International Journal of Pediatric Otorhinolaryngology

43 (1998) 11 – 20

Gastroesophageal reflux association with

laryngomalacia: a prospective study

Carla Giannoni a, Marcelle Sulek b,*, Ellen M. Friedman b,

Newton O. Duncan III c
a
Department of Otolaryngology Uni6ersity of Florida 1600 SW Archer Road, Rm. M228, Gains6ille,
FL 32610, USA
b
Departments of Otorhinolaryngology and Communicati6e Sciences and Pediatrics,
Baylor College of Medicine, Houston, TX 77030, USA
c
Texas ENT Consultants, 6550 Fannin, Ste. 2001, Houston, TX 77030, USA

Received 25 April 1997; received in revised form 10 October 1997; accepted 12 October 1997

Abstract

Objecti6e: To identify the incidence and clinical role of gastroesophageal reflux (GER) in
patients with laryngomalacia. Design: Prospective evaluation of consecutive infants with a
new diagnosis of laryngomalacia with an initial questionnaire, a barium esophagram or 24 h
pH probe and record of their subsequent clinical course. Setting: A large, tertiary pediatric
referral center and its associated outpatient clinic. Patients: New diagnosis of laryngomalacia
in 33 consecutive infants were evaluated by questionnaire and 27 of these were evaluated for
GER. Results: GER was observed in 64% of patients and was significantly associated with
severe symptoms and complicated clinical course (P= 0.0163). The presence of smokers in
the infant’s household negatively impacted his or her clinical course and symptomatology
(P=0.013) as did the presence of other major, concurrent medical problems (P = 0.065).
Conclusions: In patients with laryngomalacia, GER was significantly associated with severe
symptoms (a complicated clinical course), as was smoking in an infant’s household and other
significant medical problems. © 1998 Elsevier Science Ireland Ltd. All rights reserved.

Keywords: Laryngomalacia; Gastroesophageal reflux; Severe symptoms; Stridor

* Corresponding author. Tel.: +1 713 7703267; fax: + 1 713 7703251.

0165-5876/98/$19.00 © 1998 Elsevier Science Ireland Ltd. All rights reserved.

PII S 0 1 6 5 - 5 8 7 6 ( 9 7 ) 0 0 1 5 1 - 1
12 C. Giannoni et al. / Int. J. Pediatr. Otorhinolaryngol. 43 (1998) 11–20

1. Introduction

‘The infant who appears normal in all other respects is noticed shortly after birth
to have noisy breathing. The noise consists of a croaking sound accompanying
inspiration, which rises to a high pitched crow when a longer or more vigorous
breath is taken. The stridor increases in loudness during the first few months,
gradually lessens and disappears during the course of the second year’. Thomson,
1892 [3].

Congenital anomalies account for 87% of cases of neonatal stridor. Overall,

laryngomalacia is the leading cause, accounting for 60% of cases. Other diagnoses
that should be entertained include: craniofacial anomalies, choanal atresia, en-
cephalocele, turbinate hypertrophy, vocal cord paralysis, congenital or acquired
subglottic stenosis, laryngeal webs, cysts or clefts, papillomatosis, tracheomalacia,
vascular ring, mediastinal masses, foreign bodies and gastroesophageal reflux
(GER) [10].
Although usually a self-limited disorder, serious sequelae of laryngomalacia can
occur. Airway interventions (more frequent hospitalizations and endotracheal intu-
bations) occur in up to 22% of infants and some require surgical intervention,
tracheotomy or laser supraglottoplasty, to improve their airways [7]. Feeding
difficulties, obstructive sleep apnea with bradycardia, failure to thrive, cor pul-
monale and pectus excavatum may result. In addition, a significant number of
infants (12 – 27%) have synchronous airway lesions and half of these are below the
vocal folds [9]. Intellectual compromise has not been statistically associated with
this disorder as was previously thought [1].
Multiple authors have written about the concurrent findings of GER in up to
80% of patients with laryngomalacia [3,4,11,14]. It has been associated with surgical
failure of supraglottoplasty. Despite these inferences that GER may be associated
with laryngomalacia, to date no definitive study has been performed. To identify
the incidence and clinical role of GER in infants with laryngomalacia, we under-
took a prospective evaluation of infants who presented to the Baylor College of
Medicine Pediatric Otolaryngology clinic with a diagnosis of laryngomalacia.

2. Materials and methods

New diagnosis of laryngomalacia in 33 consecutive infants were evaluated by an

entrance questionnaire, examination, pH probe or barium swallow (when permit-
ted) and clinical course (Table 1). The barium esophagram (barium swallow) results
were evaluated by a pediatric radiologist, blinded to the child’s participation in this
clinical study and recorded as: No GER; Borderline GER; (three or fewer episodes
of GER, all below thoracic inlet); or High Grade GER; at or above the thoracic
inlet. The pH probe results were evaluated by a pediatric gastroenterologist, blinded
to the other clinical information about each patient. The score was based on the
 C. Giannoni et al. / Int. J. Pediatr. Otorhinolaryngol. 43 (1998) 11–20 13

Table 1
Questionnaire

Child’s full name:

Today’s date:
Birth weight:
Today’s weight:
1. First episode of noisy breathing (stridor)
No. days after birth:

