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Pediatric Airway Unit and Division of Pediatric Surgery, Pediatric Institute of the Heart, Doce de Octubre, University Hospital, Madrid, Spain
Received 13 September 2007; received in revised form 16 January 2008; accepted 18 January 2008.
* Corresponding author. Address: C/Vallehermoso, 20.7° Aizda, Madrid 28015, Spain. Tel.: +34 91 4451516; fax: +34 91 3908375. (Email: janton.hdoc{at}salud.madrid.org).
| Abstract |
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Key Words: Tracheobronchomalacia Tracheal stenosis Stent Bronchoscopy Children
| 1. Introduction |
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The purpose of this study is, therefore, to review the results of a single institution experience with endoscopic stent placement in children with benign tracheobronchial obstruction, and with special concern on safety and clinical effectiveness.
| 2. Material and methods |
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Bronchoscopy, either rigid or flexible, was the most important diagnostic tool and was performed in all the patients. A chest radiography was obtained in every case too, and a gastroesophageal reflux (GER) study was performed in most patients (15/21, 71%). Other image diagnostic tests (CT, MRI) were indicated on an individual basis attending to the type of lesion observed at bronchoscopy and to the presence of associated anomalies.
The following data were evaluated: age, type of obstruction, associated malformations, stent properties, technical and clinical success, complications and related reinterventions, outcome and follow-up.
Selection of the type of stent depended on the anatomic site of the lesion, the patient's age, and stent availability when time of presentation. Both metallic balloon-expandable stents and silicone-type devices were used in the series. Stent selection has evolved with time due to the increasing availability of new devices. Our present policy consists of using Palmaz metallic stents for bronchial obstruction (malacia or stenosis), and silicone Dumon devices for tracheal obstructive lesions if the patient is at least 3 months of age.
In infants under 1 year of age stents of 7–10 mm diameter were used in the trachea and devices of 4–6 mm wide were placed in the main bronchi. As a general rule, each stent was oversized approximately 10–20% compared with the adjacent normal diameter. Stents were progressively dilated as the child grew, if metallic, or replaced by a bigger one in case of a silicone stent. In older children larger stents were used. Stent removal was carried out when complications, such as migration or pronounced granulation tissue formation, occurred or when the device was no longer necessary.
Technical success was defined as successful stent placement at the appropriate site in one session. Clinical success was deemed when ventilatory weaning and extubation were achieved, or an improvement in the patient's respiratory status was observed. In the follow-up, a patient who showed no symptoms from a respiratory standpoint or only mild symptoms with infection was considered a good result.
2.1 Stent placement technique
All the stenting procedures were undertaken under general anesthesia in the operating room or in the catheterization laboratory. Prior to stent placement, the type, location, severity, and length of the obstruction were assessed by means of bronchoscopy, conventional chest radiographs, CT scan (with multiplanar reconstruction when available) or MRI.
Metallic balloon-expandable Palmaz stents (Johnson & Johnson Interventional Systems, Warren, NJ) were placed using a Storz rigid pediatric bronchoscope (Karl Storz, Germany; size range, 2.5–6.0) with fluoroscopic monitoring. These stents are available in a wide range of diameters and lengths and its design allows progressive transversal expansion with concomitant stent shortening. The airway prosthesis was mounted, in its non-dilated format, on the outside of an appropriate sized balloon catheter. The whole device was inserted into the rigid bronchoscope and placed in the desired position as determined radiographically. When possible, a 2.7 mm Hopkins 0° lens was placed parallel to the catheter in the bronchoscope channel allowing direct visual control too. The stent was expanded and deployed by inflating the balloon to its full diameter. Then, the catheter was removed by deflating the balloon completely, and a careful bronchoscopic exploration was performed to check the final aspect (Fig. 1 ).
