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Unilateral vs Bilateral Supraglottoplasty for Severe Laryngomalacia in Children
Deepkaran K. Reddy, MD;
Bruce H. Matt, MD, MS, FAAP
Arch Otolaryngol Head Neck Surg. 2001;127:694-699.
ABSTRACT
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Objectives To study the efficacy of unilateral supraglottoplasty in comparison
with bilateral supraglottoplasty for the treatment of severe laryngomalacia
in children and to study factors that may be predictive of major complications
or the need for a subsequent contralateral or revision procedure.
Design Retrospective medical record review.
Setting University tertiary care pediatric hospital.
Patients One hundred six consecutive pediatric patients, aged 9 days to 18 years,
who had undergone unilateral or bilateral supraglottoplasty for severe laryngomalacia.
Main Outcome Measures Resolution of clinically significant laryngomalacia, development of
major complications (supraglottic stenosis or aspiration), and an association
between study variables (demographics, medical comorbidities, synchronous
airway abnormalities, sites of excision, and techniques of excision) and the
need for subsequent contralateral or revision supraglottoplasty.
Results We achieved a high success rate (95.7%), a low complication rate (8.5%),
and observed the need for a contralateral procedure in 7 (14.9%) of the 47
patients who underwent initial unilateral supraglottoplasty. Two patients
who underwent initial bilateral supraglottoplasty developed supraglottic stenosis.
No significant association existed between our study variables and the development
of complications or the need for contralateral or revision supraglottoplasty.
Conclusions Unilateral supraglottoplasty was associated with a high success rate,
low complication rate, and the avoidance of supraglottic stenosis in our study
population. The percentage of patients requiring a subsequent contralateral
procedure was comparable to that reported in the literature, and no major
complications were associated with the second operation in these patients.
Therefore, unilateral supraglottoplasty seems to be a reasonable option for
initial surgical management of pediatric patients with severe laryngomalacia.
INTRODUCTION
LARYNGOMALACIA is the most common congenital laryngeal anomaly
and the most common cause of stridor in infants. The condition consists of
collapse of any of the major components of the supraglottis, including the
epiglottis, aryepiglottic folds (AEFs), and arytenoid mucosa. This collapse
leads to varying degrees of upper airway obstruction.
The primary clinical manifestation of laryngomalacia is inspiratory
stridor, which can vary in description from the usual high-pitched "hiccuping"
or "squeaking" sound to the less frequently described sounds such as "croaking"
or "crowing."1 This stridor is usually first
noticed at or shortly after birth, increasing over the next several months,
and gradually resolving by 2 years of age, although it may persist into later
childhood. Most cases of laryngomalacia are mild, self-limiting, and usually
unassociated with respiratory distress. Such cases do not require surgical
intervention and are managed expectantly. However, 10% to 15% of the patients
may present with signs of respiratory difficulty and may develop associated
medical problems, including failure to thrive, obstructive sleep apnea, and
cor pulmonale resulting from chronic hypoxia.2
In these severe cases, surgical intervention is required. The most common
procedure performed consists of excision of floppy supraglottic tissues or
supraglottoplasty.
Supraglottoplasty may be unilateral or bilateral; indeed, all of the
earlier reports on the subject described bilateral procedures. Controversy
has recently arisen about the safety of bilateral supraglottoplasty, specifically
with reference to the severe, although rare, risks of postoperative supraglottic
stenosis and aspiration. At our institution, many supraglottoplasties have
been performed over the past 10 years. During the past few years, we have
performed the unilateral procedure in most patients. In our experience, the
incidence of severe complications following either bilateral or unilateral
supraglottoplasty has been low. However, we recently noted that a few patients
who underwent unilateral supraglottoplasty required a contralateral procedure
at a later date, due to persistent laryngomalacia. Owing to concerns regarding
the need for a second operation in these patients, we decided to study the
outcomes of unilateral and bilateral supraglottoplasty in our patient population,
including the efficacy of each procedure in eliminating clinically significant
laryngomalacia, the need for revision supraglottoplasty, and the development
of complications, specifically supraglottic stenosis and aspiration. Ultimately,
we sought to determine if unilateral supraglottoplasty is sufficiently efficacious
and safe to recommend as the initial procedure of choice in the surgical management
of patients with severe laryngomalacia.
