From the Divisions of General Thoracic Surgery and Cardiac Surgery, Massachusetts General Hospital, and the Department of Surgery, Harvard Medical School, Boston, Mass.

Received for publication April 23, 2001. Revisions requested June 19, 2001; revisions received July 24, 2001. Accepted for publication July 27, 2001. Address for reprints: Hermes C. Grillo, MD, Massachusetts General Hospital, Blake 1570, Boston, MA 02114 (E-mail: pguerriero{at}partners.org).


    Abstract
 Top
 Abstract
 Introduction
 Patients and methods
 Results
 Discussion
 References
 
Objective: The difficult problem of congenital tracheal stenosis is infrequent and has been managed with several methods. Patch tracheoplasty has been favored in recent years. Alternative experience with a simpler program of slide tracheoplasty for long-segment stenosis or resection and reconstruction for short-segment stenosis is described and proposed as preferable. Long-term growth after slide tracheoplasty was studied.
Methods: Eleven consecutive patients aged 10 days to 23 years with varied patterns of stenosis (including concurrent pulmonary artery sling, anomalous right upper lobe bronchus, and bridge bronchus) had their stenoses corrected, 8 by means of slide tracheoplasty and 3 by means of resection and anastomosis. Retrospective review was made of hospital course, complications, and long-term results, with observation of growth in 4 patients (from more than 11/2-73/4 years).
Results: All patients are alive and enjoy good airways. Only 3 patients who needed concomitant cardiovascular procedures and 1 with poor ventricular function required bypass. Eight were extubated immediately or on the day of the operation, 1 at 3 days, and 1 at 8 days. A patient with complex anomalies needed 10 days of ventilation. Three had anastomotic granulomas successfully treated by means of a single bronchoscopy. Long-term airway growth was entirely satisfactory after slide tracheoplasty in 4 infants and small children (aged 10 days, 3 months, 6 months, and 31/2 years, respectively).
Conclusions: Slide tracheoplasty gives excellent short- and long-term results because long congenital stenosis is reconstructed with native tracheal tissue and is therefore immediately stable and lined with normal epithelium, and the operation is accomplished more simply and with a generally more benign postoperative course. Wholly satisfactory growth of the repaired segment occurs. Less common short congenital stenosis is effectively managed with resection and anastomosis.


    Introduction
 Top
 Abstract
 Introduction
 Patients and methods
 Results
 Discussion
 References
 
Congenital tracheal stenosis occurs in varied lengths, which are most often characterized by complete cartilaginous O rings. The stenotic segment may be located in upper or lower trachea, may be funnel shaped, and may involve different percentages of the length of trachea.Go 1 Stenosis may also involve the main bronchi and may be accompanied by an anomalous right upper lobe bronchus or by a bridging bronchus below, such an anomalous bronchus connecting with horizontal bronchi to the remainder of the right and left lungs.Go 2 A left pulmonary artery sling may be found with either a short segmental stenosis or a more complex form of stenosis.Go 3 Other variants also occur. Multiple anomalies, including cardiac malformations, are frequently associated. Patients may be symptomatic immediately after birth or may manage with limited exercise tolerance until later in life.

There is no effective medical treatment. Forceful dilation may result in splitting the cartilage O rings, with the possibility of perforation or restenosis. Three surgical techniques have been used for correction. First, early reports of tracheal resection and reconstruction in children by DeLorimier and colleagues,Go 4 Grillo and Zannini,Go 5 and Carcassonne and coworkersGo 6 included patients with congenital stenosis. It was demonstrated experimentally and clinicallyGo Go 7,8 that anastomosis of the juvenile trachea grew satisfactorily. However, only a finite length of trachea, approximately 25% to 30%, can be resected and followed by end-to-end suture before excessive anastomotic tension may result in separation.Go 9

Second, long congenital stenosis beyond the compass of safe resection and reconstruction was treated by Kimura,Go 10 Tsugawa,Go 11 and their associates by means of a linear anterior incision of the stenotic segment and insertion of a patch of costal cartilage. Idriss and colleaguesGo 12 and successors modified this approach by substitution of a pericardial patch. The flexible pericardial insert must be suspended to the mediastinum, and all patch techniques require prolonged stenting with intubation during early healing. Patch techniques were also reported by Bando,Go 13 Jaquiss,Go 14 and their associates. Granulation tissue arising from the mesenchymal surface of the patches required multiple bronchoscopic debridements and necrosis or collapse of the patch sometimes occurred.Go Go 15,16 Reoperation was necessary in 7 of 28 patients.Go 17

