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Phenytoin-induced lingual tonsil hyperplasia causing laryngeal obstruction.

The effects of phenytoin sodium on lymphoid tissue have been known for some time. However, its effect on the lymphoid tissue of Waldeyer's ring is unreported in the otolaryngology literature. We present the case of a 78-year-old woman who was treated with phenytoin for generalized tonic-clonic seizures for 4 months and who subsequently developed lingual hyperplasia that caused laryngeal obstruction eventually requiring a tracheotomy. Of note, no history of airway compromise or difficulty with intubation from a prior operation 20 years ago was reported; these findings suggest that the lingual tonsillar hyperplasia was a consequence of phenytoin therapy and not a preexisting condition. The findings of histopathologic evaluation were consistent with lymphoid hyperplasia without evidence of malignancy. We also report our operative management and follow-up of this rare condition.

Aged↗

Prevention of anterior glottic restenosis in a canine model with topical mitomycin-C.

To develop a model for recurrent anterior glottic stenosis and to test the efficacy of topical mitomycin-C in preventing restenosis, we induced anterior glottic stenosis with a CO2 laser in 5 dogs. In 3 dogs, recurrence was established after surgical lysis. Subsequently, the 3 dogs received a single topical 3-minute treatment with a 1% solution of mitomycin-C after a second surgical lysis. In a parallel experiment, the other 2 dogs received a single topical 3-minute treatment with a 1% solution of mitomycin-C after the initial surgical lysis. An anterior glottic web was induced in all 5 dogs with the CO2 laser. The 3 dogs experienced restenosis at the anterior glottis after surgical lysis alone. Mitomycin-C prevented anterior glottic restenosis in 2 of the 3 dogs treated twice and in both of the dogs treated once (p = .02). We conclude that a recurrent stenosis of the anterior glottis may be induced reproducibly in the canine model with the CO2 laser. Application of topical mitomycin-C after lysis of an anterior glottic stenosis produces a statistically significant reduction in the rate of restenosis as compared to surgical lysis alone.

Administration, Topical↗

Surgery for pediatric subglottic stenosis: disease-specific outcomes.

To set the foundation to develop a disease-based, operation-specific model to predict the outcome of pediatric airway reconstruction surgery, we performed a retrospective database review of children operated on at a single, tertiary-care children's hospital. Over the 12-year period 1988 to 2000, a total of 1,296 airway reconstruction procedures were performed. Out of these, charts were identified for 199 children who underwent laryngotracheal reconstruction for a sole diagnosis of subglottic stenosis. Children were excluded from the study if their disorder included supraglottic, glottic, or upper tracheal disease. The main outcome measures were Myer-Cotton grade-specific decannulation and extubation rates, including both operation-specific and overall results. There were 101 children who underwent double-stage laryngotracheal reconstruction. The operation-specific decannulation rates for Myer-Cotton grades 2, 3, and 4 were 85% (18/21), 37% (23/61), and 50% (7/14) (chi2 analysis, p = .0007). The overall decannulation rates were 95% (20/21), 74% (45/61), and 86% (12/14) (chi2 analysis, p = .04). There were 98 children who underwent single-stage laryngotracheal reconstruction. The operation-specific extubation rates for Myer-Cotton grades 2, 3, and 4 were 82% (37/45), 79% (34/43), and 67% (2/3) (chi2 analysis, p = .63). The overall extubation rates were 100% (45/45), 86% (37/43), and 100% (3/3) (chi2 analysis, p = .03). Logistic regression analysis showed no effect of age (less than or greater than 2 years of age) on operation-specific or overall outcome parameters. We conclude that laryngotracheal reconstruction for pediatric subglottic stenosis remains a challenging set of procedures in which multiple operations may be required to achieve eventual extubation or decannulation. Children with Myer-Cotton grade 3 or 4 disease continue to represent a significant challenge, and refinements of techniques are being examined to address this subset of children. Disease-based, operation-specific outcome statistics are the first step in the development of a meaningful predictive model.

Adolescent↗

Virtual laryngotracheal endoscopy based on geometric surface modeling using spiral computed tomography data.

