Eur J Cardiothorac Surg 2001;19:777-784
© 2001 Elsevier Science NL
Tracheal surgery in children: an 18-year review of four techniques
Carl L. Backera,
Constantine Mavroudisa,
Mark E. Gerberb,
Lauren D. Holingerb
a Division of Cardiovascular Thoracic Surgery, Children's Memorial Hospital: Department of Surgery and Otolaryngology, Head and Neck Surgery, Northwestern University Medical School, Chicago, IL, USA
b Division of Otolaryngology, Children's Memorial Hospital: Department of Surgery and Otolaryngology, Head and Neck Surgery, Northwestern University Medical School, Chicago, IL, USA
Received 9 October 2000;
received in revised form 26 February 2001;
accepted 4 April 2001.
Corresponding author. Tel.: +1-773-880-4378; fax: +1-773-880-3054
e-mail: c-backer{at}nwu.edu
 |
Abstract
|
|---|
Objective: Review the short- and long-term outcomes of a single institution experience in infants with congenital tracheal stenosis, comparing four different operative techniques used from 1982 through 2000. Methods: Hospital and clinic records of 50 infants and children who had surgical repair of congenital tracheal stenosis secondary to complete tracheal rings were reviewed. Age at surgery ranged from 7 days to 72 months (median, 5 months, mean 7.8±12 months). Techniques included pericardial patch tracheoplasty (n=28), tracheal autograft (n=12), tracheal resection (n=8), and slide tracheoplasty (n=2). All procedures were done through a median sternotomy with cardiopulmonary bypass. Seventeen patients had a pulmonary artery sling (35%), and 11 had an intracardiac anomaly (22%). Results: There were three early deaths (6% early mortality), two after pericardial tracheoplasty and one after autograft. There were six late deaths (12% late mortality), five after pericardial tracheoplasty and one after slide tracheoplasty. Length of stay (median) was 60 days (pericardial tracheoplasty), 28 days (autograft), 14 days (resection), and 18 days (slide). Reoperation and/or stent placement was required in seven patients (25%) after pericardial tracheoplasty, in two patients (17%) after autograft, in no patients after resection, and in one patient (50%) after slide tracheoplasty. Conclusions: Our current procedures of choice for infants with congenital tracheal stenosis are resection with end-to-end anastomosis for short-segment stenoses (up to eight rings) and the autograft technique for long-segment stenoses. Associated pulmonary artery sling and intracardiac anomalies should be repaired simultaneously.
Key Words: Tracheal stenosis • Tracheal autograft • Complete tracheal rings • Pericardial tracheoplasty • Tracheal resection • Slide tracheoplasty
|
1. Introduction
|
|---|
In 1984 Dr Farouk Idriss from Children's Memorial Hospital reported the first successful use of an autologous pericardial patch as a technique of tracheoplasty in five infants with tracheal stenosis secondary to complete cartilage tracheal rings [1]. At that time most case reports reported the diagnosis, but had a fatal outcome [2]. The outlook for these infants was truly dismal, with a 43% survival rate with medical management [3]. This surgical breakthrough and the strong support from our otolaryngology division established our service as a center for referrals for tracheal stenosis which has allowed the development of a comprehensive program combining the respective skills of cardiothoracic surgery and otolaryngology. Over the past 18 years we have utilized four different techniques for infants with tracheal stenosis. These include pericardial tracheoplasty [1,4], tracheal autograft [5,6], and slide tracheoplasty [7,8] for infants with long segment stenosis; and tracheal resection for short segment stenosis [9,10]. Our techniques have evolved over time as we have gained experience with the advantages and disadvantages of the different techniques. The pericardial tracheoplasty was used nearly exclusively from June 1982 to November 1995; the tracheal autograft from January 1996 on. The two slide tracheoplasties were performed in 1996 and the tracheal resections from 1990 through 2000. The objective of this study was to review the short- and long-term outcomes of a single institution experience with infants with congenital tracheal stenosis, comparing four different operative techniques used from 1982 through 2000.
|
2. Patients and methods
|
|---|
Between June 1982 and September 2000, 50 infants and children with tracheal stenosis secondary to complete tracheal rings underwent surgical repair at Children's Memorial Hospital. There were 29 males and 21 females. Age ranged from 7 days to 72 months (mean age, 7.8±12 months, median age, 5 months). These children presented with respiratory distress, stridor, apnea, cyanosis, and as ‘difficult intubations.’ Most of these patients were initially managed either by neonatologists or pediatric intensivists prior to diagnosis. Once the diagnosis was made they were managed jointly by the pediatric intensive care service, the pediatric otolaryngology service, and our service (pediatric cardiovascular-thoracic surgery). In all cases diagnosis was by rigid bronchoscopy and all recent patients had echocardiography to rule out a pulmonary artery sling. All procedures were performed through a median sternotomy approach with the use of cardiopulmonary bypass. For isolated tracheal surgery, patients were placed on cardiopulmonary bypass with an aortic cannula and a single atrial cannula and cooled to 32°C. Patients remain in normal sinus rhythm throughout the procedure. Choice of operative technique was based on the length of the stenosis and the time period in which the child presented. In general pericardial tracheoplasty was used for long segment stenosis from 1982 through 1995. The autograft technique was used for long-segment stenosis from 1996 through 2000. Two patients had a slide tracheoplasty in 1996. Tracheal resection was used for short segment tracheal stenoses. If the patient has an associated pulmonary artery sling, repair is performed on cardiopulmonary bypass prior to the tracheal repair. The left pulmonary artery is transected, moved anterior to the trachea, and reimplanted into the main pulmonary artery [11]. If there is an associated intracardiac lesion, bicaval venous cannulation is used and the intracardiac lesion is repaired during a period of aortic cross-clamp time prior to the tracheal repair. The cardiac and pulmonary artery sling repairs are both done before opening the potentially contaminated tracheal lumen.
