Ann Thorac Surg 2004;78:1446-1448
© 2004 The Society of Thoracic Surgeons
Case report
Extratracheal Biodegradable Splint to Treat Life-Threatening Tracheomalacia
Philippe Bugmann, MDa,*,
Peter C. Rimensberger, MDb,
Afksendyios Kalangos, MDa,
Constance Barazzone, MDb,
Maurice Beghetti, MDb,
Florian J. W. Lang, MDc
a Clinic of Pediatric Surgery, Hôpital des Enfants, Geneva, Switzerland
b Department of Pediatrics, Hôpital des Enfants, Geneva, Switzerland
c Department of ENT and Head and Neck Surgery, CHUV, Lausanne, Switzerland
Accepted for publication July 10, 2003.
* Address reprint requests to Dr Bugmann, Hôpital des Enfants, 6 Rue Willy-Donzé, 1211 Geneva 14, Switzerland
philippe.bugmann{at}hcuge.ch
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Abstract
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A 9-month-old girl presented with life-threatening acute respiratory failure 1 week after the surgical correction of a double aortic arch, which was due to a severe bulging of the pars membranacea into the lumen of the trachea that produced a complete obstruction of the lower trachea. Under cardiopulmonary bypass, a Y-shaped posterior biodegradable splint was placed behind the trachea and sutured to the posterior trachea, and a simultaneous right aortic arch aortopexy was performed. Thereafter, the child recovered normal respiratory function. Follow-up bronchoscopy showed a posterior dip at the splint level and an asymptomatic persistent posterior compression of the right main bronchus.
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Introduction
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Symptomatic tracheal compression caused by vascular rings, such as double aortic arch, requires surgical release of the vascular compression. However, as many as 70% of the patients may have persistent symptoms after surgery, ranging from very mild residual dyspnea during exercise to severe persistent respiratory failure [1, 2]. Adverse surgical outcomes with immediate or progressive development of life-threatening respiratory distress have also been reported, particularly in patients with double aortic arch [3]. In this rare instance, when airway damages are severe enough to warrant reintervention, choices between the surgical options are difficult. The different options are tracheostomy, aortopexy, tracheal resection and reconstruction, external splinting, or internal stenting, or a combination of these procedures. We present a case with unfavorable evolution after surgical correction of a right dominant double aortic arch, which was operated on using an external posterior biodegradable splint combined with an anterolateral aortopexy to restore a patent tracheal lumen.
A 9-month-old girl presented with a life-threatening acute hypoxic respiratory failure 1 week after surgery for a double aortic arch. Bronchoscopy revealed total obstruction of the inferior trachea due to persisting tracheo-malacia and a bulging of the pars membranacea into the lumen, requiring endotracheal intubation. Enormous difficulties in maintaining adequate ventilation were encountered: a critical positioning of the tip of the endotracheal tube exactly at the carina was necessary to obtain satisfactory gas exchanges, thus using the endotracheal tube as an internal stent. It was decided to wait 3 weeks for a possible spontaneous improvement, all the more so as a test for respiratory syncitial virus antigen proved positive. However, this conservative treatment failed. A rigid bronchoscopy confirmed the persistence of a severe lower tracheal compression associated with a bulging flaccid pars membranacea (Fig 1, A and B). Thus, surgical treatment combining an aortopexy and an external tracheal splint was planned.
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Fig 1. (A) Aspect of the lower trachea before surgery showing complete obstruction with bulging of the tracheal membrane into the lumen and external anterior and posterior compression. (B) The rigid bronchoscope is pushed through the obstructed tracheal segment to disclose the carina. (C) Operative endotracheal view of the flattened tracheal membrane after the suture of the posterior polydioxanon splint. The distal tracheal lumen is still occluded. (D) Aspect of the same segment during right aortic arch aortopexy. The carina is now visible. (E) On follow-up bronchoscopy at 6 months, the tracheal membrane shows a dip on the right side above the carina; the left main bronchus is wide open. (F) Persistent posterior compression of the right main bronchus at 6 months.
