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Ann Thorac Surg 1996;62:981-989
© 1996 The Society of Thoracic Surgeons
Sections of Cardiothoracic Surgery, Pediatric Otolaryngology, Pediatric Radiology, and Division of Pediatric Pathology, James W. Riley Hospital for Children and Indiana University Medical Center, Indianapolis, Indiana
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Abstract |
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Methods. To assess the intermediate and long-term outcomes of patients having repair with anterior pericardial tracheoplasty, we reviewed case histories of 12 patients (1984 to present). The median age was 6.7 months (range, 1 to 98 months), and the median weight was 6.0 kg (range, 0.97 to 42 kg). All patients underwent anterior pericardial tracheoplasty through a median sternotomy during partial normothermic cardiopulmonary bypass. An average of 13 tracheal rings (range, five to 23) were divided anteriorly, and a patch of fresh autologous pericardium was used to enlarge the trachea by 1.5 times the predicted diameter for patient age and weight.
Results. There was one hospital death, and all but 2 patients are long-term survivors. All but 1 current survivor remain asymptomatic, with no bronchoscopic evidence of airway obstruction or granulation on the pericardial patch. All survivors examined have normal tracheal growth and development, with a median follow-up of 5.5 years (range, 1 to 11 years).
Conclusions. Anterior pericardial tracheoplasty for congenital tracheal stenosis provides excellent results at intermediate to long-term follow-up.
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Introduction |
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TopFootnotesAbstractIntroductionPatients and MethodsResultsCommentReferences |
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Long-segment tracheal stenosis in infants and children is difficult to manage and can be life-threatening. Patients frequently have associated cardiac, respiratory, or other gastrointestinal anomalies that may confuse the diagnosis at initial presentation. The rarity of congenital tracheal stenosis has not allowed sufficient experience for the development of a standard treatment protocol. Several operative techniques have been described, including slide tracheoplasty [1, 2], rib grafting [3, 4], and pericardial patch techniques [5–7], but have had varying results. The assessment of optimal repair remains controversial because of a lack of midterm to long-term follow-up data. Anterior pericardial tracheoplasty has been used at our institution over the last 11 years. This report describes the intermediate to long-term outcomes of anterior pericardial tracheoplasty for the management of long-segment tracheal stenosis.
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Patients and Methods |
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TopFootnotesAbstractIntroductionPatients and MethodsResultsCommentReferences |
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). Eight patients (67%) had associated cardiac anomalies: combined ventricular septal defect and patent ductus (3), pulmonary arterial sling (1), congenital aortic stenosis (1), complete atrioventricular canal defect (1), tetralogy of Fallot (1), and patent ductus (1) (Table 2). The stenosis involved five to 22 complete rings (average, 13 rings) in all patients. Sixteen percent to 53% of the predicted normal diameter was measured in these patients preoperatively (Table 3). Ten patients required mechanical ventilation from 8 days to 2 years before tracheoplasty. Three of these had prior tracheostomy. Two were allowed to heal after oral intubation and before anterior pericardial tracheoplasty. One was left open with a tracheal button. Three patients initially underwent complete repair of associated cardiac anomalies (patient 3) or palliation (patients 4 and 10) (see Table 2). Severe tracheal stenosis was found during intubation in these 3 patients, which prevented extubation after repair. One premature baby (patient 2), born at 28 weeks' gestation with a weight of 970 g, had emergency anterior pericardial tracheoplasty at the age of 1 month, and had completed repair of ventricular septal defect and patent ductus arteriosus ligation 6 months later.
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). A 20-month-old boy (patient 7) with congenital aortic stenosis had four episodes of respiratory arrest at home, necessitating cardiopulmonary resuscitation. An 8-year-old boy (patient 11) had been diagnosed as having patent ductus arteriosus and long-segment tracheal stenosis at 4 years of age. He underwent patent ductus arteriosus ligation at age 4, but his tracheal stenosis was left untreated because he was minimally symptomatic. However, progression of his shortness of breath necessitated tracheoplasty 4 years later. In patient 12, a pulmonary artery sling was found at the time of tracheoplasty, and a combined repair was performed.
Fiberoptic and rigid bronchoscopy were the principal techniques used for confirming tracheal stenosis preoperatively. Tracheal diameter and length of the stenosis were measured preoperatively, when possible, and confirmed in the operating room after initiation of cardiopulmonary bypass. A separate tracheal origin of the right upper-lobe bronchus was identified in 3 patients (see Table 2). Neither pulmonary agenesis nor tracheoesophageal fistula was encountered. Conventional computed tomography (CT) was performed preoperatively in 2 patients (6 and 8) but did not provide additional useful information. Cardiac catheterization was performed in all patients to identify associated cardiac anomalies and to rule out the presence of a vascular ring or pulmonary arterial sling.
