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Ann Thorac Surg 2003;76:175-179
© 2003 The Society of Thoracic Surgeons

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Original article: general thoracic

Elective extracorporeal support for complex tracheal reconstruction in neonates

^a Departments of Cardiothoracic Surgery and Extracorporeal Support Services, Brenner Children’s Hospital, Wake Forest University School of Medicine, Winston-Salem, North Carolina, USA

^* Address reprint requests to Dr Hines, Department of Cardiothoracic Surgery, Brenner Children’s Hospital, Medical Center Blvd, Winston-Salem, NC 27157 USA.
e-mail: mhines{at}wfubmc.edu

Presented at the Forty-eighth Annual Meeting of the Southern Thoracic Surgical Association, San Antonio, TX, Nov 8–10, 2001.

	Abstract

Top Abstract Introduction Material and methods Results Comment Discussion References

 
BACKGROUND: Congenital obstructive anomalies of the trachea present unique challenges in reconstruction and perioperative airway management. Complications include anastomotic breakdown, leak and granulation formation related to the complexity of the repair, and difficulties with perioperative airway management. We describe our technique of elective intraoperative and postoperative extracorporeal support to improve surgical exposure and postoperative healing.

METHODS: We have performed complex tracheal reconstructions in 4 newborns (2.2 to 4.3 kg) for long segment tracheal stenosis and complete tracheal rings, diagnosed with bronchoscopy and computerized tomography. Three of the 4 infants had other significant anomalies including complex congenital heart disease, hydrocephalus, encephalomalacia, left lung agenesis, facial anomalies, vertebral anomalies, and hand and hip anomalies. The repairs were performed through a median sternotomy using an extracorporeal membrane oxygenation circuit for support. Venoarterial support was used for the sliding tracheoplasty reconstruction. Extracorporeal membrane oxygenation was converted to venovenous for postoperative "airway rest." After diuresis, the lungs were reexpanded and the 4 patients were ventilated and removed from extracorporeal membrane oxygenation at 4, 5, 8, and 9 days postoperatively. Bronchoscopy was performed to evaluate the airway.

RESULTS: All patients had excellent healing of the trachea without granulation tissue. There were no complications of extracorporeal membrane oxygenation support or bleeding issues. All 4 patients survived the surgery and immediate postoperative period with 2 late deaths. The child with congenital heart disease expired after 8 weeks after having hepatorenal failure develop. The child with Goldenhar’s syndrome and a single left lung died after 5 months in the hospital. The other 2 patients survived. Two of the infants required late tracheostomy for facial and laryngeal anomalies.

CONCLUSIONS: Extracorporeal membrane oxygenation provides an excellent environment for complex tracheal reconstruction and promotes postoperative healing by minimizing trauma to the reconstructed airway.

	Introduction

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Congenital tracheal stenosis is a very rare anomaly that usually presents very early in life with severe respiratory distress. In lesions too long to consider simple resection and end-to-end anastomosis, surgical repair provides considerable challenges in reconstruction because of patient size, tissue strength and availability, and intraoperative and postoperative airway management. Several techniques have evolved over the last 2 decades including esophageal tracheoplasty [1], pericardial patch tracheoplasty [2, 3], rib cartilage graft tracheoplasty [4, 5], and the slide tracheoplasty [6–11]. Complications have been reported with all of the techniques and have included separation, restenosis, mediastinitis, and granulation formation with hemorrhage or obstruction [4, 5, 12–14]. Controversy remains as to which technique is ideal, including debate over the need for cardiopulmonary bypass [2, 3, 6, 7]. It has been suggested [2] that many of the complications may not be related to surgical technique, but rather exacerbated by airway manipulation and ventilation through the newly reconstructed trachea, creating irritation, inflammation, suture line stress, and direct trauma. Although this problem can be avoided with early extubation, this is rarely possible in neonates with complex reconstructions. We opted to use venoarterial extracorporeal membrane oxygenation (ECMO) support to allow precise surgical reconstruction with a tubeless tracheal field, with the continued use of venovenous (VV) ECMO postoperatively for airway rest. By providing complete gas exchange, VV ECMO minimizes manipulation of the airway by suction, ventilation, and reintubation, potentially improving conditions for tracheal healing. We describe our technique and strategy, using elective intraoperative and postoperative extracorporeal support to manage long segment tracheal stenosis, and our initial results in 4 neonates.