This prospective study describes the clinical utility of virtual endoscopy based on geometric surface modeling of the laryngotracheal lumen. Eighteen children with dyspnea related to either subglottic angioma (n = 5) or laryngotracheal stenosis (n = 13) were included. All patients underwent video-recorded operative endoscopy, spiral computed tomography, and 3-dimensional reconstruction of the laryngotracheal lumen. Modeling was achieved by contour detection on spiral computed tomographic images and reconstruction using a geometric shape-recognition algorithm. The generated surface was used for diagnosis and measurement using interactive and automatic tools. Findings of virtual endoscopy and operative endoscopy were compared. Virtual endoscopy confirmed narrowing of the airway in all cases. In 9 cases, high-grade stenosis prevented complete operative endoscopy, but virtual endoscopy allowed accurate assessment and measurement of the stenosis. The findings of operative and virtual endoscopy were concordant in 9 cases. We conclude that surface modeling provides valuable information for preoperative evaluation of laryngotracheal narrowing. The ability to assess extraluminal anatomy provides a clearer picture of overall disease involvement. In the future, virtual endoscopy will probably be used in conjunction with operative endoscopy for therapeutic decision-making. Noninvasive virtual endoscopy could become an alternative to traditional endoscopy under general anesthesia for therapeutic follow-up.

Adolescent↗

Subglottic stenosis complicated by allergic esophagitis: case report.

Allergic esophagitis is a known entity that had been described in patients with dysphagia. It has not been previously described in association with subglottic stenosis. We report the case of a 2-year-old girl with symptoms suggestive of allergic esophagitis who suffered from subglottic stenosis that recurred despite surgical measures. Her esophageal pH monitoring results were normal, and she did not respond to antireflux medications. She did respond dramatically to corticosteroid therapy with improvement of both her esophageal and laryngeal symptoms. Allergic esophagitis as a clinical entity is discussed.

Child, Preschool↗

Long-term results of the sternohyoid myocutaneous rotary door flap for laryngotracheal reconstruction.

In the past 8 years, 45 patients with laryngotracheal stenosis have undergone reconstruction with the sternohyoid myocutaneous rotary door flap (RDF) technique at Tang Du Teaching Hospital in Xi'an, China. All patients were tracheostomy-dependent before reconstruction. Thirty-six patients had previously undergone 2 to 7 surgical procedures. Forty-one of the 45 patients (91%) were successfully decannulated. Decannulation failed in the other 4 because of RDF prolapse into the tracheal lumen or because of hypertrophic scar in the operative region. The follow-up period ranged from 1 to 8 years. Forty of the 41 patients had a stable airway and effective voice, and 1 patient had restenosis 3 years after decannulation. The results demonstrate that the RDF technique is a relatively simple and effective way to treat patients with laryngotracheal stenosis.

Adolescent↗

Idiopathic subglottic stenosis revisited.

Idiopathic subglottic stenosis (ISS) is a rare inflammatory process of unknown cause, usually limited to the subglottic region and the first 2 tracheal rings. We performed a retrospective analysis of our experience with a series of patients with this condition. The study involved retrospective review of the records of patients with ISS. A series of criteria that included patient clinical history, laboratory tests, flexible nasolaryngoscopic examination, and biopsies were used to establish a diagnosis. Symptoms, treatment, and outcome were examined. A total of 16 patients were identified; 14 were female (87.5%), and 2 were male. The mean follow-up time was 75.5 months. Fourteen patients required surgical treatment for respiratory compromise. Nine of these cases have been controlled effectively with endoscopic laser techniques. Endoscopic management failed in 5 patients, and 2 patients underwent laryngotracheal resection and reconstruction. Mitomycin-C was used in 6 patients as an adjuvant to endoscopic laser surgery. Patients in whom endoscopic treatment failed were noted to have thicker (>1 cm) and more complex stenoses. The diagnosis of ISS is a diagnosis of exclusion. A complete workup of the patient must be performed to rule out other causes of stenosis. Endoscopic laser surgery is a valid initial approach for thinner, noncomplicated lesions. Patients with thicker, complex lesions in whom endoscopic treatment fails are best managed with laryngotracheal resection and reconstruction. However, adjuvant use of mitomycin-C may prove beneficial in the treatment of these patients. Estrogen may play a role in the pathogenesis of ISS by altering the wound healing response.