2.1. Pericardial tracheoplasty
An autologous pericardial patch is harvested from the diaphragmatic surface to the reflection of the pericardium at the great vessels. This patch is typically 4–6 cm in length and 2–3 cm in width. After cardiopulmonary bypass has been initiated an #11 blade is used to incise the trachea through the extent of the tracheal rings under bronchoscopic guidance. The pericardial patch is then sutured in place using interrupted 6.0 Vicryl (Polyglactin 910, Ethicon, NJ) sutures (Fig. 1). The suturing starts at the most inferior aspect of the incision adjacent to the carina and is carried superiorly. This helps to prevent the accumulation of blood and secretions in the tracheal lumen. Rigid bronchoscopy is performed after patch placement to confirm relief of stenosis and to suction the tracheobronchial tree clear. Then the endotracheal tube is reintroduced so that the tip of the tube is just above the carina. The endotracheal tube stents the patch open. Two small hemoclips are placed at the upper and lower aspect of the patch for radiographic identification of the patch location in the postoperative period. The mediastinum is filled with saline and the patient is ventilated with airway pressures up to 35–40 cm of water pressure to assess for leaks in the patch. The patient is ventilated and weaned from cardiopulmonary bypass. The patient is kept paralyzed, intubated, and ventilated for 1 week. Bronchoscopy is performed at 1 week postoperatively to assess the healing of the patch, sites of residual stenosis, and to remove any granulation tissue that has formed. Over the next week the patient is weaned from the ventilator and the patient is usually extubated 2–3 weeks following the procedure.
View larger version (52K):
[in this window]
[in a new window]
|
Fig. 1. Pathology complete tracheal rings, pericardial tracheoplasty. The pathology of complete tracheal rings is shown in the left panel. The normal tracheal cartilage with a posterior membranous portion and a normal lumen is shown superiorly. A complete tracheal ring with compromised tracheal lumen is shown inferiorly. A completed pericardial patch tracheoplasty is shown in the right panel. In cross section, the tracheal lumen is now normal.
|
|
2.2. Tracheal autografts
The entire trachea is extensively mobilized circumferentially. The isthmus of the thyroid gland is divided. Inferiorly the dissection is carried below the carina well onto the right and left main stem bronchi. The anterior trachea is incised with a #11 blade through the extent of the complete tracheal rings (Fig. 2A). After this an assessment is made as to how much trachea can be resected without causing excessive tension at the anastomosis (Fig. 2B). The excised midportion of the trachea is used as the free tracheal autograft (Fig. 2C). The two remaining ends of the trachea are anastomosed posteriorly with multiple interrupted 6.0 PDS sutures (Ethicon, Inc., Somerville, NJ) (Fig. 3A). These sutures are placed to keep the sutures and knots out of the tracheal lumen. The tracheal autograft previously harvested is used to patch the trachea anteriorly. The corners of the autograft are trimmed and it is anchored in place with interrupted 6.0 PDS sutures (Fig. 3B,C). If the autograft is not long enough to augment the entire anterior opening in the trachea, the inferior portion of the anterior tracheal opening (adjacent to the carina) is patched with the autograft. The upper aspect of the tracheal incision is patched with fresh autologous pericardium anchored with 6.0 PDS sutures in an interrupted fashion (Fig. 3B,C). Tissell glue (Baxter Health Care Corp., Glenlake CA) is used to seal the suture lines and the patch is checked for air leaks. Small hemoclips are placed in the soft tissue adjacent to the upper and lower aspects of the autograft for radiographic identification postoperatively. Our strategy for the endotracheal tube placement varies somewhat from the pericardial tracheoplasty. We keep the endotracheal tube either (a) above the autograft if there is no pericardial augmentation or (b) in the midportion of the autograft in patients that have had pericardial augmentation. In the patients where pericardium is used to augment the upper aspect of the tracheotomy the endotracheal tube acts as a stent for the pericardial patch. No patient had a neck brace or a chin stitch. The patient is kept paralyzed and ventilated for 3–5 days at which time bronchoscopy is performed. If the tracheal lumen is patent and the child is hemodynamically stable his paralysis is stopped and he is weaned from the ventilator over the next 3–5 days. Follow-up endoscopy is routinely performed just before extubation, just before hospital discharge, at 3 and 6 months postoperatively, and then yearly. In patients who have granulation tissue formation, residual stenosis, or other airway issues, bronchoscopy is performed more frequently.
