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Autoantibodies against constituents of neutrophils in the diagnosis and treatment of (isolated) subglottic stenosis.

Six patients had a subglottic stenosis either as a presenting symptom or as a manifestation of a systemic disease. All patients had in common the presence of circulating autoantibodies against constituents of neutrophils on indirect immunofluorescence. Cytoplasmic and perinuclear staining patterns were recognized. Such autoantibodies have been reported in Wegener's granulomatosis, microscopic polyarteritis, (idiopathic) glomerulonephritis, and Churg-Strauss syndrome. However, only one of the six patients fulfilled the criteria for these conditions. Because a positive test for autoantibodies against constituents of neutrophils is rare in other conditions and because other diseases had been excluded, we suggest that this places subglottic stenosis within the spectrum of necrotizing (granulomatous) vasculitis. The consequences for therapy are discussed.

Adolescent↗

Treatment of laryngotracheal stenosis with anterior and posterior cartilage grafts. A report of 41 children.

Forty-one tracheotomy-dependent children with moderate and severe laryngotracheal stenosis (Cotton grades III and IV) underwent laryngotracheal reconstruction with simultaneous use of costal cartilage grafts in the anterior and posterior subglottis and posterior glottis. The indications for this procedure are the presence of stenosis in the posterior part of the glottis, subglottis, or both in combination with anterior subglottic or upper tracheal narrowing. Other indications are the presence of circumferential subglottic stenosis, bilateral subglottic shelves, or total subglottic obstruction. The complications of this procedure included pneumothorax (one patient) and granulation tissue formation (five patients). There were two deaths unrelated to the reconstructive procedure. This technique offers an excellent opportunity for repair of laryngotracheal stenosis with a better than 90% tracheotomy decannulation rate after a single procedure within a short period after removal of the stent.

Cartilage↗

Endoscopic assessment in children with tracheotomies.

Pediatric tracheotomy is a safe procedure, and the tube can be maintained for a prolonged period. There are, however, few guidelines regarding the endoscopic assessment of the airway following tracheotomy. From January 1988 through December 1989, the Division of Otolaryngology of The Children's Hospital of Philadelphia (Pa) performed tracheotomies on 81 patients. Thirty-two children had direct laryngoscopy and bronchoscopy at the time of the procedure, 1 week later, and at 6-month intervals for a minimum of 18 months. Four children had normal airways while 21 had intraluminal stomal granulomas, 16 had development of subglottic stenosis, nine had collapse of the anterior tracheal wall, and three had development of distal tracheal granulations. Sixteen children had multiple lesions. Anatomic changes occur in the airways of the majority of children with long-term tracheotomies, and endoscopic evaluation is an essential part of their care.

Airway Obstruction↗

Carbon dioxide laser serial microtrapdoor flap excision of subglottic stenosis.

The recent development of microspot micromanipulators for the carbon dioxide laser with 250-microns spot sizes has greatly facilitated the extension of endolaryngeal procedures to children and neonates. It is possible to accurately develop microtrapdoor flaps within the limited exposures in the pediatric subglottis. We have used serial microtrapdoor flaps in the excision of subglottic scar tissue in eight children. In five children, the stenosis was improved with relief of stridor. In three children, the flaps were used as an adjunct to laryngotracheoplasty. The surgical techniques of this procedure are detailed in this report.

Child↗

Size of endotracheal tube and neonatal acquired subglottic stenosis. Study Group for Neonatology and Pediatric Emergencies in the Parisian Area.

OBJECTIVE: Risk factors for acquired laryngotracheal stenosis in newborn infants are poorly known. The extreme scarcity of acquired laryngotracheal stenosis in young infants in France, compared with the published rate in the English literature, suggested that these patients may be treated in a different way in France. A prospective study was performed to analyze local features. DESIGN: Six-month multicenter inception cohort study. SETTING: Seven neonatal intensive care units of referral centers in the area of Paris, France. PATIENTS: A total of 247 intubated surviving neonates in a consecutive sample during 6 months. INTERVENTION: A file was completed with sex, birth weight, gestational age, underlying disease, and every feature concerning the intubation period and the outcome. MAIN OUTCOME MEASURES: All the collected data were compared with those available from previous studies in the English literature. RESULTS: After extubation, respiratory outcome was uncomplicated in 242 cases. The five patients with dyspnea were treated medically and recovered. A much higher rate of extubation difficulties was reported in the English literature. The rate of stenosis in this series was significantly lower than in previously reported studies. The differences between this and other series seem to be higher birth weights, shorter duration of intubation, and, most important, a smaller tube size (2.5 mm in neonates weighing less than 2500 g and 3.0 mm in those weighing 2500 g or more). CONCLUSIONS: Although this is a limited series and many unknown variables can differ from those in previous studies, the size of the endotracheal tube appears to be a major risk factor for acquired laryngotracheal stenosis in the neonate. Further studies seem necessary to point out additional factors.