Adult↗

Cicatricial pemphigoid in the upper aerodigestive tract: diagnosis and management in severe laryngeal stenosis.

Pemphigoid is a group of rare, acquired, autoimmune subepithelial blistering diseases. The condition has been subclassified into bullous pemphigoid and cicatricial pemphigoid (CP). Diagnosis is based on clinical presentation, evidence of subepithelialvesicles or bullae on routine histologic analysis, and direct and indirect immunofluorescence studies. Cicatricial pemphigoid is characterized by linear deposition of immunoreactants, principally IgG and complement factor 3, along epithelial basement membranes. Cicatricial pemphigoid usually leads to mucosal scarring. We present a case of severe CP that led to laryngeal and subglottic stenosis and involvement of both eyes and the oral, nasal, and nasopharyngeal mucosae. Treatment with dapsone, corticosteroids, azathioprine sodium, cyclosporine A, cyclophosphamide, methotrexate sodium, and mycophenolate mofetil between 1997 and 2001 only resulted in temporary disease control. The patient has been treated with leflunomide for the past 8 months, and there have been no relapses. Treatment of CP with leflunomide has not been described in the literature until now.

Adult↗

Laryngotracheal growth following cricotracheal resection combined with laminotomy. An experimental study on rabbits.

Cricotracheal resection (CTR) has been shown to be successful in the treatment of pediatric subglottic stenosis. Whether CTR combined with posterior midline division or partial resection of the cricoid plate affects the stability and growth of the larynx is still questionable. The study included 24 infant female New Zealand White rabbits that were divided into 4 groups of 6 animals each. Group 1 was the unoperated control group. Group 2 underwent removal of three quarters of the anterior cricoid circumference, followed by a primary anastomosis (CTR). In addition to CTR, the rabbits in group 3 underwent a laminotomy by splitting of the cricoid plate midline. Group 4 underwent both CTR and partial resection of the lamina. No stents were used in this study. After 6 months, the experiment was terminated, and the following observations were made. Groups 2 and 3 presented a large and stable subglottic lumen, comparable to that of the control group. After partial cricoid plate resection, 4 of the rabbits in group 4 suffered from a subglottic collapse that caused an airway obstruction and resulted in death. These results support the conclusion that CTR alone, as well as combined with laminotomy, can be performed in a relatively safe and effective manner without interfering with the development of the larynx in young rabbits. Cricotracheal resection together with partial resection of the cricoid plate, however, leads to an unstable situation with an impaired airway.

Age Factors↗

Successful treatment of idiopathic laryngotracheal stenosis by resection and primary anastomosis.

We studied the early and long-term response of idiopathic laryngotracheal stenosis (ILTS) to treatment by 1-stage laryngotracheal or tracheal resection and reconstruction in 73 patients. Nineteen of the 72 (26%) noted no difference in their voices or any difficulty in breathing after reconstruction. Forty-seven (64%) described loss of ability to project their voices as loudly as before or noted some difficulty in singing as well as they did before. Five (7%) had various degrees of dyspnea or stridor on effort. One needed continued dilation. The median follow-up was 8 years. Surgical treatment gave good or excellent results in 90% of these patients with ILTS. Recurrence or progression of stenosis was not evident.

Adolescent↗

Repair of chronic subglottic stenosis with autogenous thyroid cartilage.

Chronic subglottic stenosis continues to be a difficult management problem. Conservative management with bougienage, laser therapy, mitomycin-C, and/or steroid injections frequently fails. Definitive resection of the subglottis with thyrotracheal anastomosis has increased in popularity, but carries significant operative risks and postoperative complications. In the past 9 years, we have used a modification of a technique to successfully widen the subglottis, which we previously published 20 years ago. By a laryngocricotracheofissure approach, the cricoid cartilage is split vertically, both anteriorly and posteriorly. Cartilage grafts, obtained from the upper thyroid laminae anteriorly, are inserted in these splits. The posterior graft is fixed in place with specially designed pins, which improves graft fixation by adding rigidity and shortens the procedure. The repair is held in place for 3 to 4 weeks with either a silicone T tube or a conforming laryngeal stent. The charts of 21 consecutive patients treated with open repair of chronic subglottic stenosis were reviewed. The overall decannulation rate was 95%. We conclude that autogenous grafting is a useful tool in the treatment of chronic subglottic stenosis. Posterior cartilage graft fixation with our pin technique offers a significant advantage over suture fixation by improving graft stability and decreasing operative time, and has a high rate of decannulation.