MATERIALS AND METHODS
We reviewed the medical records of all patients who had undergone endoscopic
supraglottoplasty at our pediatric tertiary care institution by one of us
(B.H.M.) from July 1, 1989, to September 30, 1997. We studied variables deemed
to have a potential influence on the outcome of supraglottoplasty. These variables
included general demographics (age at time of initial procedure, sex); medical
comorbidities; additional upper airway procedures performed either preoperatively,
postoperatively, or concurrently with supraglottoplasty; areas of excision
of supraglottic tissue; and operative techniques. We also reviewed outcomes,
including the resolution of laryngomalacia; persistent laryngomalacia requiring
either revision or contralateral supraglottoplasty; postoperative supraglottic
stenosis; and postoperative aspiration.
All patients were referred to the Division of Pediatric Otolaryngology–Head
and Neck Surgery, Indiana University School of Medicine, Indianapolis, for
evaluation of stridor associated with various disorders, including obstructive
sleep apnea, failure to thrive, and episodes of apnea or cyanosis. All patients
were diagnosed as having severe laryngomalacia on the basis of flexible and/or
rigid endoscopic findings, as well as the presence of associated disorders,
as verified by the appropriate diagnostic evaluations (eg, polysomnogram,
scintiscan, and others). All patients underwent direct microrigid laryngoscopy
and bronchoscopy at the time of supraglottoplasty to evaluate the airway for
synchronous lesions. Supraglottoplasty was performed using the apneic or spontaneous
respiration technique in most cases, and all procedures were performed with
microscissors or the carbon dioxide laser under suspension microlaryngoscopy.
Postoperatively, the vast majority of patients were extubated and observed
in the intensive care unit for at least 24 hours, and a regimen of antibiotic
agents and antireflux medications was started for at least 2 weeks. This medical
regimen was prophylactic and intended to decrease the risk of supraglottic
infection and edema, which could lead to airway obstruction in either group,
or scarring and supraglottic stenosis in patients with bilateral excision
sites. Postoperative follow-up included fiberoptic laryngoscopy, as well as
diagnostic studies for associated conditions, when indicated.
RESULTS
DEMOGRAPHICS
Data were obtained for a total of 106 consecutive patients. These patients
were grouped into 2 main categories: those initially undergoing bilateral
(group 1) or unilateral (group 2) supraglottoplasty. Group 1 consisted of
59 patients and group 2 consisted of 47 patients. The mean age at the initial
procedure in group 1 was 27.1 months (age range, 2 weeks to 18 years), and
in group 2 was 38.3 months (age range, 9 days to 15 years). Sixty-five patients
were male and 41 female.
MAJOR INDICATIONS FOR SUPRAGLOTTOPLASTY
Although most patients had more than 1 contributing medical diagnosis
or comorbidity, the decision to proceed to supraglottoplasty was based on
1 of 4 major indications (Table 1).
The vast majority of patients had stridor and obstructive symptoms during
sleep, and the diagnosis of obstructive sleep apnea was confirmed by polysomnography
or oxypneumocardiogram. Other major indications included failure to thrive,
episodes of apnea/bradycardia/cyanosis, or cyanosis during feedings.
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Table 1. Major Indications for Supraglottoplasty in 106 Study Subjects
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COMORBIDITIES
Of the comorbidities associated with laryngomalacia, gastroesophageal
reflux was the most common (Table 2).
Others disorders, in decreasing order of frequency, were neuromuscular, respiratory,
and cardiovascular.
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Table 2. Associated Comorbidities
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AREAS OF EXCISION
The supraglottis consists of 3 distinct sites: the epiglottis, AEFs,
and arytenoid mucosa. From these 3 sites, 7 specific combinations of resection
were possible (Table 3). The amount
and location of tissue excised depended on preoperative flexible fiberoptic
laryngoscopy findings, as well as findings during intraoperative evaluation.
Within group 1, 22 patients had excision of all 3 sites, 12 patients had excision
of the epiglottis and AEFs together, and 9 patients had excision of the AEFs
alone. Other combinations were less common. Within group 2, the most common
combinations were AEFs together with arytenoid mucosa (16 patients) and AEFs
alone (13 patients).