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The more detailed technique of airway stent placement and removal has been extensively described in previous reports [1,4,6].
| 3. Results |
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3.2 Technical and clinical success
Stent placement was technically successful in all but one patient, in whom a complete left lung atelectasis developed after placing two metallic stents in the left main bronchus (Fig. 2
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3.3 Complications
Granulation tissue formation was the most frequent complication (5/5), and in two cases stent migration was detected (in two patients both complications occurred). Stent related respiratory infection has not been a frequent complication, and only in one patient bronchial metallic stents were supposed to be the probable cause of pneumonia (Table 2
). Granulation tissue was more severe in the three patients with metallic stents. In the first case this complication occurred in the trachea and main bronchi and several bronchoscopies were needed in order to remove the inflammatory tissue or to flatten it with balloon dilation. Stent removal seemed extremely risky because the three stents (one tracheal and two bronchial) were completely buried in the airway wall and covered with granulation tissue (Fig. 3
). This patient eventually died due to this complication. In the other two cases metallic tracheal stents were bronchoscopically removed 2 and 5 years after placement. The two patients with silicone-type stents (Dumon and Montgomery T-tube) showed a moderate inflammatory reaction located in one of its ends compelling to removal in both of them.
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3.4 Stent removal
Overall, stents were successfully withdrawn in nine patients (9/21, 42%): four patients with metallic stents and six with silicone devices (one patient had both types). In the group of patients with clinical improvement, stents were removed in seven cases (7/18, 38%): in four due to complications, and in three patients electively because they were no longer necessary. Stents were also removed in two of the three cases in which there was no clinical improvement. Bronchoscopic removal of metallic stents was much more difficult and risky than silicone stent withdrawal. In one case, a tracheal stent was removed with peripheric cardiopulmonary bypass standby although finally it was not needed, and a tracheotomy was necessary to withdraw the metallic devices in another patient because they got stuck in the trachea during bronchoscopic removal. Some degree of self-limited mucosal tracheal bleeding occurred in the two patients in which long-standing metallic stents were removed (after 2 and 5 years).
3.5 Outcome and follow-up
Eight patients (8/21, 38%) in the series died during follow-up, but only in one case (1/21, 4%) it was related to airway stenting. The remaining seven patients had functional airways and died because of other severe congenital or associated anomalies: encephalopathy (two patients), syndromic diseases [2], bilateral lung hypoplasia [1], cardiac malformation [1], and multifactorial chronic neumopathy [1].
Overall, 13 patients (13/21, 62%) are alive with a mean follow-up period of 39 months (range, 1–13.8 years). If we consider only those cases which showed clinical improvement after airway stenting, the ratio is 72% (13/18). Eleven patients (11/18, 61%) are symptom-free or show only mild symptoms with respiratory infections (including the two patients in whom stents were placed because of postoperative tracheal stenosis), and the remaining two need some type of respiratory therapy, either bronchodilators or nocturnal CPAP, because of persistent symptoms (one with bronchial stents in place and in the other the Dynamic stent was removed). Eight children (8/21, 38%) have still their airways stented, six with metallic devices and two with silicone-type stents, the longest for more than 6 years. These patients undergo close surveillance in our Unit and bronchoscopy is performed at least once a year.
| 4. Discussion |
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Aortopexy and surgical relief of external compression are still the mainstay of treatment in severe TBM. Excellent results have been reported with aortopexy, especially in cases of tracheomalacia associated to congenital TEF [12,13,16,17], but with malacia involving the bronchi it seems to be not so effective. Filler et al. reported an 80% failure rate in cases of TBM and isolated bronchomalacia [16]. Pexy operations can be done with other mediastinal vessels, such as the pulmonary arteries, or even with the trachea and main bronchi, but experience is short and information scarce [18,19].
Intraluminal stent therapy aims to support the airway till it regains its structural integrity with growth [20]. In our experience, stenting has not substituted arterio/aortopexy as a first line treatment in patients with tracheomalacia. In fact, we indicate stent placement when aortopexy has failed or it is not recommended because of previous cardiac surgery with mid-sternal access. Bronchomalacia is another of our current indications for stenting. As it has been stated before, aortopexy is usually unsuccessful when trying to correct this condition, and there is very little experience with bronchopexy. In some instances, patients with tracheostomies were referred from other institutions to our Unit. The tracheostomy tube acts as an internal splint in cases of tracheal malacia and has also a place in its management. In four patients of our series the tracheostomy tubes were unable to support effectively the malacic trachea and respiratory distress developed. They were replaced by Montgomery T-tubes in three cases with immediate clinical improvement. The remaining patient refused tracheostomy and two silicone stents were placed in her airway.
Stenosis after pediatric airway reconstruction is another well established indication for airway stenting [2]. We have placed Dumon tracheal stents in two children with recurrent stenosis: one in the postoperative period, and the other one after several bronchoscopic dilations. Stents were removed during follow-up (one month and 3 years after insertion respectively) and both patients are asymptomatic from a respiratory standpoint.