Adolescent↗

Pediatric airway reconstruction: principles, decision-making, and outcomes at the University of Iowa hospitals and clinics.

Pediatric airway stenosis challenges the treating surgeon, from the initial evaluation and decision-making process to the final postoperative care and follow-up setting. As our approach to these cases evolves, we must critically evaluate our outcomes in reference to our stated goals. We describe our process in treating this population and critically examine our outcome data from 1990 to the present. Emphasis is placed on selection of procedure and postoperative management.

Cartilage↗

Revision single-stage laryngotracheal reconstruction in children.

In this report, we discuss indications, technique, outcome, and complications of revision single-stage laryngotracheal reconstruction (SSLTR), formulate guidelines to avoid or prevent procedure failure, and establish a protocol for the management of procedure failure. We retrospectively reviewed the charts of 122 patients between the ages of 8 months and 9 years who underwent SSLTR between January 1992 and September 2001 in 2 tertiary care children's medical centers in different cities and assessed the outcomes of patients who underwent revision SSLTR. A total of 122 patients underwent SSLTR, of whom 48 patients underwent anterior and posterior grafting. Of the 122 patients, 13 had revision SSLTR; 8 of these 13 underwent the initial laryngotracheal reconstruction at another institution. Five patients had anterior grafting laryngotracheal reconstruction without stenting, 7 had anterior and posterior grafting with 1 to 21 days of endotracheal intubation, and I had cricotracheal resection and anastomosis. Of the 13 patients, 5 had anterior wall or graft collapse (grade IV stenosis), 4 had subglottic stenosis (grade IV), 2 had circumferential subglottic stenosis (grade III), and 2 had subglottic and glottic stenosis (grade IV). The overall success rate for all patients was 86% (105 of 122). The success rates for the 122 patients were as follows: anterior grafting, 100%; anterior and posterior grafting, 83% (40 of 48); and revision cases, 70% (9 of 13). We conclude that laryngotracheal reconstruction with a costal cartilage rib graft should be considered the procedure of choice for the management of subglottic stenosis. We believe that patients in whom the first procedure fails should have a high chance of success with revision SSLTR if strict guidelines and protocols are followed.

Cartilage↗

Transforming growth factor-beta1, myofibroblasts, and tissue remodeling in the pathogenesis of tracheal injury: potential role of gastroesophageal reflux.

Subglottic stenosis (SGS) is characterized by the obliteration of the tracheal lumen due to excessive deposition of connective tissue. We hypothesize that tracheal injury triggers the early production of transforming growth factor-beta1 (TGF-beta1), a factor implicated in fibroproliferative disorders. In turn, TGF-beta1 stimulates the transformation of tracheal fibroblasts into myofibroblasts with increased matrix production and scar contraction that might be influential in laying the foundation for the development of SGS. Consistent with this hypothesis, histologic analysis of tracheas from humans with SGS and from rats with experimental tracheal injury revealed increased alpha-smooth muscle actin-positive cells as compared to control tracheas, suggesting increased myofibroblast differentiation. Rat tracheal fibroblasts exposed to TGF-beta1 or gastric juice in vitro showed increased expression of alpha-smooth muscle actin, alterations in the expression of matrix molecules, and increased contraction of collagen gels. These findings suggest that gastric juice or other agents of tracheal injury promote tissue remodeling through the stimulation of the differentiation of fibroblasts into myofibroblasts.

Actins↗

Cricoid regeneration using in situ tissue engineering in canine larynx for the treatment of subglottic stenosis.

The purpose of the present study was to evaluate the efficacy of cricoid regeneration via in situ tissue engineering in a canine larynx for the treatment of subglottic stenosis. As the tissue scaffold, a Marlex mesh tube coated by collagen sponge was used for a rigid airway framework and for tissue regrowth around the tube. On 5 dogs, the larynx was exposed and the anterior third of the cricoid cartilage was resected. The tube was anastomosed to the lower edge of the thyroid cartilage and to the first tracheal cartilage. By postoperative endoscopic examination at 3 to 7 months, no airway obstruction was observed in any of the dogs. There was granulation tissue in 2 dogs and slight mesh exposure in 1 dog, but they were asymptomatic. Confluent regeneration of the epithelium over the scaffold and good incorporation of the scaffold mesh into the host tissue were observed after surgery.