Third, to solve this vexing problem, Tsang and associatesGo 18 proposed slide tracheoplasty. The stenotic segment is transected at its midpoint, the upper and lower stenotic segments are incised vertically anteriorly in one segment and posteriorly in the other, the corners of these splayed segments are trimmed, and the two are slid together and sutured(Figure 1). The circumference of the trachea is doubled, and the cross-sectional area is quadrupled.Go 19 The stenotic segment is shortened by half. The trachea is thus repaired with tracheal wall containing native cartilages and is immediately lined with normal tracheal epithelium. Satisfactory subsequent growth was demonstrated experimentally and clinically.Go Go 20,21



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Fig. 1. Technique of slide tracheoplasty. A, The extent of stenosis is identified precisely. The stenotic segment is divided transversely in its midpoint after circumferential dissection at that locus only. The upper stenotic segment is incised vertically posteriorly, and the lower segment is incised anteriorly for the full length of stenosis. B, The right-angled corners produced by these divisions are trimmed above and below. A stay suture near the tip of the superior flap is helpful, as are traction sutures at the tracheobronchial angles or main bronchi below. Minimal dissection of leteral blood supply is performed. C, The two ends are slid together after placement of individual anastomotic sutures around the entire oblique circumference of the tracheoplasty. D and E, The circumference is doubled, resulting in quadrupled cross-sectional area. Reprinted with permission from the Society of Thoracic Surgeons The Annals of Thoracic Surgery 1994;58:613-21.)

 
Since the introduction of slide tracheoplasty at Massachusetts General Hospital in 1991, 8 patients ranging in age from 10 days to 19 years with stenoses varying in length and location and with and without additional bronchial or cardiac anomalies, including left pulmonary artery sling, have undergone surgical correction with this technique(Figure 2). Three additional patients with congenital stenosis underwent tracheal resection and reconstruction by means of direct end-to-end anastomosis during this period(Figure 3). In these 3 patients simple anastomotic technique seemed to be safely applicable, and in 2 patients it was judged that slide tracheoplasty was inappropriate for additional reasons. The patients were operated on by members of the General Thoracic Surgical Division (H.C.G. and C.D.W.) with cooperation of the Cardiac Surgical Division (G.J.V. and T.E.M.) when required. Complete preoperative delineation of airway and cardiovascular abnormalities is essential. Critical studies include bronchoscopy, spiral computed tomography, echocardiography, and cardiac catheterization with angiography in some patients.



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Fig. 2. Diagrams of anatomic findings in 8 patients who underwent slide tracheoplasty. Figures indicate percentage of stenotic trachea. Scale differs because of wide range of age and tracheal size. Details are listed in Table 1.

 

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Table 1. Patients undergoing slide tracheoplasty
 


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Fig. 3. Diagram of anatomic findings and reconstruction performed in 3 patients who underwent resection. Details in Table 2.

 

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Table 2. Patients undergoing resection and reconstruction
 

    Patients and methods
 Top
 Abstract
 Introduction
 Patients and methods
 Results
 Discussion
 References
 
Patients
Slide tracheoplasty
Data on patients undergoing slide tracheoplasty are given inTable 1.Go The 4 younger patients (operated on at ages 10 days, 3 months, 6 months, and 31/2 years) were seriously symptomatic (patients 7, 3, 6, and 4). The 10-day-old infant was transferred from another hospital intubated and in respiratory distress. The 3-month-old child had stridor and episodes of cyanosis and desaturation. The 6-month-old child had a similar history and was received intubated receiving heliox with elevated PCO2. The 6-month-old had respiratory arrest and was transferred intubated from another hospital. A 101/2-year-old (patient 8) had dyspnea, stridor, cyanosis, and impaired ventricular function. She had undergone a bidirectional Glenn procedure earlier in life. Her airway was corrected in anticipation of a Fontan procedure. Maximal stenosis in these patients was between 1.9 and 3 mm in diameter. The 3 teenaged patients (patients 1, 2, and 5) were severely limited in activities of all types. One had been seen previously, but repair initially was deferred because only patch procedures were then available.