Pediatric airway stenting is a relatively recent technique and follows the wide experience acquired in the field of adult pneumology. Montgomery first described the use of silicone T-tubes in the adult trachea in 1965 [21]. Since then, stent placement has become a common endoscopic procedure for benign and malignant airway obstructive diseases in adults. An ideal airway stent should have the following properties: (1) easy placement and removal; (2) effective airway expansion; (3) good tolerance with minimal granulation tissue formation, (4) no interference with the respiratory mucociliary function; (5) good adherence to the tracheobronchial wall; and (6) a wide range of sizes so that they can be used in neonates as well as in adults [22]. Unfortunately, no single stent shows all these properties at the present time.
Silicone-type stents are the most frequently used in adult patients. Placement and removal is not difficult and they are usually well tolerated. On the other hand, they can become obstructed by inspissated secretions and stent migration may happen [23]. Their large size and rather unfavourable wall to lumen ratio makes them probably less adaptable in a smaller infant airway [20]. Nevertheless, we have successfully placed the smallest Dumon stent (6/7 mm diameter, internal/external) in a 4 month-old infant with a tracheal postoperative stricture. A 6 mm diameter Polyflex device was placed in the trachea of another patient of the same age who had tracheomalacia secondary to congenital TEF, and in whom a previous aortopexy had failed. This stent migrated and was changed to a Dumon type (Fig. 4 ). Tracheobronchial Y-shape Dynamic stents were placed in two patients, aged 8 and 12 years, with tracheal and bronchial malacia (Fig. 5 ). Tolerance was good in both cases although one child died due to unrelated causes during follow-up.
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Stent availability for use on children has increased with time. In 1993, when airway stenting was started in our Unit, metallic Palmaz stents were the most suitable because of their small size and straight-forward insertion [4]. New stents are currently available and others have adapted their size and properties to the airway requirements of small children. At the present time, we use Palmaz metallic stents for bronchial obstruction (malacia or stenosis), and silicone Dumon devices for tracheal obstructive lesions if the patient is at least 3 months of age. The smallest Dumon stent available (6/7 mm diameter) needs a 3.5 pediatric Storz rigid bronchoscope to be inserted and it is unsuitable for the airway of a neonate. If a tracheal stent is necessary in an infant under 3 months of age, either we keep him/her on ventilatory airway support until a silicone device can be placed or we insert a Palmaz metallic stent of the appropriate size. When TBM is present, we prefer to place a Y-shape Dynamic stent if it matches the size of the patient's airway. This type of tracheobronchial stent is not available for the airway of small children and infants, so two or three individual stents should be used in these cases.
Unfortunately, none of the currently available endoluminal airway stents is free of complications. Nitinol stents exhibit shape memory and are softer and more elastic than Palmaz stents. Although they are deemed to have high tissue biocompatibility [20], two of the five patients with nitinol devices reported by Nicolai et al. [6] showed pronounced granulation tissue. This stent cannot be dilated so it must be replaced by another one as the airway grows. Even though it is a more appealing alternative than other metallic devices, the size of the smallest nitinol stent available (6 mm diameter and 2 cm long) makes it less suitable for bronchial placement in a neonate. The recently available polyurethane covered nitinol stent is easier to remove and seems to cause less granulation tissue [3].
New airway stents are undergoing assessment. Endoluminal devices with improved biocompatibility or even completely resorbable ones [25] may soon replace the currently available stents.
The present study has the following limitations: It is indeed a retrospective review, and the number of patients is small. Long-term prospective multicentre studies are the best option to establish the definite therapeutic value of airway stenting.
We conclude that intraluminal airway stents are a satisfactory therapeutic option in the management of severe tracheobronchial obstruction. They can be life-saving in extremely sick children in which other ways of treatment have failed or are not indicated. Selection of the type of stent should be tailored to each particular case depending on the patient's age, clinical situation, and anatomic location of the lesion. In neonates and small infants metallic stents are the most adequate although removal may involve difficulty and risks. A close long-term survey is recommended in every case, including periodical bronchoscopic explorations and pulmonary function tests.
| Appendix A |
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Dr E. Rendina (Rome, Italy): A very high success rate. However, metallic stents have a very bad reputation especially for benign disease and especially in growing individuals. Now, Im impressed by the percentage of patients in whom you used them. Can you comment, especially on this conclusion that you make that they are the most adequate for small boys. Can you comment on that and say your reasons for this choice?
