Steroid Therapy for Tracheal Stenosis in Children

Transcription

1 Steroid Therapy for Tracheal Stenosis in Children Clinical Experience in 4 Children with Severe Strictures H. Biemann Othersen, Jr., M.D. ABSTRACT Recently a refinement in the treatment of tracheal stenosis in children has been described by Dr. H. H. Birck. He advocates a combination of dilation, intraluminal stenting, and steroid administration in high doses. In this regimen a polyvinyl (Portex) endotracheal tube is used to fashion an intraluminal stent, and prednisone is administered orally in a dose of 4 mg. per kilogram of body weight per day for two weeks. The steroid dose is gradually diminished over the ensuing four weeks. We have used this regimen, or a modification of it, in the successful treatment of 4 children with severe tracheal strictures and with tracheostomies which could not be removed. The clinical course of these children is presented. All had successful tracheal decannulation at the completion of therapy. T difficult racheal stenosis in children may be congenital, inflammatory, or traumatic; sources of trauma include iatrogenic injury from intratracheal tubes. By whatever means the stenosis occurs, it is very to treat. Until recently the only therapeutic options were repeated dilations, stenting, or surgical resection and anastomosis. In 1970 Birck [ 13 reported the cases of 7 children with laryngeal stenosis whom he treated with intraluminal stents and systemic steroid administration. Since his report we have treated 4 children who had tracheal strictures with complications. The therapeutic regimen was patterned after that recommended by Birck, but there were a few modifications. These clinical case histories are reported to illustrate the problems involved in the use of intraluminal stents combined with steroid therapy. The various modifications of Birck s technique will be reported in more detail in a forthcoming publication. Clinical Experience Patient 1. A 4I/2-month-old girl had had stridor and episodes of cyanosis since birth. An ear, nose, and throat evaluation had been obtained, and bronchoscopic examination revealed tracheomalacia. Tracheostomy had been performed, and it was thought that there was an anomalous vessel cross- F.rom the Department of Surgery, Medical University Hospital, 80 Barre St., Charleston, S.C Presented at the Twentieth Annual Meeting of the Southern Thoracic Surgical Association, Louisville, Ky., Nov. 1-3, THE ANNALS OF THORACIC SURGERY

2 Steroid Therapy for Tracheal Stenosis in Children ing anterior to the trachea and possibly producing the obstruction. Angiograms demonstrated no anomalous vessels; and visualization with the bronchoscope showed the previously described subglottic tracheomalacia, but there was no anterior compression of the trachea. It was necessary to perform a second and higher tracheostomy because the first was very low in the neck. The child was discharged, and over the ensuing nineteen months multiple attempts were made to remove the tracheostomy tube. A tight subglottic stenosis remained, and the stricture could be dilated only up to 18F. After communication with Dr. Howard Birck, it was decided to correct the patient s problem by insertion of an intraluminal stent and the systemic administration of steroids. The trachea was dilated to the size of a 22F catheter, and a stent was constructed from a polyvinyl (Portex) endotracheal tube. Repeat endoscopical evaluation and dilation to 24F were performed. The previously constructed 4.5 mm. stent (external diameter 7 mm.) was inserted and fixed into place. The patient received prednisone orally as follows: 4 mg./kg./day for the first two weeks 3 mg./kg./day, third week 2 mg./kg./day, fourth week 1.5 mg./kg./day, fifth week 1 mg./kg./day, sixth week The stent remained in place for the six weeks and was then removed with direct visualization. The steroid doses were then rapidly decreased during the next two weeks, and no corticotropin was given. The tracheal lumen was adequate and had a normal appearance. The tracheostomy tube was removed. The child has been followed closely for three years since the tube removal, and she has developed normally without recurrence of the difficulty. Patient 2. A 7-year-old boy with tetralogy of Fallot underwent openheart repair. The postoperative course was complicated, and use of a nasotracheal tube with respirator support was necessary for 11 days. The tracheostomy was deferred because of a generalized bleeding problem. Over the next 30 days he developed stridor and evidence of tracheal stricture: and in spite of dilation of the trachea, tracheostomy was necessary. During the year ensuing from the time of tracheostomy until September, 1971, thirteen separate attempts at dilation of the stricture were made. On occasion the trachea could be dilated up to 26F, but stenosis rapidly recurred. After one year dilation could be performed to 20F only with considerable difficulty. He was then considered for therapy with intraluminal stenting and steroid administration; however, preexisting problems and severe osteomyelitis of the sternum with a superimposed fungus infection contraindicated the use of systemic steroids. The trachea was dilated to 22F, and 100 mg. (2.5 ml.) of triamcinolone was injected into the stricture in four