View larger version (44K):
[in this window]
[in a new window]
|
Fig. 2. Tracheal Autograft. (A) An anterior longitudinal incision is performed through the complete extent of the tracheal rings. (B) The midportion of the trachea is excised to be used as the autograft. (C) The two remaining ends of the trachea are re-approximated posteriorly.
|
|
View larger version (41K):
[in this window]
[in a new window]
|
Fig. 3. Tracheal Autograft.(A) The posterior anastomosis is performed with interrupted PDS sutures, with the knots kept out of the tracheal lumen. (B) The autograft is prepared by trimming the corners of the graft. The pericardial patch is cut to size. (C) The autograft is sutured in place inferiorly at the carina. The pericardial patch is inserted superiorly to complete the repair.
|
|
2.3. Tracheal resection
After the trachea is mobilized, bronchoscopic identification of the stenosis is performed using a #25 needle to penetrate the trachea if needed. In most cases an anterior tracheotomy with a #11 blade is carefully performed through the stenotic rings to confirm the location of the complete tracheal rings. The complete tracheal rings are resected. If a more extensive resection than was predicted preoperatively is required, the patient can be converted to the autograft technique. Sufficient mobilization of the trachea with bilateral hilar release and freeing of the pulmonary artery from the pericardium is performed. In one case (the 72 month-old child) a hyoid release was used. End-to-end anastomosis is performed with multiple interrupted 6-0 PDS sutures. In some cases the endotracheal tube was positioned above the anastomsis, in some cases it was passed through the anastomosis. These patients were kept paralyzed for 24–48 h and then weaned from the ventilator and extubated. Surveillance bronchoscopy is performed prior to extubation and prior to hospital discharge.
2.4. Slide Tracheoplasty
The trachea is mobilized in its entirety. Bronchoscopic identification of the midportion of the stenosis is performed and the trachea is transected at this midpoint. The superior trachea is incised posteriorly through the extent of the tracheal rings and the inferior trachea is incised anteriorly through the extent of the tracheal rings. The two tracheal components are then ‘slid’ together and anastomosed with multiple interrupted 6.0 PDS sutures. The endotracheal tube is positioned in the midportion of the trachea. These patients were managed postoperatively in a similar fashion to the patients with tracheal resection.
|
3. Results
|
|---|
Pericardial patch tracheoplasty was performed in 28 patients, tracheal autograft in 12 patients, tracheal resection in eight patients, and slide tracheoplasty was performed in two patients. There were three early deaths (6% early mortality), two after pericardial tracheoplasty and one after autograft. There were six late deaths (12% late mortality), five after pericardial tracheoplasty and one after slide tracheoplasty. Seventeen patients (35%) had a pulmonary artery (PA) sling and 11 (22%) had a significant intracardiac anomaly. The associated intracardiac anomalies are shown in Table 1. Intracardiac anomalies were repaired at the time of tracheal repair, except in a few cases when patients were referred after cardiac surgery elsewhere. The number of complete tracheal rings involved ranged from two to 18 rings (mean 13.6±5). Nine patients (18%) had a tracheal right upper lobe bronchus. Eight patients had a patent ductus arteriosus, two had an absent right lung, two an absent left lung, and two a severely hypoplastic right lung. There were no complications specifically related to the use of cardiopulmonary bypass and no patients required reoperation for bleeding. The length of stay (median) was 60 days for pericardial tracheoplasty, 28 days for tracheal autograft, 14 days for tracheal resection, and 18 days for the slide tracheoplasty. Follow-up is complete in all patients. Mean follow-up after pericardial patch tracheoplasty is 10.9±4.7 years, after tracheal autograft is 2.6±1.5 years, after tracheal resection is 3.3 ± 3.2 years, and after slide tracheoplasty is 4 years. More specific details of the results are given for the individual procedures.
3.1. Pericardial patch tracheoplasty
This operation was performed from 1982 through 1995 on 28 patients. Mean age of the patients was 6.5±5.5 months, median age was 6 months. The number of tracheal rings involved ranged from 6 to 18, mean 15.2±4, median 18. There were two early deaths. One was at 2 weeks postoperatively in a 9 month-old that developed pseudomonas mediastinitis after having a significant air leak from the patch 6 days postoperatively. This was the only patient in the series where we used a running suture technique for the patch. The second death occurred in a 2-week-old infant who with an absent right lung had severe pulmonary hypertension following the procedure. This child was placed on extracorporeal membrane oxygenation (ECMO) in the operating room and died of an interventricular hemorrhage 1 week following the procedure. There were five late deaths after pericardial tracheoplasty. One was a complication from a tracheostomy that was performed 5 months after the tracheoplasty. Two patients died of complications of perforation of the trachea at the time of bronchoscopy (at 7 and 12 months postoperatively) at the referring institution following discharge from our hospital. One child died 15 months postoperatively of pulmonary hypertension. This child had Down's syndrome and had had simultaneous repair of a complete atrioventricular canal defect. The final late death was a child with multiple anomalies that died of colonic obstruction, bowel perforation, and sepsis 22 months following pericardial tracheoplasty. Six patients required reoperation for recurrent tracheal stenosis following pericardial tracheoplasty [12]. Two patients had a revision of their pericardial patch on cardiopulmonry bypass with a new patch of pericardium for recurrent tracheal stenosis. Four patients had placement of a rib cartilage graft on cardiopulmonary bypass in the distal trachea 2 to 6 months following pericardial tracheoplasty for distal tracheal and right mainstem bronchial stenosis. Three patients (two after reoperation) required five balloon expandable Palmaz stents following their pericardial tracheoplasty [13]. Six patients required a tracheostomy. Two patients had reoperations for air leaks within the first postoperative week. One of those patients had a pectoralis major muscle flap at the time of the reoperation and survived. The other patient died of mediastinitis as mentioned above. Significant risk factors for reoperation in the pericardial patch group were younger age at initial repair, associated pulmonary artery sling, and tracheal right upper lobe bronchus [13].
