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The incidence of postoperative aspiration among children undergoing supraglottoplasty for laryngomalacia.

Abstract

We conducted a retrospective study to determine the incidence of aspiration after supraglottoplasty at Riley Hospital for Children in Indianapolis. We reviewed the charts of 468 patients--281 males and 187females, aged 2 days to 20 years--who had undergone supraglottoplasty for the treatment of laryngomalacia; most patients (69.9%) were aged 28 days to 2 years. A total of 47 patients (10.0%) experienced aspiration after supraglottoplasty; the overall association between supraglottoplasty and aspiration was not statistically significant (p = 0.25). Aspiration was positively correlated with age younger than 18 months, the performance of a revision procedure, the presence of an underlying neuromuscular disorder (n = 20), and the need for a postoperative gastrostomy tube (p < 0.001 for all). When the 20patients with a neuromuscular disorder were excluded from our data analysis, the incidence of aspiration after supraglottoplasty fell to only 5.8% (27/468). We conclude that supraglottoplasty is a safe and effective procedure for the treatment of laryngomalacia. It does not significantly increase the overall incidence of aspiration in children, and thus the risk of aspiration should not be considered a deterrent to surgery, even in children with neuromuscular problems.

Introduction

Laryngomalacia is the most common congenital laryngeal anomaly and the most common cause of stridor in infants. (10) In this population, laryngomalacia leads to failure to thrive, obstructive sleep apnea, awake apneas with bradycardias and cyanosis, and/or cyanosis with feedings. Most cases resolve by the time a child reaches 18 months of age. In severe cases of laryngomalacia, however, surgical treatment with supraglottoplasty is recommended. This procedure includes aryepiglottoplasty, as well as other laryngeal procedures. Supraglottoplasty has been extensively described by other authors. (2-15)

Although supraglottoplasty relieves symptoms of upper airway obstruction, it is not without risk. The most serious complication is supraglottic stenosis. (16) Moreover, supraglottoplasty has been reported to increase the risk of aspiration, owing to limited motion and decreased sensation at the level of the supraglottic tissues. (16) For these reasons, many authors prefer unilateral supraglottoplasty to a bilateral procedure as the initial operation. (17)

Few studies have examined the incidence and management of aspiration after supraglottoplasty, regardless of the technique used. Schroeder et al found that the rate of newly diagnosed aspiration in children who were evaluated by videofluoroscopic swallow studies after bilateral supraglottoplasty was 28%. (16) In a previous study at Riley Hospital for Children, the rate of aspiration was 8.5% after unilateral supraglottoplasty and 3.4% after bilateral supraglottoplasty. (17) We interpreted the lower rate of aspiration after the bilateral procedure to represent a statistical anomaly because very few patients had undergone the bilateral procedure.

In this article, we describe a new study of aspiration after supraglottoplasty at our hospital.

Patients and methods

We retrospectively reviewed the charts of all patients who had undergone supraglottoplasty for the treatment of laryngomalacia in surgeries performed by the senior author (B.H.M.) during a period of 9.5 years at Riley Hospital for Children in Indianapolis. All patients had been referred to the Division of Pediatric Otolaryngology at the Indiana University School of Medicine for evaluation of stridor associated with various disorders, including failure to thrive, obstructive sleep apnea, awake apneas, and/or cyanosis.

All patients had been diagnosed with severe laryngomalacia on the basis of flexible and/or rigid endoscopy, as well as the presence of associated disorders verified by the appropriate diagnostic evaluations (e.g., polysomnography, etc.). These patients also had a myriad of comorbidities, which included synchronous airway abnormalities, congenital syndromes, ear disease or abnormalities, brain abnormalities (including cerebral palsy), gastrointestinal abnormalities and disorders (including gastroesophageal reflux disease), foot and limb deformities, skin lesions, cardiac defects, genitourinary abnormalities, eye abnormalities, neural tube defects, bleeding disorders, endocrine disorders, chromosomal abnormalities, intrauterine exposure to infections and toxins (including cocaine and alcohol), and psychiatric disorders.