Operative Technique
Details of our operative technique have been described previously [7]. A median sternotomy was performed, and the thymus was divided between lobes and retracted laterally. The pericardium was then opened and the innominate vessels were mobilized. The anterior trachea was exposed between the ascending aorta and the superior vena cava from the cricoid to the carina. The lateral blood supply to the trachea and the recurrent laryngeal nerves were not disturbed. Patients were given heparin and were cannulated for cardiopulmonary bypass through the right atrium and ascending aorta. Normothermic cardiopulmonary bypass was instituted, and the endotracheal tube was removed. Fiberoptic bronchoscopy was then performed to confirm the degree and extent of stenosis. The trachea was incised in the anterior midline through the entire stenotic segment. One normal ring of the trachea was opened superiorly and inferiorly to ensure complete relief of obstruction. This incision occasionally extended onto the mainstem bronchi. Three to four tacking stitches were placed on the anterolateral margins of the trachea to separate the tracheal edges further. A rectangular piece of fresh autologous pericardium was harvested and tailored to enlarge the tracheal lumen to 1.5 times the predicted age-adjusted normal diameter (Fig 1). The pericardium was sutured to the outer three fourths of the tracheal edge with continuous running 6-0 polydioxanone (PDS) suture (Ethicon, Inc, Somerville, NJ). Care was taken not to place the sutures in the tracheal mucosa so as to avoid any suture material in the lumen of the airway, which would stimulate granulation tissue formation (Fig 2, inset). Several partial-thickness sutures were placed on the outer surface of the pericardial patch anteriorly to suspend it to the surrounding mediastinal tissues (Fig 3). After completion of the tracheal suture line, the anesthesiologist reinserted the endotracheal tube and increased the tracheal airway pressure temporarily to 50 mm Hg to confirm an airtight anastomosis. Additional sutures were sometimes placed on each side of the trachea to distract the edges as another measure to prevent postoperative migration of the tracheal edges (see Figs 2, 3). However, we did not cover or buttress the pericardial repair. An endotracheal tube was placed under direct visual guidance, with its distal end positioned either proximal to the pericardial patch for a distal short-segment repair, or at the level of the midpatch for long repairs. Mechanical ventilation was then begun, and the patient was weaned from bypass. After heparin reversal, the sternotomy was closed over a mediastinal drain in a standard fashion.
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Long-Term Follow-up
The growth of the repaired trachea was evaluated using spiral CT scan (Elscint Twin; Elscint Ltd, Haifa, Israel) without intravenous contrast. From the CT data, 3-dimensional shaded surface display images and coronal and sagittal multiplanar reconstructions were created. The coronal diameter and the cross-sectional area of the native uninvolved trachea as well as the reconstructed segment were measured in three different axial images. The mean values of the diameter and the cross-sectional area were then calculated for the native and reconstructed segments of the trachea. These measurements were compared with normal values [8]. Bronchoscopic examination was performed routinely at 1 month after operation in all patients, and selectively thereafter in patients with clinical indications. Complete pulmonary function tests were performed postoperatively in 4 patients able to cooperate with the examination (>5 years old). Postoperative functional status was determined by direct evaluation of the patient by either a pediatric pulmonologist, otolaryngologist, or the cardiothoracic surgeon. If recent (<6 months) follow-up had not been obtained, the patients were contacted by telephone during the month of January 1996.
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Results |
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TopFootnotesAbstractIntroductionPatients and MethodsResultsCommentReferences |
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). The diameter of the stenotic segment ranged from 16% to 53% (mean, 30%) of the normal expected tracheal diameter. The mean cardiopulmonary bypass time was 108 minutes, with a range of 73 to 162 minutes. Postoperative bronchoscopy was performed at a median frequency of twice per patient. In 2 patients (patients 2 and 10), marked stenosis developed at their previous tracheostomy sites, which necessitated repeat anterior pericardial tracheoplasty (see Table 2). In another 2 patients (patients 11 and 12), a small amount of granulation tissue developed at the distal site of their repair, but this did not cause substantial stenosis and could be removed easily at bronchoscopy (Table 4). We have encountered no necrosis of the pericardial patch in any of these 12 patients.