3.2. Results of tracheal autograft
This operation has been performed in 12 patients since January 1996. The mean age of these patients was 5.9±6 months, median age was 4.5 months. Eight of 12 patients were out-of-state referrals. The number of tracheal rings involved ranged from 6 to 18, mean 14.8±4, median 16 rings. The length of the autograft ranged from 1.3 to 2.5 cm (mean 2.0±0.5 cm). Pericardial augmentation was required in five of 12 patients. The mean length of the pericardial patch was 2.4±0.6 cm. There was one early death (8% early mortality) and there have been no late deaths. The early death occurred in a 6-month old child that had tetralogy of Fallot, an anomalous left anterior descending coronary artery arising from the right coronary artery, and agenesis of the left kidney. He had previously had two median sternotomies for a right ventricular outflow tract patch and later a modified Blalock–Taussig shunt. Following tracheal autograft and intracardiac repair of tetralogy of Fallot, he had low cardiac output and required ECMO support 12 h postoperatively for 6 days. He died 26 days postoperatively of sepsis and anasarca. He had an autopsy which revealed multiple myocardial infarctions and necrotizing pneumonia. At the time of autopsy his trachea was widely patent and the autograft had healed. There were two significant complications following the tracheal autograft. One 2 month old boy had complete repair of pulmonary artery sling, tetralogy of Fallot, and tracheal autograft; he also had a tracheal right upper lobe bronchus. He was the only child in the series in which I divided the autograft in half longitudinally and used both halves separately to patch the tracheal opening in an effort not to use pericardium. He had disruption of an infected autograft during the time of bronchoscopy 4 days postoperatively. He underwent emergent re-sternotomy, placement on cardiopulmonary bypass, and replacement of the entire autograft (lysed by pseudomonous) with a cryopreserved aortic homograft. The right pectoralis major muscle was brought through an intercostal window to treat the mediastinitis. He required placement of a balloon expandable Palmaz tracheal stent at 4 weeks and another Palmaz tracheal stent at 3 months postoperatively. He also required a tracheostomy 7 months postoperatively. He had a prolonged hospital course of nearly 11 months, but is currently at home and doing well. The second patient with significant postoperative complications had recurrent tracheal stenosis related to the pericardial patch and required a Palmaz stent at 3 months postoperative and a tracheostomy at 4 months postoperative. That patient also had a prolonged (5 months) hospital course. She is now 3 years postop and is doing well at home. Two other patients required a temporary tracheostomy. The other patients are currently asymptomatic from a respiratory standpoint. On serial bronchoscopic examination the trachea appears to be growing normally with the patient and the autograft is difficult to differentiate from the surrounding trachea.
3.3. Tracheal resection
Eight tracheal resections for shorter segment of tracheal stenosis were performed from 1990 through 2000. The mean age of the tracheal resection patients was 16.5±26 months, the median age was 4 months (2 outliers, 42 and 72 months old). The number of complete tracheal rings ranged from two to eight, mean 5.5±2.2, median five rings. There were no early or late deaths after tracheal resection. No patient required a tracheostomy. The mean hospital stay was 14 days and there were no significant postoperative complications.
3.4. Slide tracheoplasty
Two slide tracheoplasties were performed in 1996. One child was 3 months old, had 18 complete tracheal rings, and had simultaneous repair of double outlet right ventricle. She was discharged 18 days postoperatively. That child has had no further complications. The other child had pulmonary atresia and ventricular septal defect, and had 12 complete tracheal rings. She had a simultaneous modified Blalock–Taussig shunt. The child had recurrence of tracheal stenosis secondary to granulation tissue and a ‘figure of 8’ configuration of the trachea cross-section. The child required a postoperative Palmaz tracheal stent and a tracheostomy. She was hospitalized for 5 months, never able to come off the ventilator, and died of aortic valve endocarditis.
|
4. Discussion
|
|---|
Infants with tracheal stenosis secondary to complete tracheal rings often present with life-threatening respiratory distress. As recently as 20 years ago most case reports described this rare anomaly, but had a fatal outcome [2]. In June 1982 Dr Farouk Idriss first performed a successful pericardial patch tracheoplasty at our institution for an infant with complete tracheal rings [1]. That surgical breakthrough and the skilled bronchoscopic support from our otolaryngology division led to our institution becoming a referral center for infants with tracheal stenosis and led to the experience with four different techniques in 50 patients that is reported herein.