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Table 3. Sites of Excision of Supraglottic Tissues
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OPERATIVE TECHNIQUES
Most of the earlier procedures were performed using microscissors, with
carbon dioxide laser excision becoming the preponderant method in more recent
cases. In group 1, 23 patients were treated by microscissor excision and 36
patients by laser. In group 2, only 5 patients were treated by microscissor
excision and 42 patients by laser.
ADDITIONAL UPPER AIRWAY PROCEDURES
Many patients in both groups underwent procedures for treatment of synchronous
upper airway anomalies, preoperatively, postoperatively, or concurrently with
initial or revision contralateral supraglottoplasty. Forty patients in group
1 underwent bilateral supraglottoplasty only; 24 patients in group B underwent
unilateral supraglottoplasty only.
MAIN OUTCOME MEASURES
We defined outcomes in terms of the resolution or persistence of clinically
significant laryngomalacia, the development of severe complications (supraglottic
stenosis and aspiration), the association between study variables and complications,
and between study variables and the need for revision supraglottoplasty. Success
rate was defined by resolution or improvement of laryngomalacia, as evidenced
by follow-up laryngoscopy. Failure was defined by persistent laryngomalacia
requiring tracheostomy or failure to decannulate.
Of the 59 patients in group 1, 55 (93.2%) had clinical resolution of
laryngomalacia after either the initial or the revision procedure. Three patients
required a revision supraglottoplasty for persistent laryngomalacia after
the initial procedure. The revision procedures were unilateral in 2 of these
patients and bilateral in the third, who subsequently required tracheostomy
for persistent laryngomalacia. Another patient underwent tracheostomy concurrently
with supraglottoplasty and was unable to be decannulated owing to persistent
laryngomalacia. Two patients developed supraglottic stenosis that required
surgical intervention. One was treated with initial carbon dioxide laser release
of AEF scarring, followed by tracheostomy for persistent stenosis; and 1 was
treated with tracheostomy alone. Two patients in group 1 developed postoperative
aspiration that was not documented preoperatively. Tracheostomy was performed
for other medical conditions, such as ventilator dependence due to pulmonary
abnormalities, in 9 patients.
Of the 47 patients in group 2, 45 (95.7%) had clinical resolution of
laryngomalacia after either the initial or the contralateral procedure. Seven
patients required a contralateral procedure for persistent laryngomalacia
after the initial unilateral procedure. Six of these patients had resolution
of laryngomalacia following the contralateral procedure, and 1 required tracheostomy
for persistent laryngomalacia. Four patients demonstrated postoperative aspiration
not documented preoperatively, including 1 of the 7 patients requiring contralateral
supraglottoplasty. One of these patients eventually required a tracheostomy
2 months later for "sudden (near) death" episodes. Overall, 2 patients required
tracheostomy related to their laryngomalacia. Four patients required tracheostomy
for other medical conditions.
Patients who developed supraglottic stenosis or aspiration or who required
a revision or contralateral supraglottoplasty were analyzed using the same
study variables as the other patients in groups 1 and 2. Of the 3 patients
who required revision supraglottoplasty following an initial bilateral procedure,
ages at the time of the initial procedure were 1, 7, and 14 months (mean age,
7.3 months). Of the 7 patients who required contralateral supraglottoplasty
following an initial unilateral procedure, the age range was 9 days to 32
months (mean age, 11.4 months). Of the 2 patients who developed supraglottic
stenosis, 1 was 3 weeks of age at the time of the initial procedure and had
a supraglottic laser release at 6 weeks of age for stenosis; this was followed
by tracheostomy at 2 months for persistent stenosis. The other patient was
2 months of age at the time of the initial procedure and had tracheostomy
performed at 3 months of age for stenosis. The average age at the time of
the initial procedure for these 2 patients was 5.5 weeks. Of the 2 patients
who developed aspiration following a bilateral procedure, ages at the time
of supraglottoplasty were 7 weeks and 11 months (mean age, 6.5 months). Of
the 4 patients who developed aspiration following a unilateral procedure,
ages at the time of supraglottoplasty were 3, 15, 18, and 23 months (mean
age, 14.8 months); the youngest of these patients developed aspiration after
undergoing a subsequent contralateral procedure at 3 months of age.