3 OTHERSEN different quadrants. The boy weighed 17.3 kg., so this amount represented a dose of 5.8 mg. per kilogram of body weight. A polyvinyl endotracheal tube with an internal diameter (ID) of 4.0 mm. was fashioned as a stent and fixed into place. Three weeks later the small stent was removed, the trachea was dilated to 28F, and a 5.0 mm. ID tube was inserted. No steroids were injected during that procedure. After an additional two weeks the stent was removed and the trachea dilated to 28F. The stricture was much more pliable. It was injected with 20 mg. (2 ml.) of triamcinolone, and a 5.5 mm. ID stent was inserted. The plan was to leave the stent in for another two weeks, but the child coughed up the tube after one week. The stent had remained in place for a total of 39 days. When the trachea was found to be of normal size with no stenosis, the tracheostomy tube was then removed. The child did well except for a very small tracheocutaneous fistula which drained small amounts of mucus. Two years later a small, persistent tracheocutaneous fistula was excised; the trachea appeared normal at that time. The child continues to do well. Patient 3. An 8-year-old boy was riding a minibike at dusk and failed to see a barbed wire fence. The wire struck him in the middle of the neck, and he was thrown from the bike. He complained of some pain, and on the way to our emergency room he went into respiratory arrest. During resuscitative efforts subcutaneous emphysema was noted. Immediate emergency tracheostomy was performed, and the child responded promptly. The subcutaneous emphysema was extensive for a few days, and pneumoperitoneum developed. His sensorium cleared, and an esophagogram showed no evidence of injury. He improved gradually, and 8 days after injury a bronchoscopic examination was performed. No lumen could be found in the trachea just below the cricoid cartilage. At that point the trachea was severely comminuted, and a ureteral catheter was passed from the tracheal stoma in order to demonstrate the lumen. The trachea was dilated, and a 4.5 mm. polyvinyl stent was inserted. The child was discharged with instructions to take prednisone orally, 4 mg. /kg./day. Two weeks later the patient was readmitted, and a 5 mm. stent was inserted into the trachea. One month later an attempt was made to insert a short stent which did not protrude through the vocal cords. The stent was left open and was not packed with gauze. The following day the stent became dislodged, and it was replaced with a long one. At that time the trachea was dilated to 26F, and 120 mg. (3 ml.) of triamcinolone was injected into the area of scar. Since the patient s weight was 22.7 kg., this injection represented a single dose of 5 mg. per kilogram of body weight. The boy was then discharged with instructions to take 200 mg. of prednisone for five doses (4 mg./kg./day); 100 mg. for five doses (2 mg./kg./day); and 50 mg. for five doses (1 mg./kg./day) on alternate days. 256 THE ANNALS OF THORACIC SURGERY

4 Steroid Therapy for Tracheal Stenosis in Children Three weeks later considerable granulations in the trachea were cauterized and a 5.5 mm. Portex stent was inserted. He was discharged with directions to take prednisone, 50 mg. every other day (1 mg./kg./day), and after 14 days tracheal granulations were again cauterized and a 5 mm. stent was inserted. Prednisone was continued in the same dosage, and after the stent had been in place for 91 days it was removed. Respiratory difficulty occurred as the tracheostomy was being reduced. The stenosis had recurred and could be dilated to 24F. At that time vocal cord movement was evaluated, and bilateral vocal cord paralysis was believed to be producing the respiratory problem. This observation was confirmed one week later. At that time the trachea was dilated to 26F and 100 mg. (2.5 ml.) of triamcinolone was injected into the area of stricture. A 4.5 mm. stent was then inserted. The patient continued to take prednisone in a dose of 2 mg./kg./day; two weeks later the stricture was again injected wtih 100 mg. (2.5 ml.) of triamcinolone and a 5 mm. stent was inserted. Prednisone was continued at 2 mg./kg./day. After three weeks granulations were cauterized; 80 mg. (2 ml.) of triamcinolone was injected into the stricture, which was much softer; and more granulation tissue was excised. After the stent had been in place for a total of 147 days, the trachea was dilated and 100 mg. of triamcinolone (2.5 ml.) was injected into the stricture. Simultaneously a right arytenoidopexy was performed. Over the next two weeks the steroids were discontinued and the tracheostomy tube was removed. At the present time the child is undergoing speech therapy and has a good airway and an acceptable voice. Patient 4. A 14-month-old boy was admitted with a diagnosis of severe laryngotracheobronchitis. Respiratory distress necessitated insertion of an endotracheal tube. A tube 5 mm. in internal diameter and 8 mm. in external diameter had been inserted and was found to fit snugly. The tube remained in place for 3 days, and when it was removed, respiratory distress ensued. Tracheostomy was performed, and bronchoscopic examination revealed subglottic stenosis which would admit only a size 14F catheter. Three days later the stricture was dilated, and bronchoscopy showed that the stricture could be dilated to 24F. A 4 mm. polyvinyl stent was inserted. The child had been on prednisone, 3 mg./kg./day for the first week of hospitalization and 2.5 mg./kg./day for the second week. When the stent was inserted, he was given 50 mg. of prednisone daily (4 mg./kg./day) for the first two weeks. For the next two weeks he was given 3 mg./kg./day, and then the dosage was reduced to alternate days. At the end of four and one-half weeks, cauterization of tracheal granulations was performed and a 4.5 mm. stent was inserted. Two weeks later the stent was removed and 40 mg. (1 ml.) of triamcinolone was injected into the area of stricture. The stent had been in place for 48 days. Steroid doses were rapidly tapered off and discontinued over the ensuing week.