At the time that the pericardial tracheoplasty was first being successfully performed at our institution the only other described successful surgical option for these infants was a cartilage tracheoplasty [14]. Although we used cartilage grafts in four patients for reoperation after pericardial patch tracheoplasty, we have not felt it as easy to work with as pericardium for long segment stenoses. It tends to maintain its natural ‘torque’, which does not necessarily conform to the longitudinal tracheal opening. The cartilage is rigid and it is sometimes difficult to obtain an airtight seal. We and others have had the experience where the cartilage seems to ‘fall’ into the trachea and actually obstruct the lumen. A summary of several series using the cartilage tracheoplasty is shown in Table 2. The overall mortality in 22 patients was 27%. We elected to continue with the pericardial tracheoplasty because of our success with this operation. Pericardium is readily available autologous material, is quite easy to obtain an air-tight seal, and becomes re-epithelialized with ciliated pseudostratified columnar epithelium [18]. In the first report from our institution by Idriss from 1984, 5 consecutive infants had successful patch tracheoplasty [1]. However, as we gained more experience with this technique and applied it to more patients, some with more extensive disease (i.e. absent right or left lung, associated pulmonary artery sling, associated intracardiac anomalies) we began to note more complications of the technique [4]. Although these patients survived the operation (early mortality only 7%) they often had a prolonged hospital stay secondary to patch tracheomalacia, recurrent stenosis, and exuberant granulation tissue. Mean hospital stay was 60 days. Granulation tissue was particularly problematic at the distal end of the trachea where the pericardial patch interfaced with the native trachea and carina where frequently the end of the endotracheal tube was lodged. The outlook for these patients was an 18% long-term mortality and a 25% incidence of either re-operation for recurrent stenosis or endoscopic placement of a Palmaz stent. Bando and associates reported good results with the pericardial patch tracheoplasty, 12 patients between 1984 and 1995 with one operative and one late death; a 17% mortality [19]. Andrews, however, reported a 47% mortality in 13 patients treated from 1985 to 1991 with a pericardial patch tracheoplasty [20]. A summary of the various series results with pericardial patch tracheoplasty is shown in Table 3, overall mortality in 53 patients was 28%.
The slide tracheoplasty was first described in two patients by Tsang and Goldstraw in 1989, one patient survived, the other died [7]. Grillo reported good results with the slide tracheoplasty in four patients (two patients, 19 years old) [21]. We attempted this technique in two patients in 1996 [8]. In one patient the technique was quite successful and the child was discharged in 18 days. However, the other child had significant complications from the slide tracheoplasty necessitating a Palmaz tracheal stent and a tracheostomy, and the child was never weaned from the ventilator and died 5 months postoperatively. We have several concerns with the slide tracheoplasty. The slide tracheoplasty procedure requires precise identification of the midportion of the stenosis before the initial tracheal transsection. This is not always easy to identify either bronchoscopically or from external examination. If the trachea is transected initially at the wrong site, the two portions of trachea will not correspond and there will be a residual stenosis. When performing the slide tracheoplasty the upper trachea has to be lifted anteriorly for an initial series of posterior sutures that are difficult to see and tie. Air leaks along the posterior suture line are particularly difficult to deal with. In the report by Grillo he noted having to perform an anterior vertical tracheal incision after the repair to deal with a persistent air leak ‘in an inaccessible spot’ [21]. When the slide tracheoplasty is completed the spring of the tracheal rings tends to form a ‘figure of eight’ contour in cross-section. This unnatural architectural configuration is a set-up for forces leading to granulation tissue and tracheomalacia. Published results of the slide tracheoplasty are shown in Table 4, with a mortality of 20% in only ten patients.
In late 1995 we became aware of the efforts of several groups in Europe to use tracheal homograft for rescue procedures on patients that had failure of previous operations for congenital tracheal stenosis. The landmark report by Elliott et al [23] showed us the utility of a tracheal homograft in these critically ill patients. At this same time we operated on a patient with an extremely long trachea and decided to resect a number of the tracheal rings and perform an end-to-end anastomosis posteriorly prior to the pericardial tracheoplasty. In this particular case the removed tracheal rings were simply sent to pathology. After reading the report by Elliott et al I was intrigued by the fact that the piece of the trachea that I discarded looked exactly like the tracheal homograft that Elliott was so successful in using. Because of this in the next patient that underwent repair of tracheal stenosis I elected to use the excised midportion of the trachea as a distal ‘autograft.’ That particular child had an extraordinarily diminutive trachea and also had a pulmonary artery sling and a ventricular septal defect. The upper portion of the trachea was augmented with pericardium. The postoperative course of that child was particularly good; the child was discharged from the hospital 20 days following the surgery and did not require readmission. This first initial successful experience caused us to continue to use the autograft technique in the next 11 patients in our series with long segment congenital tracheal stenosis. We were initially concerned with the possibilities of dehiscence between the autograft and pericardium and autograft ischemia. However, no patient has had either of these complications. In fact the autograft usually appears pale but pink at the time of operating room bronchoscopy, and by 5 to 7 days postoperatively appears to be revascularized and a normal pink color. In a model of the tracheal autograft developed by Dr Dodge-Khatami in our laboratory, 30 rabbits have had a successful autograft procedure (six tracheal rings) with no autograft dehiscence or ischemia [24]. We demonstrated in this model that the topical application of VEGF (vascular endothelial growth factor) enhanced healing of the autograft with reduced luminal stenosis, submucosal fibrosis, and inflammatory infiltrate. Tracheal architecture was preserved in VEGF treated animals and they had greater microvascular density.
In the autograft group one patient died from myocardial infarction and pneumonia 26 days after tetralogy repair, tracheal autograft, and postoperative ECMO support. Another child had dehiscence of the autograft but was successfully rescued with an aortic homograft patch of the trachea. Four patients required tracheostomies. The other patients in this series are currently asymptomatic from a respiratory standpoint. The principles of the tracheal autograft which make it our current procedure of choice for infants with long-segment congenital tracheal stenosis are that it: (1) uses all autologous material for the repair; (2) is technically easy to perform; (3) is architecturally sound; (4) the autograft is already lined with respiratory epithelium; (5) the cartilage intrinsically maintains its contour; (6) there is potential for growth, and (7) it is readily available, the trachea in these infants is often excessively long.