No association was found between any of the variables and the development
of supraglottic stenosis, aspiration or the need for a revision or contralateral
procedure, although patients in this group were younger, in general, than
other patients in groups 1 and 2.
The overall success rate (Table 4) for bilateral supraglottoplasty was 93.2% (55 of the 59 patients)
and for unilateral supraglottoplasty was 95.7% (45 of the 47 patients). Failures
excluded those patients requiring a tracheostomy for associated problems,
such as pulmonary disease requiring ventilatory assistance or tracheomalacia.
None of the patients in our series developed immediate complications such
as airway obstruction, cardiovascular complications, or bleeding. None developed
operative site infection. Overall, the complication rate for group 1, which
included the 2 patients who developed supraglottic stenosis and the 2 with
postoperative aspiration, was 6.8% (4 of the 59 patients). The complication
rate for group 2, which included the 4 patients with postoperative aspiration,
was 8.5% (4 of the 47 patients). The revision rate for bilateral supraglottoplasty
was 5.1% (3 of the 59 patients), and for unilateral supraglottoplasty (contralateral
procedure) was 14.9% (7 of the 47 patients).
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Table 4. Overall Results in 106 Study Subjects
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Success, failure, revision, and complication rates were compared using  2 analysis ( = .05). None of the results were found to be significantly
different between groups 1 and 2. Although patients requiring a revision or
contralateral procedure were notably younger than those not requiring a procedure,
this difference was not found to be statistically significant.
COMMENT
Although laryngomalacia is widely regarded to be the most common cause
of congenital infantile stridor, constituting 60% to 75% of the cases,3 its true incidence is unknown. Indeed, the very definition
of the term is not always uniform.1 Laryngomalacia
is rarely diagnosed in the general population and is more commonly diagnosed
at tertiary care pediatric hospitals. This is probably because of the frequency
of comorbidities that characterize this patient population, as well as an
increased familiarity with the diagnosis among physicians at tertiary care
centers. For example, Belmont and Grundfast4
reported that 80% of the patients had associated gastroesophageal reflux,
23% had neurologic disorders, and 23% had obstructive sleep apnea episodes.
The vast majority of cases of laryngomalacia are considered benign,
with no need for surgical intervention and complete resolution of symptoms
during early childhood, usually by 2 years of age. However, 10% to 15% of
the cases are considered severe enough to require surgical intervention owing
to serious complications such as upper airway obstruction, cor pulmonale,
and failure to thrive.5 Tracheostomy has been
the surgical criterion ("gold") standard for bypassing the obstruction and
allowing unimpeded airflow. However, tracheostomy is not without risks, such
as the development of infection or fatal accidental decannulation. In addition,
tracheostomy care requires an enormous amount of commitment and cooperation
among family members, and also involves a certain financial cost. For these
reasons, alternative procedures have been developed to treat laryngomalacia.
Iglauer, in 1922, was the first to describe partial epiglottectomy for
severe laryngomalacia,1 and several subsequent
studies have proposed various techniques of excision of supraglottic tissues
for this condition.3, 5, 6, 7, 8, 9, 10, 11
The functional value of supraglottoplasty was studied prospectively by Marcus
et al,12 who showed polysomnographic evidence
that obstructive sleep apnea resolved in 4 of 6 patients following supraglottoplasty.
These studies and others described the bilateral excision of supraglottic
tissues, with only a few complications reported in the literature, including
the description of 2 infants who developed supraglottic stenosis by Solomons
and Prescott,9 and a description of interarytenoid
scarring in 1 patient by Jani et al.3 The first
article to systematically analyze results in patients undergoing unilateral
supraglottoplasty was put forth by Kelly and Gray13
in 1995. They had a 94% success rate and no major complications. Three (17%)
of their 18 patients required a contralateral supraglottoplasty at a later
date owing to persistent laryngomalacia.
