Ann Thorac Surg 2001;72:943-944
© 2001 The Society of Thoracic Surgeons
Case report
Dental bar tracheopexy in a neonate
Pedro A. Catarino, FRCSa,
Satoshi Saito, MDa,
Stephen Westaby, FRCSa
a Department of Cardiac Surgery, Oxford Heart Centre, John Radcliffe Hospital, Oxford, United Kingdom
Accepted for publication September 22, 2000.
Address reprint requests to Dr Westaby, Department of Cardiac Surgery, Oxford Heart Centre, John Radcliffe Hospital, Oxford OX3 9DU, United Kingdom
e-mail: catarino{at}doctors.org.uk
 |
Abstract
|
|---|
Double aortic arch often presents with tracheoesophageal compression. Management requires division of the vascular ring and, if tracheomalacia is present, some form of tracheopexy. We report here the use of a dental bar as an external splint for tracheopexy in a neonate. Follow-up to 18 months shows good tracheal growth and no erosion.
|
Introduction
|
|---|
The management of tracheomalacia secondary to a vascular ring is controversial. Division of the ring alone may be insufficient and aortopexy is commonly used to further alleviate compression. If the tracheomalacia is extensive, then some form of additional tracheal support is required either internally or externally.
A 2.4 kg female baby was born by spontaneous vaginal delivery at 38 weeks gestation. Within the first 4 hours postpartum she became markedly cyanosed with poor peripheral perfusion and was grunting with respiratory distress. She was intubated, ventilated, and transferred to the neonatal intensive care unit. Chest radiograph showed a large heart and oligemic lung fields. Initial echocardiography showed tetralogy of Fallot with a patent ductus arteriosus. An infusion of prostacyclin was commenced. She failed attempts at extubation because of respiratory stridor and was transferred to the regional cardiothoracic center.
Further echocardiography confirmed tetralogy of Fallot with pulmonary atresia and showed a double aortic arch. The dominant right aortic arch gave rise to the brachiocephalic artery and continued posterior to the trachea into the descending aorta. The left arch crossed the trachea anteriorly and gave rise to the left carotid and subclavian arteries before joining the descending aorta to complete a vascular ring. Bilateral superior vena cava, an atrial septal defect, and a large patent ductus arteriosus were also noted. The findings were confirmed by magnetic resonance imaging, which also showed significant compression of the trachea by the anterior arch.
The tetralogy of Fallot was considered unsuitable for primary repair and a modified right Blalock-Taussig shunt was performed at 6 days of age. The patent ductus arteriosus was then ligated and divided, as was the anterior aortic arch between the left common carotid and left subclavian arteries. This left the trachea free from vascular compression, and tracheopexy was deemed unnecessary, although we did not perform bronchoscopy at this time. The patient had a good hemodynamic result, but she developed stridor on extubation and fiberoptic bronchoscopy confirmed a segmental tracheomalacia affecting predominantly the left anterior segment of the trachea midway from the glottis to the carina. With increased respiratory effort this was seen to close down the tracheal lumen to 10% in expiration.
The infant underwent a further left thoracotomy 18 days after her first procedure. The trachea and left main bronchus were carefully dissected, identifying the previously preserved recurrent laryngeal nerve, and revealing a soft collapsing segment of trachea 1.5 cm in length, affecting half of the circumference. Conventional tracheopexy by this stage of the difficult dissection was impossible. A 3 cm stainless steel Erich dental bar (Fig 1A) (Medicon Instrumente, Munich, Germany) was contoured to fit the junction of the trachea and the left main bronchus. This was used to externally splint the flail segment longitudinally (Fig 1B). The dental bar was placed against the trachea to which it was fixed proximal and distal to the malacic segment using multiple interrupted 5-0 polypropylene sutures. The malacic segment was then suspended from the dental bar using further interrupted sutures. No further sutures were needed to suspend either the trachea or the dental bar itself.