The technique of tracheal resection works very well for infants with shorter segment tracheal stenosis. Jonas recommended distal tracheal resection and pulmonary artery translocation for the repair of pulmonary artery sling [10]. Heinemann has reported success in nine of ten infants with resection of shorter segments of tracheal stenosis with the use of cardiopulmonary bypass [9]. Cotter reported six patients that underwent resection and end-to-end anastomosis, with excellent results [25]. In our series the patients who had a short enough segment to undergo successful tracheal resection had an excellent outcome. We have successfully resected as many as eight rings. Their time to extubation was short and their hospital stay was shorter than any of the other techniques in our series. A summary of published results of tracheal resection is shown in Table 5, 24 patients, 8% mortality.
Infants with tracheal stenosis secondary to complete tracheal rings present a significant challenge in their clinical management. Management of these infants requires very close co-operation between the pediatric cardiothoracic surgeon and the otolaryngologist. Diagnoses in our series have been by rigid bronchoscopy in all cases. All patients should have an echocardiogram to rule out a pulmonary artery sling. We recommend resection and end-to-end anastomosis facilitated with cardiopulmonary bypass for short segment stenosis (eight complete tracheal rings or less). For patients with long segment congenital tracheal stenosis (>8 rings) we recommend repair with a tracheal autograft with or without pericardial augmentation. Other surgeons have reported excellent results using the slide tracheoplasty. Pulmonary artery sling and associated intracardiac defects should be repaired simultaneously with the repair of the tracheal stenosis. The postoperative management of these patients is quite complex and requires extreme attention to detail and vigilance with regard to airway management. Patients with patch dehiscence were managed successfully with vascularized muscle flaps. The multidisciplinary approach to these patients with close collaboration and co-ordination between otolaryngology and thoracic surgery and ongoing evaluation of our techniques for these infants is the key to successful long-term outcomes for these critically ill infants.
|
Footnotes
|
|---|
Presented at the 14th Annual Meeting of the European Association for Cardio-thoracic Surgery, Frankfurt, Germany, October 7–11, 2000.
|
Appendix A. Conference discussion
|
|---|
Dr A. Corno (Lausanne, Switzerland): In the University Hospital of Lausanne, we fully agree with your indication of treating congenital tracheal stenosis and associated congenital difficulty at the same time, but we disagree with your other conclusion regarding the technique. We adopted slide tracheoplasty in the last period, and we have a series of all infants with severe stenosis like your patients, that means, 2, 3 mm internal diameter up to 80% of the tracheal length and with long-term segment tracheal stenosis. In all of them we performed the slide tracheoplasty in the same simultaneous cardiopulmonary bypass period for repair of heart defect from a simple VSD to complex double outlet right ventricle requiring a cardiac conduit. In all of them, the internal tracheal diameter has been enlarged up to 200 or 250% of the previous diameter, and in all of them extubation was obtained in less than 3 days.
So my question is, based on what kind of hypothesis you can discard or condemn a technique which has two major advantages: the first, it can be applied to any anatomical type of tracheal stenosis, and second, it doesn't require any autologous or homologous material or a pericardial patch.
Dr Backer: I think the slide tracheoplasty is the operation that comes closest to approaching the success that we have had with the tracheal autograft. In our experience, the slide tracheoplasty worked very well for one patient. The second patient I tried it on had considerable problems with granulation tissue formation, and the spring of the cartilages tended to make the tracheal lumen look like a figure of eight. This patient required a stent and a tracheostomy and eventually died 5 months later.
The other issue that I have with the slide tracheoplasty is that I found the posterior portion of the anastomosis superiorly somewhat difficult to do. Dr Grillo in his paper mentioned that they had a significant air leak from one of their patients in this area; they had to open the trachea anteriorly to control that leak. The beauty of the autograft operation I find is all of the suturing is anterior and it is architecturally sound, and, as I mentioned, I feel easier to apply. But I think the slide tracheoplasty is an excellent technique.
Dr A. Merounko (Tomsk, Russia): I would like to ask, what was the main reasons of hospital mortality?
Dr Backer: In the pericardial tracheoplasty, one child had an air leak, developed mediastinitis, had two reoperations and died of mediastinitis. One other child that had an absent lung had severe pulmonary hypertension, was placed on ECMO, and that child died of an intraventricular hemorrhage one week post-op. The other early death in the autograft group was a child that had had two previous median sternotomies, had simultaneous repair of tetralogy of Fallot and died of myocardial infarction and pneumonia 26 days post-op. That child also required ECMO support for 6 days. Of the late deaths after pericardial tracheoplasty, two actually died of perforation of the trachea during bronchoscopy at the referring institution. I mentioned that one of the keys to our success has been the input from our otolaryngology colleagues with their expert bronchoscopy. This is not an easy patient to do an endoscopy on when they have a difficult airway situation postoperatively.