As is standard practice at Riley Hospital for Children, all patients who underwent supraglottoplasty for laryngomalacia also underwent microdirect laryngoscopy and bronchoscopy at the time of supraglottoplasty to evaluate the airway for synchronous lesions. (18) If floppiness of the epiglottis or aryepiglottic folds was seen while the patient was breathing spontaneously, trimming of the mucosa of the aryepiglottic fold(s) was performed to allow for increased airflow.

As is also standard practice, the vast majority of patients were extubated, and a regimen of antibiotic agents plus either a proton-pump inhibitor or an H, blocker was prescribed for at least 2 weeks. For patients with suspected postoperative aspiration, videofluoroscopic swallow studies were performed if indicated to document the presence of aspiration and to evaluate the safety of oral feeding.

A total of 2,360 patients had undergone supraglottoplasty during the study period (table 1). After we excluded patients who did not meet our eligibility requirements, our study population was made up of 468 patients. This group included 281 males (60.0%) and 187 females (40.0%), aged 2 days to 20 years. A breakdown of the age distribution follows:

* <28 days: 58 patients (12.4%);

* [greater than or equal to] 28 days to <2 years: 327 patients (69.9%);

* [greater than or equal to] 2 years to <13 years: 75 patients (16.0%); and

* [greater than or equal to] 13 to [less than or equal to] 20 years: 7 patients (1.5%).

A total of 380 patients were younger than 18 months, and 88 were 18 months or older.

In addition to demographic data, we also compiled information on medical comorbidities, areas of excision, surgical technique, and postoperative aspiration symptoms and their treatment. We also examined patients who had required postoperative gastrostomy tube placement to determine if this had been performed because of aspiration or aspiration risk.

We excluded patients who had chronic cough, pneumonia, chronic lung disease, and suspected or documented aspiration before supraglottoplasty. In order to minimize confounding factors, we also excluded patients who had undergone other airway procedures at the same time as the supraglottoplasty.

Approval for our study protocol was obtained from the Institutional Review Board at the Indiana University School of Medicine.

The chi-square test was used to determine statistical significance (SPSS, v. 17.0).

Results

Postoperative aspiration. Postoperative aspiration developed in 47 patients (10.0%). The association between supraglottoplasty and aspiration was not statistically significant (p = 0.25).

Table 2 shows the statistical analysis of the relationship between aspiration and several variables.

Sex. There was no statistically significant difference between males and females in terms of aspiration (p = 0.93).

Age. Of the 47 patients who experienced aspiration, 45 were younger than 18 months (9.6%) and 2 were 18 months or older (0.4%). The difference between the two groups was statistically significant (p = 0.007).

Laterality. Of the 468 patients, most (n = 366 [78.2%]) underwent right-sided supraglottoplasty, since the standard Oxford laryngoscope blade allows for more exposure of the right aryepiglottic fold. Of the remainder, 49 patients (10.5%) underwent left-sided supraglottoplasty and 53(11.3%) had a bilateral procedure. Most of the unilateral supraglottoplasties were performed with a laser, and all of the bilateral procedures were performed with laryngeal scissors.

Postoperative aspiration developed in 38 of the 366 patients who underwent right-sided supraglottoplasty (10.4%), in 7 of the 49 who underwent a left-sided procedure (14.3%), and in 2 of the 53 cases of bilateral surgery (3.8%). There was no significant difference in the incidence of aspiration between the unilateral and bilateral cohorts (p = 0.17).

Revision procedures. A revision procedure had been performed in 27 of the 468 patients (5.8%). Revisions were usually laser-assisted. Postoperative aspiration developed in 4 of these 27 patients (14.8%). The difference in the incidence of aspiration between those who did and did not undergo a revision procedure was not statistically significant (p = 0.61).

Neuromuscular disorders. A preexisting neuromuscular disorder was present in 113 of the 468 patients (24.1%) before surgery; aspiration occurred in 20 of these patients (17.7%). Put another way, of the 47 patients who experienced postoperative aspiration, 20 (42.6%) had a neuromuscular disorder. The correlation between neuromuscular disorders and postoperative aspiration was statistically significant (p < 0.001).