Weaning from the ventilator was achieved 7 days later. Postoperative recovery was slowed by the development of a necrotizing enterocolitis and a concurrent lower respiratory tract infection. She was discharged from the hospital 4 weeks later. She has made a good recovery, her growth lies on the fifth centile, and she has normal developmental milestones at 18 months of age. She will be admitted for correction of tetralogy of Fallot in about 1 year.
|
Comment
|
|---|
Double aortic arch in association with tetralogy of Fallot is a rare condition [1]. Double aortic arch often presents with tracheoesophageal compression and the severity of the symptoms depending on the degree of compression. The more severe patients present with respiratory difficulty in infancy, whereas less severe patients present with dysphagia in older children and adults [2]. Tetralogy of Fallot characteristically presents with cyanosis and a heart murmur, without altered breath sounds. Where the conditions coexist, there may be some diagnostic difficulty but careful echocardiography provides the mainstay of diagnosis and determination of the severity of each element.
The optimal management of tracheomalacia is ill defined, particularly in the very young infant. Management strategies vary from conservative therapy to tracheal resection and reconstruction. Where tracheomalacia is clearly acquired, as in the case of vascular rings, a conservative approach may be successful and division of the vascular ring is often sufficient to relieve the compression [3]. However aortopexy is commonly used to further alleviate the tendency for tracheal collapse [4]. Internal stenting by tracheostomy with a long silicone internal stent has also been used but is miserable for a child and almost impossible in the neonate. Expandable metal endoluminal stents are used as a last resort [5] and their removal can be very difficult and bloody. Another strategy is the use of a circumferential external support [6]. The main concern in any of these cases is the uncertainty over the scope for normal tracheal growth in infants. In the case of foreign bodies there is also a potential for chronic inflammatory reactions and erosion of adjacent structures.
Our case report emphasizes the need to consider the problem of the airway in all cases of vascular ring. We initially opted to treat the trachea conservatively in view of the short history and discrete area of involvement. However, it became obvious that some form of stabilization was required to achieve independence from the ventilator. After a bloody and difficult dissection, we found that a sutured tracheopexy alone provided insufficient support. Encouraged by a previous report of the use of dental arch bars for circumferential tracheal support in an adult [7], we elected to use an improvised dental bar as an external splint. In view of discrete segmental nature of the tracheomalacia, we anticipated that placing the splint longitudinally would interfere least with growth. Nevertheless, the defined angulation imposed on the neonate’s left main bronchus remained a concern for us. The comparatively small supporting surface of the dental bar reduced the risk of erosion and was sufficient to suspend the malacic airway segment in our patient, although a more extensive segment might require circumferential support.
Follow-up has now reached 18 months with good tracheal growth, no asymmetry, and no evidence of erosion.
|
References
|
|---|
-
Virdi I.S., Keeton B.R., Shore D.F., Monro J.L. Surgical management in tetralogy of Fallot and vascular ring. Pediatr Cardiol 1987;8:131-134.[Medline]
-
Gross R.E. Arterial malformations which cause compression of the trachea or esophagus. Circulation 1955;11:124-134.[Medline]
-
Becker C.L., Ilbawi M.N., Idriss F.S., DeLeon S.Y. Vascular anomalies causing tracheoesophageal compression. J Thorac Cardiovasc Surg 1989;97:725-731.[Abstract]
-
Roberts C.S., Otterson H.B., Sade R.M., Smith C.P., III, Tagge E.P., Crawford F.A., Jr Tracheoesophageal compression from aortic arch anomalies. Analysis of 30 operatively treated children. J Ped Surg 1994;29:334-338.[Medline]
-
Filler R.M., Forte V., Fraga J.C., Matute J. The use of expandable metallic airway stents for tracheobronchial obstruction in children. J Ped Surg 1995;30:1050-1056.[Medline]
-
Hagl S., Jacob H., Sebening C., van Bodegom P., Schmidt K., Zilow E., et al. External stabilization of long-segment tracheobronchomalasia guided by intraoperative bronchoscopy. Ann Thorac Surg 1997;64:1412-1421.[Abstract/Free Full Text]
-
Waldhausen J.A., Kastl D., Orisini M.A., Conner G.H. The flat trachea: a cause of tracheal obstruction and its surgical management. Ann Thorac Surg 1982;34:461-465.[Abstract]
Top
Abstract
Introduction