Dr T. Tlaskal (Prague, Czech Republic): I have experience with reconstruction of tracheal stenosis in three patients operated in course of the last 3 years. Recently we have seen an 8-month-old infant with tetralogy of Fallot, small pulmonary arteries, left superior vena cava and right aortic arch. It was only after the repair of tetralogy that severe malformation of the lower tracheobroncheal tree was disclosed. Tracheobronchoscopy and tracheobronchography demonstrated presence of the right tracheal bronchus with severe stenosis and hypoplasia of the distal part of trachea. The repair of tracheal malformation was performed on cardiopulmonary by-pass by resection of the right upper lobe and plastic repair of the hypoplastic segment of the lower part of trachea. As a big piece of pericardium had been utilised previously for repair of congenital heart lesion xenopericardium was used for patch repair of tracheal stenosis. The patient is doing well 6 months after surgery.
I would like to ask you what would you do at reoperation in this particular patient if no autologous pericardium was available and, in general, what is your policy at reoperations after repair of tracheal stenosis.
Dr Backer: Frequently, even if they have had a previous median sternotomy, I have been able to find some pericardium to use. The one patient that I could not find pericardium that dehisced the autograft, I used an aortic homograft, which was readily available for our cardiac cases, and that worked quite well.
|
References
|
|---|
-
Idriss F.S., DeLeon S.Y., Ilbawi M.N., Gerson C.R., Tucker G.F., Holinger L. Tracheoplasty with pericardial patch for extensive tracheal stenosis in infants and children. J Thorac Cardiovasc Surg 1984;88:527-536.[Abstract]
-
Janik J.S., Nagaraj H.S., Yacoub U., Groff D.B. Congenital funnel-shaped tracheal stenosis: an asymptomatic lethal anomaly of early infancy. J Thorac Cardiovasc Surg 1982;83:761-766.[Abstract]
-
Benjamin B., Pitkin J., Cohen D. Congenital tracheal stenosis. Ann Otol Rhinol Laryngol 1981;90:364-371.[Medline]
-
Cosentino C.M., Backer C.L., Idriss F.S., Holinger L.D., Gerson C.R., Mavroudis C. Pericardial patch tracheoplasty for severe tracheal stenosis in children: intermediate results. J Pediatr Surg 1991;26:879-885.[Medline]
-
Backer C.L., Mavroudis C., Dunham M.E., Holinger L.D. Repair of congenital tracheal stenosis with a free tracheal autograft. J Thorac Cardiovasc Surg 1998;115:869-874.[Abstract/Free Full Text]
-
Backer C.L., Mavroudis C., Dunham M.E., Holinger L. Intermediate-term results of the free tracheal autograft for long segment congenital tracheal stenosis. J Pediatr Surg 2000;35:813-819.[Medline]
-
Tsang V., Murday A., Gillbe C., Goldstraw P. Slide tracheoplasty for congenital funnel-shaped tracheal stenosis. Ann Thorac Surg 1989;48:632-635.[Abstract]
-
Dayan S.H., Dunham M.E., Backer C.L., Mavroudis C., Holinger L.D. Slide tracheoplasty in the management of congenital tracheal stenosis. Ann Otol Rhinol Laryngol 1997;106:914-919.[Medline]
-
Heinemann M.K., Ziemer G., Sieverding L., Baden W., Kaulitz R., Luhmer I. Long-segment tracheal resection in infancy utilizing extracorporeal circulation. In: Imai Y., Momma K., eds. Proceedings of the 2nd World Congress of Pediatric Cardiology and Cardiac Surgery. New York: Futura Publishing Co, 1998:711-713.
-
Jonas R.A., Spevak P.J., McGill T., Castaneda A.R. Pulmonary artery sling: primary repair by tracheal resection in infancy. J Thorac Cardiovasc Surg 1989;97:548-550.[Abstract]
-
Backer C.L., Mavroudis C., Dunham M.E., Holinger L. Pulmonary artery sling: results with median sternotomy, cardiopulmonary bypass, and reimplantation. Ann Thorac Surg 1999;67:1738-1745.[Abstract/Free Full Text]
-
Backer C.L., Mavroudis C., Dunham M.E., Holinger L.D. Reoperation after pericardial patch tracheoplasty. J Pediatr Surg 1997;32:1108-1111.[Medline]
-
Furman R.H., Backer C.L., Dunham M.E., Donaldson J., Mavroudis C., Holinger L.D. The use of balloon expandable metallic stents in the treatment of pediatric tracheomalacia and bronchomalacia. Arch Otolaryngol Head Neck Surg 1999;125:203-207.
-
Kimura K., Mukohara N., Tsugawa C., Matsumoto Y., Sugimura C., Murata H., Itoh H. Tracheoplasty for congenital stenosis of the entire trachea. J Pediatr Surg 1982;17:869-871.[Medline]
-
Tsugawa C., Kimura K., Muraji T., Nishijima E., Matsumoto Y., Murata H. Congenital stenosis involving a long segment of the trachea: further experience in reconstructive surgery. J Pediatr Surg 1988;23:471-475.[Medline]
-
Jaquiss R.D.B., Lusk R.P., Spray T.L., Huddleston C.B. Repair of long-segment tracheal stenosis in infancy. J Thorac Cardiovasc Surg 1995;110:1504-1512.[Abstract/Free Full Text]
-
Kamata S., Usui N., Ishikawa S., Kitayama Y., Sawai T., Okuyama H., Fukui Y., Okada A. Experience in tracheobronchial reconstruction with a costal cartilage graft for congenital tracheal stenosis. J Pediatr Surg 1997;32:54-57.[Medline]
-
Cheng A.T.L., Backer C.L., Holinger L.D., Dunham M.E., Mavroudis C., Gonzalez-Crussi F. Histopathological changes following pericardial patch tracheoplasty. Arch Otolaryngol Head Neck Surg 1997;123:1069-1072.