It is interesting that when patients with neuromuscular disorders were excluded from the data analysis of the entire study group, the association between supraglottoplasty and postoperative aspiration fell to only 5.8% (27/468).

Gastrostomy tube placement. A total of 417 patients required gastrostomy tube placement prior to surgery. Of these, 118 patients (28.3%) still required placement postoperatively. The relationship between aspiration and the presence of a gastrostomy tube was statistically significant (p < 0.001). However, it is unclear what the clinical significance of this relationship was, since only 31 of the 118 gastrostomy tube patients (26.3%) had an underlying neuromuscular disorder. Gastrostomy tubes might have been placed more for gastrointestinal issues, which is not something we specifically examined. A total of 84 of the 118 tube patients (71.2%) received their tube postoperatively.

Discussion

In 1922, the first endoscopic surgery for laryngomalacia was reported by Iglauer, who performed an epiglottectomy with a nasal snare to achieve resolution of a child's stridor. (4) In 1985, Seid et al first described the use of a C[O.sub.2] laser in excision of the aryepiglottic folds. (5) Solomons and Prescott described epiglottopexy and partial epiglottectomy to treat variations in supraglottic anatomy that cause supraglottic collapse. (6) These reports and those of others have provided an abundance of research that shows that supraglottoplasty is an effective means of treating laryngomalacia, resulting in a complete regression of symptoms in most patients. (2-17)

Nevertheless, some authors hesitate to recommend supraglottoplasty because of the fear of aspiration. Research has shown, however, that most cases of aspiration resolve in time with conservative measures. Very rarely do patients require feeding tube placement; studies have indicated that only those patients with a history of previous neurologic disorders are likely to require feeding tube placement to prevent aspiration after supraglottoplasty. (3,15-16)

Richter et al showed that cold-knife supraglottoplasty can alleviate laryngeal penetration and aspiration as assessed by pre- and postoperative functional endoscopic evaluation of swallowing, although their study was fairly small (N = 50). (19) They found that supraglottoplasty did not cause aspiration in patients who did not exhibit aspiration preoperatively. However, they also found that supraglottoplasty might not relieve aspiration in patients with multiple medical comorbidities.

The correlation between postsupraglottoplasty aspiration and neurologic disorders may be related to the laryngeal adductor reflex (LAR). The LAR is a sensorimotor reflex that starts with a peripheral afferent laryngeal sensation that travels to the brainstem and stimulates efferent vagal motor responses to cause glottic closure and airway protection during swallowing. Thompson showed that the factors that alter the peripheral and central reflexes of the LAR play a role in the etiology of signs and symptoms of laryngomalacia. (20) This may explain (1) why there are so many children with neuromuscular disorders who also have laryngomalacia and (2) why these children also have a high rate of aspiration after supraglottoplasty. Further research could also examine the role of prematurity in laryngomalacia secondary to the decreased sensorimotor integrative function of the larynx.

Olney et al found that 20% of children with laryngomalacia had severe neurologic compromise and/or multiple congenital anomalies. (8) Smith and Cooper found that upper airway obstruction can be seen years later on volume-loop curves in children who experienced a spontaneous resolution of laryngomalacia, but laryngomalacia will generally resolve within the first 12 to 18 months of life, even in children with multiple congenital anomalies and/or severe neurologic compromise. (21)

Prescott reported that 30% of children with laryngomalacia had a neurologic disorder. (12) Furthermore, Senders and Navarrete showed that children with associated neurologic or anatomic abnormalities have a more complicated course during the immediate postoperative period. (3) The high incidence of associated neuromuscular abnormalities suggests that this component plays an important role in the etiology of laryngomalacia.

One limitation of our study is its retrospective nature. To illustrate, if a patient had developed aspiration postoperatively and received care at another institution, we would not have collected that information. Nevertheless, we believe that such a circumstance is unlikely because children with multiple medical comorbidities eventually require treatment at Riley Hospital for Children, since it is the only comprehensive children's hospital in Indiana. Another limitation is that we did not document the nature of laryngomalacia (aryepiglottic fold collapse vs. epiglottic collapse), which would have been interesting to note.