Birth Weight↗

Voice quality after laryngotracheal reconstruction.

OBJECTIVE: To ascertain changes in laryngeal anatomy and function associated with poor voice outcome after laryngotracheal reconstruction (LTR) and to specifically describe dysphonia in pediatric patients treated for subglottic stenosis. DESIGN: A survey was undertaken of patients having undergone LTR for subglottic stenosis. Twelve patients were recruited during the year 1990-1991. Nine patients were able to complete the endoscopic portion of the protocol that included fiberoptic and direct laryngoscopy. Speech samples were qualitatively evaluable in only six of nine patients (vocal quality, loudness, and intelligibility) and objectively in five of nine patients (fundamental frequency and pitch perturbation). PATIENTS: All patients in this consecutive sample had undergone LTR for congenital or acquired subglottic stenosis. The average age was 6 years. Each patient had previously undergone an average of two prior open laryngeal procedures and five endoscopic procedures. OUTCOME MEASURES: Correlation between anatomic and functional changes in the post-LTR larynx and voice outcome. RESULTS: Endoscopic evaluation of the post-LTR larynx showed a 78% incidence of altered anatomy and 44% incidence of altered function. Speech sample analysis showed the following: 100% decreased vocal quality, 50% decreased intelligibility, 100% decreased volume, and 80% low fundamental frequency and increased jitter. CONCLUSIONS: Children with high-grade subglottic stenosis and multiple prior surgeries are at high risk for poor voice outcome after LTR. Future prospective studies will allow substantiation of this observation and clarification of which changes in laryngeal structure and function arise from surgery per se.

Adolescent↗

Problems in postoperative management after anterior cricoid split.

OBJECTIVE: Describe complications and postoperative management problems associated with the anterior cricoid split. DESIGN: Retrospective case series. SETTING: Urban referral hospital. PATIENTS: Nine consecutive patients with acquired subglottic stenosis believed amenable to the anterior cricoid split. INTERVENTION: The anterior cricoid split was performed on 10 occasions between June 1989 and June 1992. MAIN OUTCOME MEASURE: Extubation or decannulation. RESULTS: Six of nine patients underwent decannulation, with success. Management problems included those related to surgery or postoperative endotracheal stenting, preexisting medical conditions, and postoperative sedation requirement. CONCLUSIONS: The number of postoperative complications and management problems was highest in those patients with both bronchopulmonary dysplasia and preexisting tracheotomy.

Bacterial Infections↗

Acquired laryngeal lesions. Pathologic study using serial macrosections.

OBJECTIVE: To present the pathologic findings of acquired lesions of the larynx in infants. SETTING: The Laryngeal Development Laboratory of Children's Memorial Hospital, Chicago, Ill. MATERIALS: One hundred fifteen larynges received at postmortem examination from 1975 to 1992. MAIN OUTCOME MEASURE: Acquired laryngeal lesions from intubation trauma. TECHNIQUE: Whole-organ serial section of larynges. RESULTS: Fifty-three of the 115 specimens exhibited acquired laryngeal lesions: 36, submucosal mucous gland hyperplasia; 12, submucosal fibrosis; 10, granulation tissue; eight, ulceration; eight, fragmented or distorted cricoid; four, cricoid ossification; four, ductal cysts; three, healed furrows, and one, anterior glottic synechia. Some specimens exhibited more than one type of acquired abnormality.

Female↗

Idiopathic subglottic stenosis.