5 OTHERSEN The child was discharged after two and one-half months of hospitalization. Comment Tracheal stenosis can be a severe, life-threatening problem, and its long-term management is difficult and protracted. Many strictures can and should be prevented. If tracheal injury does occur, prompt recognition is important in order to initiate therapy before dense scarring develops. Each of the 4 cases presented illustrates the method for handling a different type of tracheal stricture. The case of Patient 1 illustrates a congenital subglottic stenosis, which is probably best treated by intraluminal stenting. Excision in this area would be extremely difficult. Patient 2 s history shows the problem of protracted endotracheal intubation with respirator attachment. The constant to-and-fro movement of the tube with respiration produces severe damage to the tracheal wall. In preventing these lesions, it is important to remember that a soft endotracheal tube may be left in place for up to two weeks, but if a respirator is needed or if the tube must remain longer than 14 days, a tracheostomy should be performed. Careful attention to the details of tracheostomy is necessary. It should be remembered that the endotracheal tube need not fit the trachea snugly and that a small air leak may actually be desirable in that it indicates the tube is not impinging on the tracheal wall. If an air leak occurs, the volume of the respirator input can be increased to compensate for the leak. Cuffed tubes are very rarely necessary in children. Patient 3 had an extensive comminuted fracture of the trachea. The trachea was not cleanly transected but was fractured over a distance of approximately 4 cm. In such injuries early recognition and prompt tracheostomy are mandatory as a lifesaving measure. The fractured trachea should then be reduced when the patient s condition is stable, preferably within 5 days. Perhaps it may have been better to perform open exploration along with the endotracheal stenting in order to secure the best reduction of the tracheal fragments. The area of injury was probably too long for resection and primary anastomosis. The history of Patient 4 shows the severe problems that may develop when a large endotracheal tube is placed through an area of inflamed trachea. Injury occurs rapidly, and these patients are best treated by early tracheostomy. In the treatment of these patients, long-term dilations were tried without lasting benefit. The only other acceptable treatment was tracheal resection with anastomosis; but the risk of injury to the recurrent laryngeal nerves from resection is considerable, and restenosis at the suture line may occur. The technique described by Birck [l] appears to be an acceptable alternative. It remains to be seen whether the use of steroids in high doses will 258 THE ANNALS OF THORACIC SURGERY

6 Steroid Therapy for Tracheal Stenosis in Children produce problems. All children receiving these high doses were kept on antacids in order to decrease the. incidence of stress ulcers. The parents were warned of potential difficulties, but in these 4 patients no complications from steroid administration were seen. We believe that the procedures described herein deserve further clinical trial in order to prove or disprove their real value. Reference 1. Birck, H. H. Endoscopic repair of laryngeal stenosis. Trans. Am. Acad. Ophthalmol. Otolaryngol. 74: 140, NOTICE FROM THE SOUTHERN THORACIC SURGICAL ASSOCIATION The Twenty-first Annual Meeting of the Southern Thoracic Surgical Association will be held at the Williamsburg Inn and Lodge, Williamsburg, Va., November 7-9, Reservations may be made by writing to the Reservations Manager, The Williamsburg Inn and Lodge, Williamsburg, Va Members wishing to participate in the scientific program should submit abstracts-typed double-spaced and in triplicate-to William H. Lee, Jr., M.D. (Chairman of the Program Committee), Medical College Hospital, 55 Doughty St., Charleston, S.C The deadline for submission of abstracts is June 1, The Council of the Southern Thoracic Surgical Association has ruled that all slides used during presentation of papers and discussions must be of 35-mm. size. If additional information is required, inquiries should be directed to Dr. Lee. Application for membership in the Southern Thoracic Surgical Association, on forms provided by the Association, should be sent directly to George R. Daicoff, M.D. (Chairman of the Membership Committee), University of Florida College of Medicine, Gainesville, Fla The deadline for application to membership is September Papers that are accepted for the program will be considered for publication in The Annals and must be submitted to the Editor by October 15, JAMES W. BROOKS, M.D. Secretary-Treasurer