We believe that the high number of laryngomalacia cases that we treated is attributable to the fact that our hospital is a tertiary care center. Many of our referring physicians are pediatric pulmonologists who send patients to us for surgical evaluation and treatment after medical management has failed.

One might also wonder about the high number of patients in our study who were older than 2 years (n = 82). We found that these patients had not outgrown their laryngomalacia. Again, surgical intervention was indicated after they had not responded to extensive medical management by their referring physician.

Finally, the children who did exhibit postoperative aspiration might have already had aspiration preoperatively, especially those with a preexisting neuromuscular disorder. We do not know how many patients had aspiration prior to surgery since we did not test for it. There also might have been children with silent aspiration that was not recognized. To accurately document the incidence of aspiration after supraglottoplasty, a prospective study with a pre- and postoperative video feeding study would need to be done.

In conclusion, we found supraglottoplasty to be a safe and effective means of treating laryngomalacia. The rate of postoperative aspiration was significantly higher in children who had a neuromuscular disorder (17.7%) but the overall rate was still low (10.0%) and should not be a deterrent to performing this operation, even in patients with a neuromuscular disorder.

Lauren C. Anderson de Moreno, MD; Sarah J. Burgin, MD; Bruce H. Matt, MD, MS

From Natchitoches Medical Specialists, Natchitoches, La. (Dr. Anderson de Moreno); the Department of Otolaryngology-Head and Neck Surgery, University of Michigan School of Medicine, Ann Arbor (Dr. Burgin); and the Department of Otolaryngology-Head and Neck Surgery, Indiana University School of Medicine, Indianapolis (Dr. Matt). The study described in this article was conducted at Riley Hospital for Children in Indianapolis.

Corresponding author: Lauren C. Anderson de Moreno, MD, Natchitoches Medical Specialists, 1029 Keyser Ave., Ste. C, Natchitoches, LA 71457. Email: lauren.moreno@nrmchospital.org

References

(1.) Plant RL, Derkay CS. Physiology of the mouth, pharynx, and esophagus. In: Bluestone CD, Stool SE, Alper CM, et al, eds. Pediatric Otolaryngology. 4th ed. Philadelphia: Saunders; 2001:1103-8.

(2.) Zalzal GH, Collins WO. Microdebrider-assisted supraglottoplasty. Int J Pediatr Otorhinolaryngol 2005;69(3):305-9.

(3.) Senders CW, Navarrete EG. Laser supraglottoplasty for laryngomalacia: Are specific anatomical defects more influential than associated anomalies on outcome? Int J Pediatr Otorhinolaryngol 2001;57(3):235-44.

(4.) Iglauer S. Epiglottidectomy for the relief of congenital laryngeal stridor, with report of a case. Laryngoscope 1922;32(l):56-9.

(5.) Seid AB, Park SM, Kearns MJ, Gugenheim S. Laser division of the aryepiglottic folds for severe laryngomalacia. Int J Pediatr Otorhinolaryngol 1985;10(2):153-8.

(6.) Solomons NB, Prescott CA. Laryngomalacia. A review and the surgical management for severe cases. Int J Pediatr Otorhinolaryngol 1987;13(l):31-9.

(7.) Toynton SC, Saunders MW, Bailey CM. Aryepiglottoplasty for laryngomalacia: 100 consecutive cases. JLaryngolOtol2001;115(1):35-8.

(8.) Olney DR, Greinwald JH Jr., Smith RJ, Bauman NM. Laryngomalacia and its treatment. Laryngoscope 1999;109(11):1770-5.

(9.) Holinger LD, Konior RJ. Surgical management of severe laryngomalacia. Laryngoscope 1989;99(2):136-42.

(10.) Zalzal GH, Anon JB, Cotton RT. Epiglottoplasty for the treatment oflaryngomalacia. Ann Otol Rhinol Laryngol 1987;96(1 Pt l):72-6.

(11.) Roger G, Denoyelle F, Triglia JM, Garabedian EN. Severe laryngomalacia: Surgical indications and results in 115 patients. Laryngoscope 1995;105(10):1111-17.

(12.) Prescott CA. The current status of corrective surgery for laryngomalacia. Am J Otolaryngol 1991;12(4):230-5.