OBJECTIVE: To review our patients with subglottic stenosis and describe a rare subclass of patients in whom the cause of subglottic scarring and narrowing remains unknown. DESIGN: A retrospective chart review and clinical update were performed on all patients seen with the diagnosis of subglottic stenosis. SETTING: The patient pool represents a collection of primary care patients within a suburb of Boston, Mass, and tertiary referrals from community and out-of-state otolaryngologists. PATIENTS: The charts of 80 patients were reviewed, and 10 of the 80 patients fulfilled the criteria for idiopathic subglottic stenosis. INTERVENTION: Diagnostic and therapeutic intervention ranged from laboratory tests, topography, flexible bronchoscopy, rigid endoscopy and biopsy, laser-assisted dilation, and resection and repair of the lesion. MAIN OUTCOME MEASURES: Attention was directed toward the character of the lesion, treatment modality, and clinical outcome. RESULTS: From 1985 to 1992, 10 patients with idiopathic subglottic stenosis were treated at the Lahey Clinic, Burlington, Mass. Eight patients required therapy for exertional dyspnea. Endoscopic laser incision and dilation were performed in all eight patients, with good initial results. Four patients were treated successfully with endoscopy alone: three required only one dilation, and the fourth required a second dilation. The remaining four patients, who had longer and more complex stenoses, have had repeated restenosis at intervals ranging from 1.5 to 12 months. Two of these patients have undergone laryngotracheal resection and reconstruction and were without evidence of restenosis 10 and 20 months after surgery. CONCLUSIONS: There exists a subclass of patients with subglottic stenosis of unknown cause. Symptomatic idiopathic subglottic stenosis may be treated successfully with laser incision and dilation if the stenosis is simple and short. More complex, longer stenoses are prone to recurrence and are more definitively managed by resection and reconstruction of the narrowed area.

Adult↗

Postoperative medical management in single-stage laryngotracheoplasty.

OBJECTIVE: To determine whether it is safe and effective to avoid the use of neuromuscular relaxants in patients who have indwelling nasotracheal tubes after undergoing single-stage laryngotracheoplasty. DESIGN: Retrospective case series. SETTING: University-based referral center specializing in the surgical management of laryngotracheal stenosis and other pathologic conditions of the airway. PATIENTS: Referred sample of 104 consecutive patients (67 males, 37 females) undergoing single-stage laryngotracheoplasty for subglottic stenosis. INTERVENTION: Single-stage laryngotracheoplasty. MAIN OUTCOME MEASURES: Incidence of accidental extubation, use of neuromuscular relaxants, incidence of reintubation after planned extubation, duration of intubation, overall rate of successful airway expansion. RESULTS: One patient self-extubated without sequelae. One patient required a brief course of neuromuscular relaxants. The success rate (without further laryngotracheoplasty) for all patients was 86% (89/104). For the 25 patients operated on in 1992, with at least 1 year of follow-up, the success rate was 92% (23/25). CONCLUSION: Single-stage laryngotracheoplasty can be done safely and effectively without using paralyzing agents in the postoperative period. This approach has certain advantages, which are discussed.

Accidents↗

The effect of gastroesophageal reflux on laryngotracheal reconstruction.

OBJECTIVES: To determine the need for preoperative evaluation for gastroesophageal reflux disease (GERD) in all children undergoing laryngotracheal reconstruction (LTR) and to assess the effect of GERD on the outcome of LTR. DESIGN: Prospective, single-blind observational study. SETTING: Tertiary care children's hospital. PATIENTS: Seventy-four pediatric patients with laryngotracheal stenosis who underwent LTR at the Children's National Medical Center, Washington, DC, from October 1, 1986, through August 31, 1994. INTERVENTION: Evaluation for and treatment of GERD, LTR, endoscopy, and removal of granulation tissue. MAIN OUTCOME MEASURES: Successful decannulation and number of endoscopies required to remove laryngeal and tracheal granulation tissue. RESULTS: Seventy-four patients underwent 82 LTRs. The senior surgeon was blinded to the status of GERD evaluation and treatment. Four groups were identified: 37 patients (40 LTRs) with no preoperative evaluation for GERD; 10 patients (11 LTRs) with normal findings on preoperative evaluation for GERD; seven patients (nine LTRs) with abnormal findings on preoperative evaluation for GERD but who failed to receive appropriate treatment; and 20 patients (22 LTRs) with abnormal findings on preoperative evaluation for GERD who received appropriate therapy. Severity and extent of stenosis as determined by multicentricity of stenosis, type of repair, and duration of stent were similar in the four groups. The effect of GERD and its treatment on the outcome of LTR was measured by the number of endoscopies necessary for removal of granulation tissue following reconstruction and successful decannulation. Statistical analyses indicate that (1) all children do not require preoperative evaluation for GERD; (2) neither the presence of GERD nor its treatment are major factors in determining the outcome of LTR. CONCLUSION: Preoperative evaluation for GERD and its treatment do not favorably affect the outcome of LTR.