	Material and methods

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Patient population
Four neonates presented to our institution with severe respiratory distress requiring early intubation. All 4 infants were maintained on mechanical ventilation and required heavy sedation or paralysis, or both. Early rigid bronchoscopy was performed documenting the presence of long segment tracheal stenosis with varying amounts of complete rings and funnel type stenosis measuring no greater than 2 mm. Because of the tenuous airways, all the children were kept sedated to maintain endotracheal intubation before repair. All infants had multisystem evaluations including genetic workup, echocardiography, cranial ultrasound, and either computerized tomography or magnetic resonance imaging of the chest including detail of the airways. All 4 patients were found to have adequate airways at the carina and beyond, except the child who had Goldenhar’s syndrome and an absent left lung. In this child, the right lung appeared to be relatively enlarged with appropriate expansion of the right main stem bronchus beyond the stenosis at the level where the carina should have been. When it was determined that the patients were candidates for repair, a date was selected based on patient stability, associated anomalies, size, and age. Patient profiles along with airway defects and associated anomalies are outlined in Table 1.

When venoarterial support was established, the endotracheal tube was carefully removed to just below the vocal cords. A sliding or advancement tracheoplasty was then performed in all 4 patients. Before completing the suture line, a new endotracheal tube (slightly larger than the previous tube) was inserted and guided carefully across and beyond the extent of the repair to act as a stent. The lungs were gently ventilated and reexpanded, and the ECMO system was converted to VV support by connecting the inflow and drainage tubing to the appropriate ends of the double lumen cannula in the right internal jugular vein. All the patients’ hemodynamics remained stable on only low dose dopamine (2 to 5 µg/kg/min). The heparin was not reversed, but the activated clotting time (ACT) was allowed to drift down to a range of 160 to 180 s. A pericardial flap was sutured in place over the repair between the trachea and great vessels. The mediastinum was drained, and the sternum and wound were closed. The patients were maintained on paralytics and narcotics and were transferred to the pediatric intensive care unit on VV ECMO support.

Postoperative strategy
Patients were maintained on VV extracorporeal support with ventilator settings designed to minimize airway trauma, making no attempt to achieve any native gas exchange. Airway rest was maintained with ventilatory rates of 6 to 8 per minute, with low peak pressures (eg, 20 to 24 cm H₂O), and elevated end expiratory pressures (10 to 14 cm H₂O). This strategy was used for at least the first 3 days to maintain some lung expansion without achieving any functional tidal volume. Gentle suctioning of the endotracheal tube was performed as needed to keep the tube clear of secretions. Diuresis was initiated with a continuous infusion of furosemide. When the chest radiograph demonstrated clearing of the radiopacity of the lung fields frequently seen after exposure to the ECMO circuit, and after lung compliance was noted to improve, ventilation was initiated with peak inspiratory pressures of about 26 to 30 cm H₂O. When compliance had improved to allow achievement of tidal volumes (approximately 8 to 10 mL/kg) with the pressures previously mentioned, then the rate was increased. The ECMO was discontinued for a trial period. If adequate gas exchange was maintained for several hours, the ECMO cannula was removed. Plans were made to perform rigid bronchoscopy on the next elective date to evaluate the airway before working toward extubation.

	Results

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All 4 neonates survived the surgery without intraoperative complication and were removed from ECMO on postoperative days 4, 5, 8, and 9 (Table 2). There were no bleeding complications or tamponade despite sternal closure with ongoing heparinization. There were no neck infections, subcutaneous or mediastinal air, suture line leaks, or breakdown. The only complication possibly attributable to the intraoperative venoarterial ECMO run was transient acute tubular necrosis in the infant with a single kidney and congenital heart disease. Bronchoscopic evaluation revealed excellent healing of the repair with only trivial granulation seen in 1 of the 4 patients. One infant had a weak right vocal cord, presumably from traction of the recurrent nerve during mobilization of the trachea, but this was transient and recovery was revealed by subsequent examination.

The second child, the premature quadruplet with congenital heart disease, seizure disorder, and a single kidney, recovered from perioperative renal dysfunction and maintained adequate hemodynamics despite an abnormal cardiac anatomy. However, during the next several weeks this child experienced multiple episodes of sepsis, subsequently having hepatorenal failure develop, and after 8 weeks without significant progress,ventilatory support was withdrawn at the request of the parents. However, during this time there were never any issues of airway healing, although the patient’s general condition precluded withdrawal of ventilatory support or extubation.