Dr Anton-Pacheco: Well, I agree. But metallic balloon-expandable Palmaz stent has been the only available for several years. So we were very short of stents to use in very small children. So we had to use these because we cannot put a silicone stent under 3 months of age, because you need a rigid bronchoscope, 3.5, and you cant put that bronchoscope in the airway of such a small baby.
Probably we are going to switch to the nitinol-covered stent instead of the Palmaz expandable stent in the next months. But until now this is our experience. And although there are complications with metallic stents we know, and we have suffered them, I think it's a life-saving procedure in some cases.
Dr D. Wood (Seattle, Washington, USA): I just want to take that same question one step further, because I have the same concern about the use of uncovered metallic stents. In the cases where you removed the stents, what was the outcome of the airway in those cases? I know that often those airways are very raw and then suffer post-stent removal stenosis. My other question is what is the long-term outcome in just the metal stents that are still in place?
Im anticipating bad long-term outcomes in both the bare metal stents that are left in place, because of lack of growth and how they create an inflammatory airway response, and in the ones that are removed because of the post-removal inflammation. Certainly the silicone stents do come in a 6 mm, that is the smallest, and may not be small enough for the infants. But I think that we have to make every effort to avoid uncovered stents because of all of the problems that youve discovered. What are your outcomes?
Dr Anton-Pacheco: We removed the metallic stents in four patients. In two cases it was very soon after placement because there was no clinical improvement or because there was a technical complication. So those stents were easily removed in one case, and a tracheotomy was required in the other case because the stent got stuck in the trachea.
And the other two patients with long-standing Palmaz metallic stents were removed by means of rigid bronchoscopy with moderate self-limited mucosal bleeding and no more incidence. Concerning the growth in the boy and the girl, they are doing okay, there has been no other complications after stent removal with these two long-term stent cases.
And the other question was related to growth. Well, we dont know what will be the long-term follow-up, probably nobody knows. We keep dilating them. And if they produce problems, they generate problems, we try to remove them. But we keep dilating the stents. And in general, bronchial metallic stents are better tolerated, that's our impression, than tracheal stents. We keep dilating.
Dr C. Sebening (Heidelberg, Germany): Im walking in the mouth of the lion here because Im a congenital heart surgeon. But Im very interested in this type of pathology and quite a number of patients have been addressed to our unit with either vascular compression syndromes or secondary malacia due to esophagus atresia type IIIb of Vogt.
So do you follow in your center more complex cases that have been surgically corrected in this context, with the use of extracorporeal circulation? Or do you see these kinds of children before surgery? Im asking, and with much respect for all the progress that has been achieved in your field: before putting a stent into a child, having a vascular compression syndrome or secondary malacia, do you evaluate the patient for treatment options alone, or in collaboration with a surgical team?
Dr Anton-Pacheco: Yes, some of these cases are due to vascular compression. And our first line of treatment is surgery, I mean aortopexy or arteriopexy or something like that, that's the first-line treatment. And only those cases that are a failure, surgical failure, or that are bronchomalacic or something like, they go into the stent group. But we first do a conservative trial, then a surgical treatment, and stenting is our third-line treatment.
Dr Sebening: So you have children that are operated by your heart surgeon in your town?
Dr Anton-Pacheco: By my heart surgeon?
Dr Sebening: Yes, I saw the name on there.
Dr Anton-Pacheco: Well, aortopexy is usually performed by the pediatric surgical service. So aortopexy is performed by us, I am a pediatric surgeon. And if it doesnt work, we place the stent, too.
Dr Sebening: The point that I would like to make is from our observation that extracorporeal circulation may be a very useful tool for long intrathoracic tracheal pathology.
Dr Anton-Pacheco: For how much time? ECMO you mean?
Dr Sebening: No, I mean the heart–lung machine. Im just asking because Im interested in this type of pathology and some cases have collected in our department.
Dr Anton-Pacheco: We have no experience. I cant tell you.
| Footnotes |
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Presented at the 21st Annual Meeting of the European Association for Cardio-thoracic Surgery, Geneva, Switzerland, September 16–19, 2007. | References |
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