Balloon Dilatation
of Long-Segment
Tracheal
Stenoses
By Charles E. Bagwell, James L. Talbert, and Joseph J. Tepas Ill
Gainesville, Florida 0 Although a rare anomaly, congenital tracheal stenosis is associated with a high mortality. Surgical approaches proposed for this life-threatening lesion have included resection of localized stenoses; tracheoplasty involving interposition of cartilage, periosteum, or pericardium via an anterior tracheal split; or esophageal anastomosis to a posterior tracheal split. None of these techniques have provided uniformly satisfactory results, and all entail significant morbidity associated with the tracheal anastomosis. We have used endoscopic balloon dilatation in four children with critical airway obstruction from long-segment tracheal stenoses. In each of these patients, symptoms began early in life (from 2 weeks to 6 months of age) and consisted of respiratory distress from upper airway obstruction or recurrent episodes of stridor and chest retractions requiring hospitalization. The location of the tracheal stenoses varied, involving the upper third, middle third, middle half, and distal half of the trachea in the patients encountered. At bronchoscopy, the lumen was judged to be 2 1 mm in three of these patients. Dilatation was accomplished using Gruentzig balloon catheters in sizes ranging from 3 mm to (eventually) 6 mm, and produced a posterior split in the complete tracheal rings responsible for the stenosis in all patients, resulting in great increases in airway lumen. All children were improved following balloon dilatation, and three are currently doing well at 2.5, 3.0, and 3.0+ years postdilatation. One infant eventually died of airway obstruction incurred by severe bronchomalacia distal to the tip of an endotracheal tube that had been passed to the carina after sequential balloon dilatations. Although management of long-segment tracheal stenoses remains a difficult and challenging problem to the pediatric surgeon, based on our experience we believe endoscopic balloon dilatation provides an alternative technique for splitting the offending stenotic cartilaginous rings while avoiding the risks of tracheal anastomoses. Copyright o 1991 by W.B. Saunders Company INDEX WORDS: Tracheal stenosis, balloon dilatation.
A
LTHOUGH THERE are many causes of acute respiratory distress in the infant, one of the most challenging to pediatric surgeons involves congenital stenosis of long segments of the trachea. Affected infants, although thankfully rare, usually present early in life with acute onset of life-threatening respiratoq distress. Controversies currently exist in methods of diagnostic evaluation and in methods of surgical management, which may include resection (of short-segment stenoses); posterior tracheoplasty to esophagus; or anterior tracheal split with interposition of cartilage, periosteum, or pericardium. Use of endoscopic balloon dilatation (EBD) in four children with long-segment tracheal stenoses prompts review of this modality in overall treatment of these complicated lesions. JournalofPediatricSurgery,
Vol26, No 2 (February), 1991: pp 153-159
CASE REPORTS
Patient 1 This 6-week-old boy was referred to the Pediatric Intensive Care Unit at Shands Teaching Hospital from a local hospital with acute respiratory distress. After initial stabilization, airway fluoroscopy showed severe stenosis involving the trachea from subglottic to supracarinal regions (Fig 1). Bronchoscopy confirmed the stenosis suspected radiographically, and showed the proximal airway lumen to be less than 1 mm in diameter (Fig 2). Repeat bronchoscopy was performed with preparation for cardiopulmonary bypass and an anterior tracheal split with cartilage interposition as described by Kimura et al,’ but endoscopic dilatation of the stenotic trachea was performed using a 3-mm Gruentzig balloon catheter (Microfasive Inc, Watertown, MA). This resulted in disruption of the posterior aspect of the complete cartilaginous rings responsible for the tight stenosis (Fig 3). Immediate improvement in ventilation was apparent (Fig 4). Though repeated endoscopic procedures with laser excision of tracheal granulation tissue and tracheostomy have been necessary in this child, at greater than 3 years post-EBD he has normal development and no airway symptoms.