Initially the third infant did well after her short 4-day course of ECMO, and after bronchoscopic evaluation demonstrated excellent healing, the infant was extubated. This patient was progressing nicely until 4 days later when reintubation was required for rapidly progressive respiratory failure and when adult respiratory distress syndrome had developed. The patient was placed on VV ECMO for adult respiratory distress syndrome and respiratory failure, but then had positive blood cultures with Staphylococcus aureus and toxic shock syndrome with full-blown desquamation. Later staphylococcal and enterococcal meningitis also developed. The sternotomy wound and neck remained free of infection. Poor perfusion required conversion to venoarterial ECMO support. After 20 days of support, the patient’s lungs and hemodynamics recovered. The patient was removed from extracorporeal support again. The meningitis improved, but left the patient with communicating hydrocephalus and seizures, requiring placement of a ventriculoperitoneal shunt. After this prolonged intubation, repeat bronchoscopy demonstrated a new left vocal cord paralysis and tracheomalacia. The patient then underwent tracheostomy, was weaned to tracheal collar, and discharged to foster care.

The fourth infant was successfully extubated after bronchoscopy, but before planned discharge the patient had a respiratory syncytial virus infection develop and was returned to the pediatric intensive care and reintubated. Over the next 5 months the patient had numerous return trips to the intermediate and intensive care units caused by an inability to handle his secretions. Although previously placed with a gastrostomy tube and Nissen fundoplication, the combination of micrognathia, a cleft soft palate, and only having a single lung continued to create upper airway problems, despite repeated bronchoscopy to clear mucous plugging that demonstrated adequate airway size and structural patency. The child expired 5 months later.

	Comment

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Numerous serious complications have been reported after complex tracheal reconstruction including tracheal leak or breakdown, infection and deep neck abscesses, airway obstruction and bleeding from granulation tissue, and restenosis [4, 5, 12–14]. Extended paralysis and mechanical ventilation [2, 3] prevent trauma from motion, but they do not avoid positive pressure ventilation and manipulation of the airway to maintain pulmonary toilet and adequate gas exchange. Intraoperative extracorporeal support provides not only an intraoperative "tubeless" environment for precise repair, but can be continued postoperatively with VV ECMO to minimize trauma to the airway. Venovenous ECMO is commonly used for severe lung disease to allow "lung rest," by providing complete gas exchange in an extracorporeal fashion, allowing reduction of the hazardous ventilatory pressures and high concentrations of oxygen. Similarly, VV ECMO provides complete gas exchange for the first several days after tracheal reconstruction to minimize use, manipulation, and trauma to the healing airway. The need for emergency maneuvers such as aggressive suctioning and urgent reintubation are eliminated because the infant is no longer dependent on the ventilator or airway for gas exchange. Although previously reported for extended postoperative support, it involved the use of venoarterial ECMO [17]. Venovenous ECMO for continued support avoids carotid ligation and potential arterial emboli, preserves pulsatile flow and normal hemodynamics, and maintains pulmonary blood flow during the resting period. In addition, the morbidity of VV ECMO is considerably less, and usually complications while on support can frequently be attributed to the patient’s underlying disease or shock before initiation of support. Because placement on support is completely elective in nature, these risks are minimized. With the use of antifibrinolytics, anticoagulation can be maintained and can still allow closure of the sternum primarily with low risk of pericardial fluid collection, tamponade, or extensive bleeding (none in our series). We believe this period of airway rest facilitates tracheal healing, reducing the risk of leak, breakdown, and granulation formation, as demonstrated by our postoperative bronchoscopy with excellent granulation free healing in all 4 patients.

Although late mortality occurred in 2 patients, these were attributed to the patients’ numerous comorbidities and other congenital defects. Two patients eventually required tracheostomy, but these were not indicated for failure of the reconstruction, but for other congenital upper airway anomalies in 1 child, and for tracheomalacia after an episode of sepsis and prolonged intubation with the subsequent illness after successful extubation in the second child. In fact, all four reconstructed airways healed without any tissue breakdown or granulation development, which was the goal of the protective strategy of elective extracorporeal support. We believe this technique offers many advantages in the management of complex tracheal anomalies in the newborn.

	Discussion

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DR DARRYL S. WEIMAN (Memphis, TN): On the adult side, Dr Grillo has taught us that we should use absorbable type sutures to do this reconstruction. What do you use?