The obvious disadvantage of a staged contralateral supraglottoplasty
is the need for a second general anesthetic, thereby subjecting the patient
to the usual risks of pediatric airway surgery. This scenario would be ideally
avoided. However, when deciding on unilateral vs bilateral procedures, one
must consider the dire consequences of supraglottic stenosis, which is theoretically
much more likely following a bilateral procedure. If the contralateral supraglottoplasty
rate is not prohibitively higher than the revision or complication rate for
bilateral supraglottoplasty, then the risk of a second airway procedure may
be justifiable. Our contralateral supraglottoplasty rate was comparable to
that in the study by Kelly and Gray.13
Although our overall success rate was high for bilateral supraglottoplasty,
and only 2 patients developed supraglottic stenosis, we have largely abandoned
the bilateral approach because of the severity of this complication in the
few patients in which it occurs. Supraglottic stenosis leads to severe airway
compromise that usually requires tracheostomy. The stenosis results from adhesion
of opposing areas of excision and the resulting scar formation and can occur
even when great care is taken not to excise directly opposing tissue sites
or the interarytenoid mucosa. Because of the recalcitrant nature of supraglottic
stenosis, decannulation in these patients is difficult, and sometimes impossible.
Supraglottic stenosis also leads to impairment of airway protection and an
increased potential for aspiration owing to limited motion as well as decreased
sensation at the level of the supraglottic tissues. Therefore, supraglottoplasty,
whether bilateral or unilateral, should involve excising only the minimal
amount of tissue necessary to improve the airway. This is particularly true
of the epiglottis, which serves a vital function in airway protection. In
addition to the risk of supraglottic stenosis, overexcision of the epiglottis
may result in frank aspiration. In our patients in whom the epiglottis was
included in the supraglottoplasty, only the lateral aspect of the epiglottis
was excised.
Unilateral supraglottoplasty avoids bilaterally opposing excision sites
and, therefore, should not lead to supraglottic stenosis. It is a more conservative
approach with excision of less overall tissue than the bilateral procedure.
A disadvantage of this procedure, therefore, is the potential for excision
of too little tissue to effect resolution of laryngomalacia. However, the
unilateral procedure has been shown to have a high success rate in our study,
and of those patients requiring a contralateral procedure, few complications
have resulted. In addition, the risk of a revision procedure also exists for
bilateral procedures, as evidenced by 3 patients in group 1. Thus, there is
no guarantee of avoiding a second operation with the bilateral procedure.
Our unilateral supraglottoplasty population (group 2) was not without
complications. Four patients developed postoperative aspiration not documented
preoperatively. One of these patients had undergone a contralateral supraglottoplasty;
aspiration in this case could be potentially due to overexcision of tissues.
The other 3 patients had mild aspiration that resolved with medical therapy,
including thickened feedings. Many patients in both groups had aspiration
documented preoperatively but not postoperatively, and some had aspiration
both preoperatively and postoperatively. The possibility remains that some
patients with postoperative aspiration actually had undiagnosed preoperative
aspiration. In any case, only 1 patient in group 2 developed postoperative
aspiration severe enough to warrant a tracheostomy.
None of our study variables identified patients at risk of requiring
a second operation. There was no indication that specific sites of excision
affected the chance of requiring a revision or contralateral procedure. One
would assume that a contralateral or revision procedure would be needed more
often in cases in which too little tissue was initially excised; however,
among the 7 contralateral supraglottoplasties, only 2 involved initial excision
of just 1 supraglottic site (AEFs). There was also no correlation between
the amount of tissue excised and the chance of supraglottic stenosis or aspiration.
Of the 2 patients who developed supraglottic stenosis, one had only the AEFs
excised; the other had the lateral epiglottis and AEFs excised.
Comorbidities also did not predict which patients were more likely to
require revision or contralateral procedures. In fact, no subset of patients
with any combination of comorbidities had an increased rate of revision procedures,
and even in patients who had multiple comorbidities, there were no trends.
Thus, the number and type of comorbid conditions did not predict which patients
would develop complications or would need a revision or contralateral supraglottoplasty.
With regard to surgical technique, all 7 patients requiring a contralateral
supraglottoplasty had undergone laser excision. However, this most likely
reflects the parallel shift from bilateral to unilateral procedures and from
microscissor to carbon dioxide laser excision over time, and probably does
not reflect a cause-and-effect relationship between the two.