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There was one late death. A 2-year-old boy (patient 5) had been hospitalized since birth (at 28 weeks of gestation) because of bronchopulmonary dysplasia and long-segment tracheal stenosis. A tracheostomy was placed at 2 weeks of age. An upper tracheal and subglottic stenosis was relieved with an autologous rib graft performed by the otolaryngology surgical service, but he continued to be ventilator dependent. His entire intrathoracic trachea was subsequently enlarged by anterior pericardial tracheoplasty, allowing him to be successfully extubated on the 18th postoperative day. He went home on postoperative day 28 with a tracheal button. He was asymptomatic for 2 years until a seizure and respiratory distress developed. He died in the emergency room after attempted cardiopulmonary resuscitation. Cultures of his blood revealed gram-negative rods, but the source of sepsis was not identified. At the time of resuscitation, a bronchoscopy was performed, which revealed a minimal amount of granulation tissue in the left main bronchus but no evidence of obstruction. Postmortem cross-sectional histologic studies of the anterior pericardial tracheoplasty repair site showed intact respiratory epithelium overlying a well-vascularized submucosa, with hypertrophic mucus-producing tracheal glands. Remnants of pericardium or suture granulomas were not identified histologically (Figs 4A, 4B).
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). The remaining 9 patients are asymptomatic, with no bronchoscopic evidence of airway obstruction or granulation on the pericardial patch (see Table 5).
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) as well as coronal and sagittal multiplanar reconstruction (Fig 7) revealed widely patent luminal areas. All 6 patients had tracheal diameters ranging from 69% to 94% of predicted normal values (see Table 5). The cross-sectional areas of both the normal and reconstructed segments of the trachea were each measured in three different axial images, and the mean value was compared with a nomogram determined by Effman and associates [8] (Fig 8). All patients had normal tracheal growth, although 2 had growth at the lower limits of normal values (but still within 2 standard deviations of the mean) (Fig 9). Complete pulmonary function tests were available in 4 survivors (all >5 years old) and demonstrated normal function in 2 patients and mild to moderate obstruction in 2 patients.
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Comment |
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TopFootnotesAbstractIntroductionPatients and MethodsResultsCommentReferences |
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Tracheoplasty for long-segment congenital stenosis using costal cartilage was initially reported by Kimura and associates [3] in 1982. Several clinical series confirmed that this technique resulted in good early and midterm outcomes, although the frequent need for repeat bronchoscopy to remove granulation tissue remained problematic [3, 10–12]. DeLorimier and associates [12] showed that cartilaginous grafts can be absorbed and replaced by fibrous tissue within 3 months of the repair. A similar observation was reported by Kimura [13] in 1 patient with a good functional result seen 10 years after operation. Unfortunately, objective data documenting growth in the reconstructed trachea with this technique are lacking.
Slide tracheoplasty is a novel and attractive approach for the management of long-segment tracheal stenosis. It was initially proposed by Tsang and colleagues [2] and has been modified by Grillo [1]. In this technique, the trachea is divided transversely through the middle of the stenosis using either a cervical incision or thoracotomy. The two ends are spatulated by a longitudinal slit on the anterior surface of one end and the posterior surface of the other end. The spatulated ends are then advanced over each other and anastomosed together in an extended end to end fashion. Distal ventilation is used for gas exchange, and stents are not used. Reports of this technique have shown that the majority of patients can be extubated in the operating room or soon thereafter in the intensive care unit. The total hospital stay in Grillo's series [1] ranged from 8 to 13 days. In addition to shortened intubation times and hospital stays, potential advantages of slide tracheoplasty when compared with either cartilaginous or pericardial patch tracheal augmentation include the avoidance of graft material, rare formation of granulation tissue, and less frequent requirements for postoperative bronchoscopy. Although all of these advantages are desirable, patient age and size may be a limiting factor. The feasibility of slide tracheoplasty in young infants and the growth potential of the long suture line remain to be determined, although this technique may be ideal for older children. Extremely long tracheal stenoses or those that involve the mainstem bronchi pose potential limitations to slide tracheoplasty repair and may not be adequately treated with this technique. Grillo [1] himself suggested that additional operative procedures might be required for 1 patient in his series who had bronchial stenosis.
In our current series, 12 patients underwent anterior pericardial tracheoplasty, with an early mortality rate of 8% (1 of 12) and an overall survival at 5 years of 84% (10 of 12). Intubation time (range, 6 to 48 days; median, 12 days) and hospital stay (range, 10 to 346 days; median, 17 days) were substantially longer than those in Grillo's report [1]. However, the duration of intubation has decreased substantially as we have gained more experience with this technique. The last 2 patients (11 and 12) were extubated on postoperative days 6 and 8 and were discharged home on postoperative days 10 and 12, respectively.