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The child was reoperated on through a median sternotomy. Cardiopulmonary bypass was instituted by cannulating the ascending aorta and right atrium, allowing for safe tracheal exploration and repair. The inner aspect of the trachea was continuously monitored during the procedure using flexible bronchoscopy. Dissection to free the left anterior aortic arch did not improve the tracheal compression. Different tractions on the two aortic arches were ineffective as well; the tracheal lumen remained obstructed by the bulging pars membranacea. Thus, the posterior aspect of the trachea was approached from the left side only, to avoid devascularization of the tracheal segment and preserve the aortotracheal adhesions for an aortopexy. A custom-made posterior biodegradable Y-shaped splint was trimmed out of a 0.25-mm thick polydioxanone plate (ZX5 PDS plate, 0.25 x 40 x 50 mm; Johnson & Johnson, Spreitenbach, Switzerland) to match the shape of the posterior wall of the lower trachea, extending 1 cm behind the two main bronchi. The tracheal membrane was sutured to the splint by 8 U-shaped polyglactin 6-0 sutures (Vicryl V134; Ethicon, Johnson & Johnson, Spretienbach, Switzerland) passed through holes previously drilled every 5 mm in the polydioxanone plate (Fig 2). Care was taken to avoid the left recurrent laryngeal nerve. An endoluminal tracheal view showed a flattening of the pars membraneacea, but the lumen was still occluded owing to the anteroposterior compression (Fig 1, C). A right lateral translation of the right aortic arch definitively opened the lumen of the carina. Therefore, a Spitz aortopexy was performed with a 3 cm x 1 cm knitted polyester patch sutured on the right aortic arch and two nonabsorbable 4-0 stitches passed through the patch and adjacent right hemisternum. These two stitches were fastened during a compression of the sternum after closure. Upon release of the sternum, the lumen of the carina opened (Fig 1, D) and expired CO2 levels immediately returned to normal (no expired CO2 could be monitored before the tightening of the aortic suspension stitches). Extubation was performed successfully 3 days after surgery. The child was discharged 3 weeks later with normal bilateral lung ventilation but a persistent inspiratory stridor that disappeared within 1 month.
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Fig 2. (Left) Anatomic situation after ligature and division of the left aortic arch distal to the left subclavian artery. (Right) Drawing of the operative procedure: the left aortic arch is not depicted for clarity; the Y-shaped polydioxanone splint is sutured to the posterior wall of the trachea through predrilled holes; a Dacron patch is sutured to the lateral aspect of the right aortic arch below the right common carotid artery and is pulled laterally by two heavy sutures that will be passed through the right part of the sternum.
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A follow-up bronchoscopy was performed 6 months later to check the aspect of the lower trachea after weakening of the biodegradable splint. The posterior aspect of the lower trachea showed a shallow dip on the right side of the tracheal membrane (Fig 1, E), probably related to the effect of the posterior splint. The lumen of the left main bronchus was normal, but an 80% posterior compression of the right main bronchus persisted (Fig 1, F). At 1-year follow-up, the child remains asymptomatic even at exertion.
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Comment
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The need for secondary intervention after surgical treatment of symptomatic double aortic arch has been reported in several follow-up studies. Horvath and associates [4] reported that 5 of 26 children presenting with a double aortic arch and tracheal compression required reoperation. An undivided ligamentum arteriosum or adhesive bands was found. Two other patients died of persistent tracheomalacia that "could have been treated by more aggressive therapy," according to the authors [4]. Aortic suspension procedures are considered the first choice in persistent vascular extrinsic tracheobronchial compression [5]. Tracheal resection and reconstruction has also been used [3], but is considered as major surgery with the risk of complications related to tracheal anastomoses, such as fibrotic stenosis, ingrowth of granulation tissue, or leakage. Endotracheal stenting is an attractive solution, but the long-term safety and tolerance is still a matter of uncertainty [6]. External airway splinting with ring-reinforced polytetrafluoroethylene prostheses has been used by Hagl and colleagues [7] with excellent results. This procedure leaves foreign material around the trachea, however, could possibly restrict the long-term growth of the airways, and exposes adjacent structure to the risk of erosion. The surgical strategy that was adopted here avoided any internal stenting of the child's airways, keeping this option as a last resort. The 0.25-mm biodegradable polydioxanone plate was chosen for its relative stiffness and ease to fashion. It successfully flattened the tracheobronchial membrane and did not damage any surrounding organ. The favorable clinical result remained stable for 1 year, and the local aspect at 6 months suggests that fibrotic tissue is replacing the vanishing plate to maintain the flattening effect on the posterior trachea. The only partial release of the compression of the right main bronchus is probably related to the insufficient space available between the two aortic arches at their bifurcation.
This case shows that a biodegradable plate sutured posteriorly to the trachea has many advantages: it may efficiently stiffen the flaccid pars membranacea, seems not to restrict the normal growth potential of the tracheobronchial tree (an important advantage for young children), and it does not leave any long-term foreign material, thereby reducing the risk of infection or erosion of adjacent structures. However, the long-term efficiency of such biodegradable plates will require further evaluation.
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Acknowledgments
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We thank Prof Beat Friedli for his kind help and for the considerable improvements he brought to the manuscript.
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References
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- ten Berge M, van der Laag J, van der Ent CK, Beek FJ. Clinical, radiological and functional follow-up after surgical decompression of double aortic arch. Pediatr Radiol. 2002;32:561–566[Medline]
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- Horvath P, Hucin B, Hruda J, et al. Intermediate to late results of surgical relief of vascular tracheobronchial compression. Eur J Cardiothorac Surg. 1992;6:366–371[Abstract]
- Corbally MT, Spitz L, Kiely E, Brereton RJ, Drake DP. Aortopexy for tracheomalacia in oesophageal anomalies. Eur J Pediatr Surg. 1993;3:264–266[Medline]
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Abstract
Introduction