Patient 2 This S-month-old boy was noted to have a “tight trachea” while undergoing general anesthetic at a local hospital for hernia repair. At this time, only a 25mm endotracheal tube could be inserted for a short distance, and had to be held in place during the procedure by the anesthesiologist. After a relatively uneventful recovery, he was readmitted 3 weeks later with severe respiratory distress and cyanosis. At the time of transfer to Shands Teaching Hospital, he was immediately begun on heliox (helium-oxygen mixture) to lower the viscosity of airflow through the stenotic orifice. Airway fluoroscopy demonstrated tracheal stenosis extending from midtrachea to (and perhaps beyond) the carina (Fig 5). Due to the length of involved trachea, sequential dilatation procedures were performed with posterior splitting of the tracheal cartilages as described in Patient 1. Following the second such procedure, sudden difficulty in ventilation was voiced by the anesthesiologist on moving the patient from the operating table onto a transport bed, with complete inability to squeeze the Ambu bag. Fearing obstruction by blood clot or tissue flap, a Jackson tracheal dilator was expediently passed via laryngoscopic visualization and restored airflow after dislodgement of the offending obstruction. However, sudden appearance of subcutaneous emphysema necessitated bilateral chest tubes for pneumothoraces and a pneumomediastinum, which suggested tracheal disruption. During the ensuing 2 weeks ventilatory assistance was necessary via an indwelling endotracheal tube cautiously passed as a stent through the site of suspected tracheal injury, and gradual resolution of the subcutane-
From the Division of Pediatric Surgety, Universityof Florida College of Medicine, Gainesville, FL. Date accepted: December 15, 1989. Address reprint requests to Charles E. Bagwell, MD, Division of Pediatric Surgery, University of Florida College of Medicine, Box J-286, J. Hillis Miller Health Center, Gainesville, FL 32610. Copyright o 1991 by W B. Saunders Company 0022-3468/91/2602-0008$03.00l0 153
154
BAGWELL, TALBERT, AND TEPAS
bronchial orifices was noted. At this time a 5-mm endotracheal tube was positioned at the carina as a stent. After an unremarkable postanesthetic recovery, the child suddenly died the same evening, presumably secondary to a distal mucous plug complicated by distal bronchomalacia. Autopsy confirmed the long-segment congenital tracheal stenosis with healing of the tracheal perforation suspected earlier (Fig 8).
Patient 3 This 17-month-old dysmorphic girl with monosomy 18 and multiple congenital anomalies was referred to Shands Teaching Hospital for “congenitally small airways and recurrent croup” since age 6 months. On arrival, she was noted to be stridulous with respiratory distress. Bronchoscopy demonstrated tight stenosis (
of Long-Segment
Tracheal
Stenoses
By Charles E. Bagwell, James L. Talbert, and Joseph J. Tepas Ill
Gainesville, Florida 0 Although a rare anomaly, congenital tracheal stenosis is associated with a high mortality. Surgical approaches proposed for this life-threatening lesion have included resection of localized stenoses; tracheoplasty involving interposition of cartilage, periosteum, or pericardium via an anterior tracheal split; or esophageal anastomosis to a posterior tracheal split. None of these techniques have provided uniformly satisfactory results, and all entail significant morbidity associated with the tracheal anastomosis. We have used endoscopic balloon dilatation in four children with critical airway obstruction from long-segment tracheal stenoses. In each of these patients, symptoms began early in life (from 2 weeks to 6 months of age) and consisted of respiratory distress from upper airway obstruction or recurrent episodes of stridor and chest retractions requiring hospitalization. The location of the tracheal stenoses varied, involving the upper third, middle third, middle half, and distal half of the trachea in the patients encountered. At bronchoscopy, the lumen was judged to be 2 1 mm in three of these patients. Dilatation was accomplished using Gruentzig balloon catheters in sizes ranging from 3 mm to (eventually) 6 mm, and produced a posterior split in the complete tracheal rings responsible for the stenosis in all patients, resulting in great increases in airway lumen. All children were improved following balloon dilatation, and three are currently doing well at 2.5, 3.0, and 3.0+ years postdilatation. One infant eventually died of airway obstruction incurred by severe bronchomalacia distal to the tip of an endotracheal tube that had been passed to the carina after sequential balloon dilatations. Although management of long-segment tracheal stenoses remains a difficult and challenging problem to the pediatric surgeon, based on our experience we believe endoscopic balloon dilatation provides an alternative technique for splitting the offending stenotic cartilaginous rings while avoiding the risks of tracheal anastomoses. Copyright o 1991 by W.B. Saunders Company INDEX WORDS: Tracheal stenosis, balloon dilatation.