In contrast to the slide tracheoplasty technique, neither the length nor the location of the stenosis is a technical limitation of anterior pericardial tracheoplasty. Extensive stenosis involving the mid to distal trachea and left mainstem bronchus has been repaired successfully (patient 5) by extending the pericardial patch across the bronchial stenosis. Although this patient died of sepsis of unknown origin 2 years after operation, bronchoscopy revealed no stenosis of either the trachea or the distal left mainstem bronchus at the time of his death.
Excessive granulation formation on the mesenchymal surface of the tracheal substitute and the requirement for frequent postoperative bronchoscopy may be disadvantages of pericardial patch or rib cartilage tracheoplasty as compared with slide tracheoplasty, which is accomplished with only native tracheal tissue. The largest experience with the anterior pericardial patch repair was reported by the group at the Children's Hospital in Chicago [14] and described up to 16 bronchoscopic sessions in patients for debridement of excessive granulation tissue. In our series, however, only 2 patients experienced marked granulation associated with restenosis. Both of these cases occurred at previous tracheostomy sites, and the luminal surface of the pericardial patch was not involved (see Table 4). In 2 patients, minimal granulation developed at the distal end of their pericardial patch repair, without any stenosis, and was treated successfully by bronchoscopic removal (see Table 4). Postoperative bronchoscopy was required a median of only twice per patient (range, two to 12 times) in this series. More important, except for 1 patient (patient 10, who had granulation at a tracheostomy site), none of the late bronchoscopic studies showed substantial granulation formation beyond 6 months postoperatively (range, 6 months to 10 years) (see Table 5). Similar findings were observed in a recent report of cartilage repair by Jaquiss and associates [4]. These results indicate that the avoidance of mesenchymal tissue for airway reconstruction may be less important than suggested by Grillo [1].
The fate of the luminal surface and the speed of reepithelialization of a pericardial patch remain controversial. Postmortem cross-sectional pathologic examination of one specimen from a late death in this series showed that the pericardial patch was not histologically identifiable after 2 years. A complete lining of epithelium and development of normal mucosal and submucosal structures, including mucous glands, vessels, and muscles, were observed throughout the repaired trachea (see Fig 4). Although further research is necessary to elucidate the mechanism of reepithelialization, these histologic results support the late bronchoscopic findings of adequate airway growth and development in these patients.
The most important finding of the current study is that of normal tracheal growth and development (by spiral CT scan assessment) after anterior pericardial tracheoplasty in the 6 long-term survivors who could be studied. Although the 2 most recent repairs (patients 11 and 12) showed growth at the lower limit of normal, no patient had an abnormally small tracheal diameter or cross-sectional area after follow-up extending as long as 11 years.
Spiral CT scan is cost effective compared with magnetic resonance imaging and does not require sedation in small children because the whole study can be done in 10 to 15 seconds. We believe this technique is one of the most efficient methods for following long-term growth of the repaired trachea. Moreover, relation with other vessels and organs can be clearly determined if intravenous contrast is used. Thus, this new technique also may be useful to rule out a pulmonary artery sling or vascular ring preoperatively.
The use of cardiopulmonary bypass for repair of the trachea in children remains controversial. We and others have found it extremely useful to achieve good operative exposure of the trachea [4–7, 14–16]. Others have preferred to avoid bypass [1–3, 11]. Certainly, cardiac surgeons are more familiar with this type of technique for maintaining oxygenation, and general thoracic surgeons are more familiar with using cross-incisional ventilation of the distal airway. In the current series, bypass times were all within an acceptable range, and no complication related to the use of bypass was observed. Although only 1 patient had concomitant repair in this series, cardiopulmonary bypass is an ideal modality of oxygenation for combined repair of long-segment tracheal stenosis and complex congenital heart disease [15, 16].
In summary, anterior pericardial tracheoplasty represents an attractive therapeutic option for infants with long-segment tracheal stenosis and offers good functional results at intermediate to long-term follow-up. Major advantages of this technique include (1) no restrictions for a patient's age or size, (2) no technical limitations as to the length and location of stenosis that can be repaired, and (3) possibility of concomitant repair of other congenital cardiac anomalies using cardiopulmonary bypass. The vast majority of survivors remain asymptomatic without important airway obstruction, and studies suggest normal tracheal growth and development after operation. We believe that anterior pericardial tracheoplasty is the procedure of choice for long-segment congenital tracheal stenosis in infants and children with or without associated congenital cardiac anomalies.
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Footnotes |
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TopFootnotesAbstractIntroductionPatients and MethodsResultsCommentReferences |
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Address reprint requests to Dr Bando, Section of Cardiothoracic Surgery, Indiana University Medical Center, 545 Barnhill Dr, EM 215, Indianapolis, IN 46202.
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TopFootnotesAbstractIntroductionPatients and MethodsResultsCommentReferences |
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