-
Bando K., Turrentine M.W., Sun K., Sharp T.G., Matt B., Karmazyn B., Heifetz S.A., Stevens J., Kesler K.A., Brown J.W. Anterior pericardial tracheoplasty for congenital tracheal stenosis: intermediate to long-term outcomes. Ann Thorac Surg 1996;62:981-989.[Abstract/Free Full Text]
-
Andrews T.M., Cotton R.T., Bailey W.W., Myer C.M., III, Vester S.R. Tracheoplasty for congenital complete tracheal rings. Arch Otolaryngol Head Neck Surg 1994;120:1363-1369.
-
Grillo H.C. Slide tracheoplasty for long-segment congenital tracheal stenosis. Ann Thorac Surg 1994;58:613-621.[Abstract]
-
Lang F.J., Hurni M., Monnier P. Long-segment congenital tracheal stenosis: treatment by slide-tracheoplasty. J Pediatr Surg 1999;34:1216-1222.[Medline]
-
Elliott M.J., Haw M.P., Jacobs J.P., Bailey C.M., Evans J.N.G., Herberhold C. Tracheal reconstruction in children using cadaveric homograft trachea. Eur J Cardio-thorac Surg 1996;10:707-712.[Abstract]
-
Dodge-Khatami A., Backer C.L., Crawford S.E., Cook K.E., Holinger L.D., Mavroudis C. Topical vascular endothelial growth factor (VEGF) enhances free tracheal autograft healing in an experimental rabbit model of tracheal reconstruction. Surg Forum 1999:146-147.
-
Cotter C.S., Jones D.T., Nuss R.C., Jonas R. Management of distal tracheal stenosis. Arch Otolaryngol Head Neck Surg 1999;125:325-328.
This article has been cited by other articles:
|
|
|
|
 
P. B. Manning, M. J. Rutter, and W. L. Border
Slide Tracheoplasty in Infants and Children: Risk Factors for Prolonged Postoperative Ventilatory Support
Ann. Thorac. Surg.,
April 1, 2008;
85(4):
1187 - 1192.
[Abstract]
[Full Text]
[PDF]
|
|
|
|
|
|
|
J. Brazdil, T. Tlaskal, P. Vojtovic, P. Pohunek, and M. Cihar
Slide Plasty of Trachea and Right Main Bronchus in a Newborn
Ann. Thorac. Surg.,
March 1, 2008;
85(3):
1096 - 1097.
[Abstract]
[Full Text]
[PDF]
|
|
|
|
|
|
|
T. Loukanov, C. Sebening, M. Gorenflo, and S. Hagl
Management of congenital tracheal stenosis in infancy
Eur. J. Cardiothorac. Surg.,
February 1, 2007;
31(2):
331 - 331.
[Full Text]
[PDF]
|
|
|
|
|
|
|
L. M. Phipps, J. A. Raymond, and T. M. Angeletti
Congenital Tracheal Stenosis.
Crit. Care Nurse,
June 1, 2006;
26(3):
60 - 69.
[Full Text]
[PDF]
|
|
|
|
|
|
|
J. L. Anton-Pacheco, I. Cano, J. Comas, L. Galletti, L. Polo, A. Garcia, M. Lopez, and D. Cabezali
Management of congenital tracheal stenosis in infancy.
Eur. J. Cardiothorac. Surg.,
June 1, 2006;
29(6):
991 - 996.
[Abstract]
[Full Text]
[PDF]
|
|
|
|
|
|
|
C. Karagiannidis, V. Velehorschi, B. Obertrifter, H.-N. Macha, A. Linder, and L. Freitag
High-Level Expression of Matrix-Associated Transforming Growth Factor-{beta}1 in Benign Airway Stenosis
Chest,
May 1, 2006;
129(5):
1298 - 1304.
[Abstract]
[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]
|
|
|
|
|
|
|
A. Macedo, E. Fadel, G.-M. Mazmanian, V. de Montpreville, M. German-Fattal, S. Mussot, A. Chapelier, and P. G. Dartevelle
Heterotopic en bloc tracheobronchial transplantation with direct revascularization in pigs
J. Thorac. Cardiovasc. Surg.,
June 1, 2004;
127(6):
1593 - 1601.
[Abstract]
[Full Text]
[PDF]
|
|
|
|
|
|
|
M. H. Hines and D. R. Hansell
Elective extracorporeal support for complex tracheal reconstruction in neonates
Ann. Thorac. Surg.,
July 1, 2003;
76(1):
175 - 179.
[Abstract]
[Full Text]
[PDF]
|
|
|
|
|
|
|
M. Ricci, G. A. Cohen, D. Roebuck, and M. J. Elliott
Management of complex tracheo-aortic fistula following neonatal tracheal reconstruction
Ann. Thorac. Surg.,
April 1, 2003;
75(4):
1325 - 1328.
[Abstract]
[Full Text]
[PDF]
|
|
|
Top
Abstract
1. Introduction