(13.) Jani P, Koltai P, Ochi JW, Bailey CM. Surgical treatment of laryngomalacia. J Laryngol Otol 1991;105(12):1040-5.

(14.) Marcus CL, Crockett DM, Ward SL. Evaluation of epiglottoplasty as treatment for severe laryngomalacia. J Pediatr 1990;117(5):706-10.

(15.) Kelly SM, Gray SD. Unilateral endoscopic supraglottoplasty for severe laryngomalacia. Arch Otolaryngol Head Neck Surg 1995;121(12):1351-4.

(16.) Schroeder JW Jr., Thakkar KH, Poznanovic SA, Holinger LD. Aspiration following C[O.sub.2] laser-assisted supraglottoplasty. Int J Pediatr Otorhinolaryngol 2008;72(7):985-90.

(17.) Reddy DK, Matt BH. Unilateral vs. bilateral supraglottoplasty for severe laryngomalacia in children. Arch Otolaryngol Head Neck Surg 2001;127(6):694-9.

(18.) Mancuso RF, Choi SS, Zalzal GH, Grundfast KM. Laryngomalacia. The search for the second lesion. Arch Otolaryngol Head Neck Surg 1996; 122(3):302-6.

(19.) Richter GT, Wooten CT, Rutter MJ, Thompson DM. Impact of supraglottoplasty on aspiration in severe laryngomalacia. Ann Otol Rhinol Laryngol 2009;118(4):259-66.

(20.) Thompson DM. Abnormal sensorimotor integrative function of the larynx in congenital laryngomalacia: A new theory of etiology. Laryngoscope 2007;117(6 Pt 2 Suppl 114):l-33.

(21.) Smith GJ, Cooper DM. Laryngomalacia and inspiratory obstruction in later childhood. Arch Dis Child 1981;56(5):345-9.
Table 1. Other procedures performed in addition to
supraglottoplasty for laryngomalacia

Procedure                                              n (%) *

Adenotonsillectomy                                   1,879 (79.6)
Adenoidectomy                                         191 (8.1)
Tracheostomy                                          126 (5.3)
Palatoplasty                                           60 (2.5)
Tonsillectomy                                          38 (1.6)
Tracheocutaneous fistula closure                       29 (1.2)
Granulation removal                                    22 (<1)
Pharyngoplasty                                         17 (<1)
Mandibular distraction                                 11 (<1)
Mitomycin application                                  10 (<1)
Choanal atresia repair                                  9 (<1)
Turbinate reduction                                     9 (<1)
Laryngotracheal reconstruction                          7 (<1)
Uvulectomy                                              7 (<1)
Tracheoesophageal fistula repair                        6 (<1)
Tracheal/subglottic dilation                            5 (<1)
Uvulopalatopharyngoplasty                               4 (<1)
Vallecular/epiglottic cyst excision                     4 (<1)
Foreign-body removal                                    3 (<1)
Subglottic/glottic cyst excision                        3 (<1)
Base of tongue reduction                                2 (<1)
Incision and drainage of retropharyngeal abscess        2 (<1)
Lymphangioma excision                                   2 (<1)
Subglottic web excision                                 2 (<1)
Papilloma excision                                      1 (<1)
Repair of airway laceration                             1 (<)

* The 2,360 patients underwent a total of 2,450 procedures;
percentages are based on the total number of patients.

Table 2. Statistical analysis of how selected variables
were associated with postoperative aspiration

Variable                                             p Value

Sex                                                    0.93
Age <18 mo * vs. [greater than or equal to] 18 mo     0.007
Bilateral vs. unilateral procedure                     0.17
Revision procedure or not                              0.61
Neuromuscular disorder * or not                       <0.001
Gastrostomy tube placement * or not                   <0.001

* Aspiration was significantly more common in this subgroup
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Title Annotation:ORIGINAL ARTICLE
Author:de Moreno, Lauren C. Anderson; Burgin, Sarah J.; Matt, Bruce H.
Publication:Ear, Nose and Throat Journal
Article Type:Report
Date:Aug 1, 2015
Words:3076
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