Animals↗

Primary cricotracheal resection with thyrotracheal anastomosis for the treatment of severe subglottic stenosis in children and adolescents.

Severe subglottic stenosis in children is best managed by laryngotracheal reconstruction or cricotracheal resection (CTR). We describe clinical outcomes with CTR and end-to-end anastomosis in pediatric patients with severe subglottic stenosis in a tertiary-care pediatric teaching hospital in Mexico City. We prospectively followed up all consecutive patients younger than 18 years of age with a Myer-Cotton grade 3 or 4 subglottic stenosis who underwent CTR between May 1, 2000, and March 31, 2003. The frequency of each clinical outcome was calculated. Twenty-two patients (16 boys [72.7%] and 6 girls [27.3%]) were included. The mean age at operation was 4.6 years (range, 11 months to 16 years). Eighteen patients (81.8%) required primary CTR, and 4 (18.2%) required extended CTR. Seventeen (77.3%) had grade 3 stenosis, and 5 (22.7%) had grade 4 stenosis. Six (27.3%) had associated clinical conditions, and 3 (13.6%) had associated vocal cord mobility defects. All were tracheostomy-dependent at presentation, and none had undergone previous airway surgery. Ten (45.5%) underwent one-stage surgery, and 12 (54.5%) had a concomitant temporary tracheotomy. No intraoperative complications occurred. Seventeen patients (77.3%) developed postoperative granulation tissue requiring endoscopic resection. The mean follow-up was 1.2 years (range, 2 months to 2.8 years). No deaths occurred. Fifteen children (88.2%) with grade 3 stenosis and 5 (100%) with grade 4 stenosis were decannulated, for an overall decannulation rate of 90.9%. Partial cricoid resection with end-to-end anastomosis has been a feasible procedure with reproducible successful results among our patients. We conclude that CTR performed as a primary procedure is an effective treatment for the management of severe subglottic stenosis in children.

Adolescent↗

Airway evaluation of conjoined twins.

Case reports in the literature on conjoined twins discuss the difficulties with anesthesia and surgical separation; however, the role of airway endoscopy as a means of evaluating the respiratory tract has not been described. This case of thoraco-omphaloischiopagus laterally conjoined twins demonstrates the importance of videoendoscopic airway evaluation in the management of conjoined twins. Direct laryngoscopy and bronchoscopy was used to evaluate ventilator dependence and demonstrated tracheal anomalies that were partially responsible for difficulties with weaning and endotracheal tube placement. Knowledge of the airway anomalies assisted in ventilator management of the twins, and the neonatalogists were able to proceed with greater confidence because no surgically correctable airway obstruction was found. Direct laryngoscopy and bronchoscopy offer valuable information about thoracopagus conjoined twins and should be included in the preoperative evaluation of planned separation of conjoined twins, as well as being used for conjoined twins who are ventilator-dependent.

Bronchi↗

Use of silicone hearing aid mold material in laryngotracheal reconstruction.

OBJECTIVES: Endolaryngeal stenting in patients with irregularly shaped larynges can be challenging. In such cases, the use of a moldable yet reasonably rigid endolaryngeal stent is desirable. The purpose of this report is to describe our experience with silicone hearing aid material as a moldable endolaryngeal stent in a patient with an atypically shaped larynx. METHODS: A patient with relapsing polychondritis that resulted in complete stenosis of the subglottic airway underwent laryngotracheal reconstruction. Moldable silicone, commonly used to prepare ear canal molds for hearing aids, was molded into the neo-endolarynx to serve as a custom-made endolaryngeal stent. RESULTS: The patient tolerated the moldable silicone stent well and had a patent airway with epithelialization 8 months after removal. CONCLUSIONS: Silicone mold material is a suitable substance for endolaryngeal stenting. It conforms to the configuration of the individual's airway, is tolerated well, can be removed relatively easily, and is a useful alternative to prefabricated stents in laryngotracheal reconstruction.

Adult↗