DR HINES: I use an absorbable PDS as a running suture.

DR ROBERT J. CERFOLIO (Birmingham, AL): I enjoyed your presentation. A couple of questions for you. We have not had the experience you have had. We have only done this in 2 patients, but we have used a much more simple approach. Even though they had long stenoses and were less than 2 weeks old, we used a cervical collar incision, avoided sternotomy, avoided bypass, and avoided extracorpeal membrane oxygenation (ECMO). We use intermittent periods of apnea and place a very small catheter that is connected to a jet ventilator. The catheter can be placed through the pediatric endotracheal tube. As you cut the trachea you pull back the tube into the proximal trachea, and then you can slide the catheter through the anastomosis into the distal trachea. You can actually jet ventilate distally while you are sewing if needed, but in general, short periods of apnea are used and the anastomsis can be done in several minutes without hypoxia. It seems that in these little newborns you are able to get all the way down to the carina through the neck (unlike adults), and you can easily get up to the hyoid bone (as in the adults). This way, described by Grillo and Goldstraw, allows avoidance of some of these suprahyoid release maneuvers that you have used. These can have significant morbidity, especially in adults. This technique seems less complicated, avoiding risk of infection to the sternum and cardiopulmonary bypass and extracorporeal membrane oxygenation. Do you have any experience with this alternative technique?

My second question is why use a lateral incision? Do you really need it? We know the blood supply to the trachea comes in posterior (laterally in the normal trachea). I agree these kids may have abnormal blood supply and I agree that, in general, kids’ tracheas can get away with more dissection with less problems of ischemia, but why not just do the sliding tracheoplasty that has been recommended by Grillo and Goldstraw using a vertical incision? This seems to work so well and it avoids the potential of injury to the blood supply, which is so important for healing without sequela?

DR HINES: Thank you for your questions. Regarding the first question, we have not tried to do this through a neck incision. All these children had either a very long segment or extensive stenosis down to the carina, and we had decided from the magnetic resonance image and computed tomography scans that exposure would be much better. I would argue that the short periods of apnea are not as good as a 20-minute period of apnea, and you can actually do a much better job without the tube and without trying to intubate these children distally, particularly the child with a single lung. We also found much better exposure with this, and with the venovenous ECMO, it provides a very good environment.

As to the second question regarding the lateral incisions, I think we all know, and it is well documented that the blood supply to the normal trachea runs from multiple sources up the lateral wall. What we found in these children is that these are not normal tracheas and there are no vessels along the lateral wall, and when you transect it, what you see is a very thick mucosa, and so this is to me almost like a stomach. Most of the blood supply runs up and down the mucosa, and I think it is very appropriate to do a lateral incision, and then the trachea seems to heal fine.

DR CONSTANTINE MAVROUDIS (Chicago, IL): This is a nice presentation on a very challenging set of 4 patients. You present a 50% mortality, but I do not think that this is the main issue. The issue is whether ECMO, postoperative elective extracorporeal membrane oxygenation, as you call it, helps to heal these tracheal anastomoses. In other words does extracorporeal support decrease the amount of granulation tissue, which has been the bane of taking care of these patients, as you know?

I wonder if you have done any patients the other way, without ECMO? I think these numbers are small and you are going to have a hard time proving that the healing rate is better this way than the more accepted method without extracorporeal support. If you experience some without the use of extracorporeal support, would you give us some idea on why you have come to this rather avant-garde therapy? I think most of us would not do what you are doing, but maybe we should be as long as you can convince us.

Thank you very much.

DR HINES: My experience really was in my training at Boston Children’s Hospital in seeing either difficult airways that we had there and problems with granulation or other children referred in with problems with this, and so when I moved back to North Carolina, I decided I wanted to approach it a little bit different. My other bias was I also did an ECMO fellowship and I was the ECMO director, so unlike a lot of cardiac surgeons, I was very familiar and comfortable with it and had decided when we got the first child that this would be a good approach.

We have done a few other children that were older children, non-neonates, with airway problems, and at the end of the repair decided that the repair was not that complex and did not leave them on ECMO and actually extubated them in the operating room. But in these complex repairs, I just felt very comfortable with this, and it provides a good environment. Also venovenous ECMO, which many ECMO centers are not comfortable with yet, turns out to be very, very simple and safe with very low morbidity.

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