A
LTHOUGH THERE are many causes of acute respiratory distress in the infant, one of the most challenging to pediatric surgeons involves congenital stenosis of long segments of the trachea. Affected infants, although thankfully rare, usually present early in life with acute onset of life-threatening respiratoq distress. Controversies currently exist in methods of diagnostic evaluation and in methods of surgical management, which may include resection (of short-segment stenoses); posterior tracheoplasty to esophagus; or anterior tracheal split with interposition of cartilage, periosteum, or pericardium. Use of endoscopic balloon dilatation (EBD) in four children with long-segment tracheal stenoses prompts review of this modality in overall treatment of these complicated lesions. JournalofPediatricSurgery,
Vol26, No 2 (February), 1991: pp 153-159
CASE REPORTS
Patient 1 This 6-week-old boy was referred to the Pediatric Intensive Care Unit at Shands Teaching Hospital from a local hospital with acute respiratory distress. After initial stabilization, airway fluoroscopy showed severe stenosis involving the trachea from subglottic to supracarinal regions (Fig 1). Bronchoscopy confirmed the stenosis suspected radiographically, and showed the proximal airway lumen to be less than 1 mm in diameter (Fig 2). Repeat bronchoscopy was performed with preparation for cardiopulmonary bypass and an anterior tracheal split with cartilage interposition as described by Kimura et al,’ but endoscopic dilatation of the stenotic trachea was performed using a 3-mm Gruentzig balloon catheter (Microfasive Inc, Watertown, MA). This resulted in disruption of the posterior aspect of the complete cartilaginous rings responsible for the tight stenosis (Fig 3). Immediate improvement in ventilation was apparent (Fig 4). Though repeated endoscopic procedures with laser excision of tracheal granulation tissue and tracheostomy have been necessary in this child, at greater than 3 years post-EBD he has normal development and no airway symptoms.
Patient 2 This S-month-old boy was noted to have a “tight trachea” while undergoing general anesthetic at a local hospital for hernia repair. At this time, only a 25mm endotracheal tube could be inserted for a short distance, and had to be held in place during the procedure by the anesthesiologist. After a relatively uneventful recovery, he was readmitted 3 weeks later with severe respiratory distress and cyanosis. At the time of transfer to Shands Teaching Hospital, he was immediately begun on heliox (helium-oxygen mixture) to lower the viscosity of airflow through the stenotic orifice. Airway fluoroscopy demonstrated tracheal stenosis extending from midtrachea to (and perhaps beyond) the carina (Fig 5). Due to the length of involved trachea, sequential dilatation procedures were performed with posterior splitting of the tracheal cartilages as described in Patient 1. Following the second such procedure, sudden difficulty in ventilation was voiced by the anesthesiologist on moving the patient from the operating table onto a transport bed, with complete inability to squeeze the Ambu bag. Fearing obstruction by blood clot or tissue flap, a Jackson tracheal dilator was expediently passed via laryngoscopic visualization and restored airflow after dislodgement of the offending obstruction. However, sudden appearance of subcutaneous emphysema necessitated bilateral chest tubes for pneumothoraces and a pneumomediastinum, which suggested tracheal disruption. During the ensuing 2 weeks ventilatory assistance was necessary via an indwelling endotracheal tube cautiously passed as a stent through the site of suspected tracheal injury, and gradual resolution of the subcutane-
From the Division of Pediatric Surgety, Universityof Florida College of Medicine, Gainesville, FL. Date accepted: December 15, 1989. Address reprint requests to Charles E. Bagwell, MD, Division of Pediatric Surgery, University of Florida College of Medicine, Box J-286, J. Hillis Miller Health Center, Gainesville, FL 32610. Copyright o 1991 by W B. Saunders Company 0022-3468/91/2602-0008$03.00l0 153
154
BAGWELL, TALBERT, AND TEPAS
bronchial orifices was noted. At this time a 5-mm endotracheal tube was positioned at the carina as a stent. After an unremarkable postanesthetic recovery, the child suddenly died the same evening, presumably secondary to a distal mucous plug complicated by distal bronchomalacia. Autopsy confirmed the long-segment congenital tracheal stenosis with healing of the tracheal perforation suspected earlier (Fig 8).
Patient 3 This 17-month-old dysmorphic girl with monosomy 18 and multiple congenital anomalies was referred to Shands Teaching Hospital for “congenitally small airways and recurrent croup” since age 6 months. On arrival, she was noted to be stridulous with respiratory distress. Bronchoscopy demonstrated tight stenosis (
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