Stenoses varied in length from 36% to 83% of tracheal length(Figure 2Go). Of these 8 patients, 3 had an anomalous right upper lobe bronchus, 1 associated with a bridging bronchus and also left pulmonary artery sling. Another anomalous pulmonary artery was in conjunction with 50% of length lower tracheal stenosis. He had a constellation of anomalies that occur with congenital tracheal stenosis, including inperforate anus. The 101/2-year-old had an anomalous retrotracheal pulmonary artery (anastomosed previously to the superior vena cava at the time of a bidirectional Glenn procedure), but it did not curve around or impinge significantly on the trachea. One showed a degree of malacia in the short segment of trachea below a bronchus suis and also in both main bronchi.

Resection and reconstruction
Data on patients undergoing resection and reconstruction are given inTable 2.Go The 3-month-old had severe symptoms from birth and had a complex of stenosis and malacia over a short tracheal segment just above the carina (patient 9). The shortness of the segment and the accompanying malacia were indications for resection and anastomosis. The 7-month-old with VACTERL*Go association was transferred intubated and ventilated by means of tracheostomy after tracheoesophageal fistula repair, modified Blalock-Taussig shunt for single-ventricle congenital heart disease, gastric fundoplication, and colostomy. He had air trapping and episodes of airway obstruction (patient 11). A bridging bronchus was very narrow throughout but showed maximal stenosis at its proximal end, making it unsuitable for slide tracheoplasty, as was done in patient 6. The segment was short enough to permit complete resection and anastomosis. The 23-year-old had been observed since infancy, when the stenosis was deemed too long for resection. He had undergone several surgical procedures for other malformations. The stenotic segment remained narrow but grew proportionally to the normal trachea. Resection was done to enhance his performance capabilities (patient 10).

Procedures
The technique of our modification of Tsang and Goldstraw's procedure for slide tracheoplasty has been described in detail.Go 19 The first 5 tracheoplasties were performed without need for cardiopulmonary bypass. Two patients who required reimplantation of the left pulmonary artery also needed a period of normothermic bypass. Because the left pulmonary artery was divided early for access, the tracheoplasty was also performed during bypass before pulmonary arterial reanastomosis. In patient 6 a segment of mild stenosis proximal to the severely stenotic bridging bronchus was not modified, and tracheoplasty was confined to the very stenotic bridging bronchus. In all but the first patient, the distal segment of stenosis was opened anteriorly. Patient 1 showed cartilage O rings in both main bronchi, but narrowing was insufficient to require enlargement. The slide technique could be modified to accomplish this were it necessary.

Patient 8 was operated on during bypass because of impaired ventricular function. The stenosis telescoped from a 3.5-cm segment with a diameter of 3 to 4 mm to a 4-cm segment of 2 to 3 mm in diameter, with an additional shelf of scar at the meeting point of the segments. This was believed to have resulted from prolonged intubation during prior operations. This 1-cm zone of maximal stenosis was resected, and slide tracheoplasty was performed on the remaining stenosis.

The thymic lobes are usually separated for tracheal exposure. With modest further mobilization, a thymic lobe was generally interposed between the tracheal suture line and the brachiocephalic artery to buttress the anastomosis and prevent potential erosion of the artery. Right sternohyoid muscle divided superiorly was used in patient 8, in whom previous cardiac operations made the thymus unavailable.

Resection and simple reconstruction was done with our conventional tracheal surgical techniques. In the patient with supracarinal stenosis and malacia (patient 9), complete sternotomy and transpericardial exposure of the carina provided excellent access. A sufficiently wide opening for generous tracheal anastomosis was created at the junction of the relatively transverse main bronchi. The anastomosis was covered with a pedicled strip of pericardium.

Resection of a connecting bridge bronchus was described by Cantrell and Guild.Go 1 Larger ovoid apertures were made at the site of origin of the stenotic bridge and at its insertion into the distal bronchial branching (patient 11). Great care was taken to widen these openings so that bronchial kinking would be minimized after approximation of the apertures. Cardiopulmonary bypass was used in this patient with single-ventricle physiology because atrial septectomy was performed concomitantly. The preexisting tracheostomy was left in place.

All patients were approached anteriorly, 3 through a cervical incision only, 3 through a collar incision with partial upper sternotomy, and 5 through a collar incision with complete sternotomy.