Finally, our study population consisted of a large number of patients
who required surgical intervention for laryngomalacia. Although one criticism
of our study may be that the threshold for surgical intervention might have
been lower than usual to accrue such a large series of operative cases, our
analysis indicates that all of the patients in our series did require supraglottoplasty
because of the severity of their condition. All of the patients had signs
of physiologic compromise secondary to laryngomalacia and airway obstruction
as manifested by their primary presenting report and as supported by diagnostic
studies. Most of these patients had already undergone intensive medical or
surgical therapy with persistent laryngomalacia. For example, more than half
of the patients with gastroesophageal reflux required a fundoplication procedure
to control their reflux, with the remainder receiving medical therapy for
at least several weeks. Patients with feeding difficulties and failure to
thrive had been treated with feeding therapy and failed. Finally, patients
with obstructive sleep apnea underwent trials of home monitoring, in some
cases with continous positive airway pressure or oxygen therapy, without success.
By definition, all of our study patients had failed medical therapy. Therefore,
surgical intervention was indeed a last resort in these patients. In addition,
the overall number of patients undergoing supraglottoplasty in our study,
on a per-year basis, compares with the numbers in some other studies.6, 8, 14
A second potential criticism involves the inclusion of patients with
synchronous lesions in our study. Some of these, most commonly hypertrophic
adenoids and tonsils, were untreated until after supraglottoplasty had been
performed. One could argue that the presence of these synchronous lesions
at the time of supraglottoplasty may have contributed to laryngomalacia, and
that treatment of the synchronous lesions may have been tried first. However,
these patients still represented a minority of cases in our study. A larger
subgroup of our study population includes those patients who underwent combined
surgery, including procedures for synchronous lesions at the same time as
supraglottoplasty. Including these patients in the analysis clearly contaminates
the data. Table 5 summarizes our
results if we were to analyze only those patients who underwent supraglottoplasty
alone, without additional procedures at the same time or at revision or contralateral
supraglottoplasty. The results, in terms of resolution of laryngomalacia,
are excellent, as with the overall study population. However, the percentage
of patients requiring a contralateral procedure following unilateral supraglottoplasty
rises to 25%, slightly higher than our overall results. This reinforces the
idea that addressing synchronous lesions at the time of supraglottoplasty,
or prior to supraglottoplasty, may improve the rate of resolution of laryngomalacia,
owing to the additive effect of synchronous lesions on laryngomalacia. Ultimately,
the point remains that patients with laryngomalacia should be carefully selected
for supraglottoplasty, with every effort made to treat associated medical
conditions first, and to address synchronous lesions with surgery specific
to those lesions prior to undertaking supraglottoplasty, when feasible.
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Table 5. Results of Supraglottoplasty Only
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CONCLUSIONS
This study was designed to review our extensive experience with supraglottoplasty
in patients with laryngomalacia and to determine the success, failure, and
complication rates of unilateral and bilateral supraglottoplasty, as well
as to determine any factors that would be predictive of complications or the
need for revision supraglottoplasty for persistent laryngomalacia. We found
that none of the study variables could predict the need for revision supraglottoplasty
or the development of complications. Our overall success rate was high, and
our complication rate was low, for both groups. None of our patients undergoing
a contralateral supraglottoplasty experienced complications attributable to
the second procedure. Therefore, avoiding the severe consequences of supraglottic
stenosis may well be worth the risk of subjecting a patient to a second procedure
following unilateral supraglottoplasty. Based on the results of our study,
we believe that unilateral supraglottoplasty should be the initial procedure
for patients with severe laryngomalacia requiring surgery.
AUTHOR INFORMATION
Accepted for publication January 17, 2001.
Presented at the annual meeting of the American Society of Pediatric
Otolaryngology, Palm Desert, Calif, April 29, 1999.
From the Department of Otolaryngology–Head and Neck Surgery,
Indiana University School of Medicine, Indianapolis. Dr Reddy is now with
the Department of Otolaryngology–Head and Neck Surgery, University of
Michigan School of Medicine, Ann Arbor.
Corresponding author: Deepkaran K. Reddy, MD, Department of Otolaryngology–Head
and Neck Surgery, University of Michigan Medical Center, 1500 E Medical Center
Dr, 1904 TC, Ann Arbor, MI 48109-0312 (e-mail: kdreddy{at}umich.edu).
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