Analysis of Variance↗

Analysis of voice outcomes in pediatric patients following surgical procedures for laryngotracheal stenosis.

OBJECTIVE: To evaluate long-term (> 2 years) voice function and corresponding anatomical features in children who have undergone augmentation procedures for laryngotracheal stenosis. DESIGN: Follow-up survey assessment. SETTING: Academic pediatric referral center. PATIENTS OR OTHER PARTICIPANTS: The families of 50 patients were contacted by mail, and completed home questionnaires. Of these 50 patients, 33 agreed to participate in a comprehensive hospital-based voice assessment, and 28 of these underwent fiberoptic endoscopy. INTERVENTIONS: Hospital-based evaluation and parental questionnaire. MAIN OUTCOME MEASURES: (1) Perceptual voice assessment from taped samples by using the Vocal Profile Analysis protocol performed by 3 speech and language therapists, (2) fiberoptic laryngeal endoscopy assessment performed by 3 otolaryngologists, and (3) parental assessment of voice by questionnaire. The conditions of the patients were categorized by etiology, the degree of stenosis, coexisting laryngeal lesions, and the type of surgical procedure that was done based on a review of medical records. RESULTS: Eight of 33 patients were judged perceptually by using an amended version of the Vocal Profile Analysis scheme to have "normal" voices. In the remaining 25 patients, abnormalities were demonstrated in the parameters, including harshness (52%), whisper (36%), ventricular band phonation (21%), continuity (27%), mean pitch (27%), and falsetto voice (12%). By using endoscopy, 3 of 25 larynges were judged to be anatomically normal. Abnormalities that were found in the other patients included altered vocal fold mobility (42%), abnormal subglottis (38%), supraglottic vibration (31%), and anterior commissure blunting (31%). Supraglottic vibration corresponded with the perceptual judgment "ventricular band phonation type," and was seen in children with glottic insufficiency. Only 12% of parents indicated by questionnaire that deterioration of voice and dissatisfaction with voice function occurred after surgical procedures were performed. CONCLUSIONS: A substantial proportion of children who have undergone laryngotracheal surgical procedures for stenosis demonstrate chronic voice and endoscopic abnormalities. The effects of these surgical procedures and preexisting laryngeal abnormalities on voice continue to be difficult to establish in the pediatric population. Careful management by speech and language therapists who are familiar with such children is essential for optimal voice function.

Adolescent↗

Ideal timing of pediatric laryngotracheal reconstruction.

OBJECTIVE: To determine whether there is an ideal age at which to perform a laryngotracheal reconstruction (LTR) in the pediatric population. DESIGN: Prospective observational study. SETTING: Tertiary care children's hospital. PATIENTS: Forty-eight patients aged 48 months or younger with laryngotracheal stenosis who underwent 50 LTRs from October 1, 1986, to June 30, 1995. Patients were divided into 2 groups: group 1, aged 8 through 24 months (22 patients); group 2, aged 25 through 48 months (26 patients). INTERVENTION: Endoscopy and LTR. MAIN OUTCOME MEASURES: Successful decannulation. RESULTS: Statistical analysis showed that (1) patients in group 2 had more severe degree of laryngotracheal stenosis as determined by duration of stenting with no difference in multiple sites of stenosis or type of repair required to correct laryngotracheal stenosis and (2) patients in group 2 were more likely to have successful decannulation. CONCLUSION: Laryngotracheal reconstruction at a younger age (< 25 months) is important for a child's speech and language development as well as for eliminating the morbidity and mortality associated with a tracheotomy. However, LTR at a younger age is associated with a higher risk of failure despite lesser degree of pathology. Therefore, although we still recommend LTR at a younger age since it may be beneficial for a child's speech and language development and avoidance of tracheotomy complications, this recommendation may be at the price of LTR failure and requirement for revision procedures.

Age Factors↗

Supraglottic stenosis in infants and children. A preliminary report.