    Results
 Top
 Abstract
 Introduction
 Patients and methods
 Results
 Discussion
 References
 
All patients survived and obtained a widely patent and stable airway. Although wholly normal tracheal crosssectional area was not expected after slide tracheoplasty, the quadrupled enlargement was sufficient to relieve all symptoms. In all juvenile patients the repaired tracheal segment continued to grow. Growth has been observed in patients operated on at age 10 days, 3 months, 6 months, and 31/2 years during, respectively, follow-up periods of 1 year and 6 months and over 8, 5, and 7 years. Growth was predictable from sequential bronchoscopic observation of unoperated stenosis (patient 10) and by means of imaging.Go 22

Two of 7 slide tracheoplasty patients and 1 resection patient had a single suture line granuloma that was removed by means of a single bronchoscopy and did not recur. Tracheal stomal granulation tissue occurred in the resected patient referred with a preexisting tracheostomy, multiple prior operations, and on a ventilator. He was troubled by postoperative atelectasis and needed repeated aspiration bronchoscopies for secretions. He has continuing medical problems, but his airway remains entirely adequate. The 2 patients who had left pulmonary artery reimplantation showed reduced arterial flow. One underwent dilation of the arterial anastomosis, with improvement in flow. One subcutaneous sternal closure suture was removed. One patient had left vocal cord paralysis postoperatively, which recovered spontaneously.


    Discussion
 Top
 Abstract
 Introduction
 Patients and methods
 Results
 Discussion
 References
 
Short-segment congenital tracheal stenosis is generally best treated with now safe and standardized methods of tracheal resection and reconstruction. Long-segment stenosis is effectively and safely treated with slide tracheoplasty, as shown here. The advantages of this method over various techniques of patch tracheoplasty lie in the immediate reconstruction of trachea with tracheal tissue, providing a stable cartilaginous wall with normal epithelial lining. When carefully executed, the operation has not led to loss of viability from interference with tracheal blood supply or to failure of healing. Most of the lateral blood supply of the trachea is preserved. Excessive tension does not develop. Direct complications have been limited to a few one-time granulomas and a single transient injury to a recurrent laryngeal nerve. The conduct of the operation is simplified because cardiopulmonary bypass is usually not needed. Bypass is necessary only for accompanying cardiovascular procedures or borderline cardiac physiology. Routine postoperative endotracheal tube splinting is unnecessary, allowing prompt or early extubation except in patients with otherwise complicated anomalies. Tracheostomy was not performed.

Given the lesser tolerance of tension in juvenile tracheal anastomosis,Go 9 slide tracheoplasty should, in our opinion, be considered for all but the shortest stenotic segments. In infants it is also likely that a slide procedure would lessen the obstructive possibility of early narrowing caused by edema in a circular suture line after resection. Slide tracheoplasty is gaining acceptance.Go Go Go 16,23-30 Although most are reports of 1 or 2 patients, overall mortality, including this series, is 9%(Table 3).


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Table 3. Slide tracheoplasty for congenital tracheal stenosis
 
Patch tracheoplasty met a previously urgent need but should now perhaps be largely reserved for unusual problems. Insertion into the tracheal wall of a devascularized mesenchymal patch of pericardium or cartilage without epithelial covering, subsequent need for prolonged postoperative airway splinting by means of intubation, proclivity to form troublesome granulations, and the possibility, although infrequent, of necrosis or collapse, would all seem to favor use of slide tracheoplasty or resection and reconstruction as preferred techniques for treating long and short congenital tracheal stenosis, respectively.

Finally, resection of a very short bridge bronchus (patient 11) deserves attention for treatment of that specific anatomic situation.Go 1


    Footnotes
 
*Vertebral abnormalities, Anal atresia, Cardiac abnormalities, Tracheoseophageal fistula and/or Esophageal atresia, Renal agenesis and dysplasia, and Limb defects. Back


    References
 Top
 Abstract
 Introduction
 Patients and methods
 Results
 Discussion
 References
 

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  8. Couraud L, Moreau JM, Velly JF. The growth of circumferential scars of the major airways from infancy to adulthood. Eur J Cardiothorac Surg. 1990;4:521-6.[Abstract]
  9. Maeda M, Grillo HC. Effect of tension on tracheal growth after resection and anastomosis in puppies. J Thorac Cardiovasc Surg. 1973;65:658-68.[Medline]
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C. D. Wright, B. B. Graham, H. C. Grillo, J. C. Wain, and D. J. Mathisen
Pediatric tracheal surgery
Ann. Thorac. Surg., August 1, 2002; 74(2): 308 - 314.
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H. C. Grillo
Tracheal replacement: a critical review
Ann. Thorac. Surg., June 1, 2002; 73(6): 1995 - 2004.
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