OBJECTIVES: To describe a condition identified in the pediatric population that narrows the supraglottic larynx. This condition has been termed supraglottic stenosis/collapse. To discuss common factors present in these children and to examine potential causes of this specific condition. DESIGN: Retrospective review of patients with supraglottic stenosis/collapse from October 1985 to June 1993. SETTING: Academic tertiary care children's hospital. PATIENTS: Seventeen patients aged 7 months to 14 years underwent laryngoscopy and bronchoscopy for evaluation or follow-up of upper airway obstruction or respiratory failure and were found to have supraglottic stenosis/ collapse. MAIN OUTCOME MEASURES: Each patient's records were reviewed for the average age at the time of diagnosis, history of prematurity, evidence of gastroesophageal reflux, history of long-term intubation, and any associated medical problems. In addition, patients were compared based on previous airway surgery (tracheostomy, laryngotracheal reconstruction, or anterior cricoid split). RESULTS: Chronic upper airway obstruction was identified in 17 patients caused by supraglottic stenosis/ collapse. All patients (100%) had undergone tracheostomy prior to the recognition of supraglottic stenosis/ collapse. Fifty-nine percent of the patients had undergone laryngotracheal reconstruction, and 53% had a history of gastroesophageal reflux. CONCLUSION: Supraglottic stenosis/collapse is a distinct entity that may be related to prior laryngeal or tracheal surgery. This diagnosis should be considered when evaluating and treating patients with chronic upper airway obstruction following laryngeal and/or tracheal surgery.

Adolescent↗

Repair of a complete glottic-subglottic stenosis with a fibular osseocutaneous free flap.

Reconstruction of extensive laryngotracheal stenosis remains a formidable challenge. The ideal reconstructive technique has not been found because of the variability in the complexity and degree of laryngotracheal stenosis and the challenge of wound healing in a contaminated tubular structure. The application of microvascular free-tissue transfer in laryngotracheal reconstruction is limited. We used a fibula osseocutaneous revascularized flap for reconstruction of a complex laryngotracheal stenosis. The clinical course, long-term follow-up, and potential advantages and disadvantages are discussed.

Adult↗

Anterior cricoid split. Use of hyoid as autologous grafting material.

OBJECTIVE: To examine the use of hyoid as a readily available autologous grafting material for the anterior cricoid split (ACS) procedure. DESIGN: Prospective analysis of 20 patients undergoing ACS with hyoid interposition grafting for subglottic stenosis over a 3-year period. The patients received at least 1 year of follow-up after surgery. SETTING: Tertiary care children's hospital. PATIENTS: Twenty infants (age range, 2-9 months) with endoscopically confirmed acquired and congenital subglottic stenosis. Presenting symptoms included stridor, failure to extubate, and recurrent atypical croup. All 20 children underwent ACS with hyoid interposition grafting. RESULTS: All 20 patients exhibited improvement in their symptoms of airway obstruction. All 12 patients in whom extubation had previously failed subsequently underwent successful extubation. The 8 patients with symptoms of stridor and atypical croup showed marked improvement in their symptoms. Serial bronchoscopy revealed mucosal healing and incorporation of the hyoid grafts. CONCLUSION: Hyoid provides a readily available and reliable grafting material for interposition grafting in the ACS procedure for neonates and infants.

Airway Obstruction↗

Management of acute epiglottitis by nasotracheal intubation.

In 40 cases of acute epiglottitis in children, intubation was the chosen method for the management of airway obstruction. Six patients were treated without the establishment of an artificial airway, and no tracheostomies were done. No patients who were admitted to the hospital died of airway obstruction, although one sustained irreversible brain damage before admission, and two died of overwhelming infectionmthe average duration of intubation was 2.days and the average hospital stay was 5.days. Two children developed subglottic granulation tissue that was removed successfully and did not recur. Nasotracheal intubation is an acceptable method of management of epiglottitis.

Acute Disease↗

Acquired subglottic stenosis in the very-low-birth-weight infant.

In the six-year period from 1975 through 1980, acquired subglottic stenosis was recognized in eight patients during the first year of life who survived intubated mechanical ventilation as newborn infants. Seven of eight cases occurred in infants who weighed less than 1,500 g at birth. This complication was not seen in the first three years of the review, when only nasotracheal intubation was practiced. In the second three years, orotracheal intubation was the predominant practice. Seven infants required tracheostomy and the outcome was poor: none has undergone decannulation and three have died. Recognition of this disorder as a cause of inability to extubate the very-low-birth-weight infant is stressed.

Child, Preschool↗