2. Does the noisy breathing (stridor) occur:

(a) At rest?
Never Occasionally Often/always
(b) During feeding?
Never Occasionally Often/always
(c) When sleeping?
Never Occasionally Often/always
(d) When lying on back?
Never Occasionally Often/always
(e) When lying on stomach?
Never Occasionally Often/always
3. Does your baby ever turn blue?
Never Occasionally Often/always
4. Does your child have a problem with vomiting?
Never Once per day ]Once per feeding
5. How is your baby’s weight gain:
Normal for age Slow Very poor
6. Have you ever seen your child stop breathing?
Never Once per week Every night
7. Has your child had any other medical problems or hospitalizations?
No Yes What kind?
8. Was your baby premature?
No Yes EGA (weeks):
9. Has anyone in your family ever had:
(a) Laryngomalacia, floppy larynx: No Yes Relation: —————
(b) Breathing problems at birth: No Yes Relation: —————
(c) Sudden infant death syndrome: No Yes Relation: —————
10. Are there any smokers in the house?
Yes No

number of GER episodes, the number of episodes greater than 5 min long, and
percentage of time pH less than four and recorded as: None; Borderline; or High
Grade GER for age. Both studies were carried out on six patients and the GER
score was based on the pH probe analysis, unless high grade GER was clearly
demonstrated on an esophagram and then a score for high grade GER was
assigned.
14 C. Giannoni et al. / Int. J. Pediatr. Otorhinolaryngol. 43 (1998) 11–20

Table 2
Severity of laryngomalacia veruses degree of GER

Laryngomalacia Uncomplicated/mild Complicated/severe Totals

Total cases 39.4% (13/33) 61.6% (20/33) 100% (n = 33)

Unable to evaluate GER 3 3 6
Cases tested for GER 10 17 27
GER (n=27)
None 22% (6/27) 11% (3/27) 33% (9/27)
Borderline 7.4% (2/27) 11% (3/27) 18% (5/27)
High grade 7.4% (2/27) 41% (11/27) 48% (13/27) P =0.0163

Infants were also evaluated for complications of their laryngomalacia. A child

was considered to have a complicated clinical course if he or she fulfilled one or
more of the following criteria: (1) moderate to severe airway symptoms necessitat-
ing multiple (greater than or equal to two) emergency center visits or hospitaliza-
tions; (2) multiple episodes (greater than or equal to two) of upper respiratory
infections or pneumonia requiring hospitalization; (3) very poor weight gain or
failure to thrive; or (4) patient required surgical intervention for severe respiratory
symptoms (supraglottoplasty or tracheotomy). These children with a complicated
course are classified as having severe laryngomalacia. Those without these compli-
cating factors are considered mild cases.

3. Results

Evaluation of 33 consecutive children with a new diagnosis of laryngomalacia

was carried out. At first presentation to the otolaryngologist 20 (61%) were males
and 13 were females (39%) with a mean age of 95 days old. The mean onset of
symptoms was 11.8 days. Other medical diagnoses were observed in five patients
(15%). Prematurely (less than 38 weeks gestation), seven (21%) were born. A
smoker lived in eight of the infants households. A sibling of one child had sudden
infant death syndrome. Testing for GER went underway in 27 of these children
(86%), either by 24 h pH probe (n=20) and/or by Barium swallow (n= 13), under
the attending physician’s choice (Table 2).Further testing was refused by four
parents and two children were too ill to undergo testing before a surgical treatment
was initiated. Severe laryngomalacia was detected in 20 of the patients (61%) when
evaluated. Of the 17 patients with severe laryngomalacia who were tested, 11 had
high grade GER, whereas only two of ten patients with mild laryngomalacia had
high grade GER (Fig. 1). To further evaluate their airway anatomy 15 children with
severe, persistent or worsening respiratory symptoms underwent direct laryn-
goscopy and bronchoscopy (DL&B). Of these children nine underwent supraglotto-
plasty for severe symptoms or complications of their laryngomalacia. Only one
child, at 2 weeks of age, failed supraglottoplasty and went on to require a
tracheotomy for severe respiratory symptoms.
 C. Giannoni et al. / Int. J. Pediatr. Otorhinolaryngol. 43 (1998) 11–20 15

One or more other significant medical problems were associated with five
patients: cardiac disorders-VSD (one); pulmonary disorders-bronchopulmonary
dysplasia (two) and asthma (one); central nervous system (CNS) disorders-central
apnea (one), cerebral palsy (two) and unknown cause (one). No second laryngotra-
cheobronchial abnormalities were found by barium esophagram or DL&B in any
infant in this study. All five infants with other medical problems had complicated
or severe laryngomalacia (P = 0.065). The presence of smokers in a child’s home
was also significantly associated with poor clinical condition (P= 0.0009).
Prematurity (P = 0.136), a family history of laryngomalacia (P= 0.131) and low
birth weight (P =0.094) were not significantly associated with complicated laryngo-
malacia, but did show noticeable trends and may become significant in a larger
patient population (Table 3).
No significant differences between the mild versus the severe groups of laryngo-
malacia patients or the gradations of GER groups were seen in regards to: the
estimated gestational age (average= 38.6 weeks, range 22–40 weeks); birth weight
(average 5.48 lbs, range 0.27 – 9.36 lbs); symptom onset (average 11.82 days, range
0–60 days); visit weight (average 9.24 lbs, range 2.38–16.0 lbs); or visit age (average
95.04 days, range 7 – 411 days). Parent reported symptoms (severity at rest, sleeping,
feeding, or in the supine or prone positions, vomiting; apnea and cyanosis) did not
correlate significantly with the incidence of complications or degree of GER.
The gradations of GER were found to be associated with the decisions to
perform DL&B in these patients (P=0.054) (Table 4). Though surgical interven-
tion was more frequent in high grade GER (eight of 13) and borderline GER (two
of five), it was not a statistically significant finding (Table 4). Other medical
problems were found to be associated with high grade GER in four of five patients
(P =0.065). Visual inspection of the data shows trends towards worse apnea,
symptoms in the supine position and lower birth weight in the cases of high grade
GER (Table 5).

Fig. 1. Degree of laryngomalacia versus degree of GER.

16 C. Giannoni et al. / Int. J. Pediatr. Otorhinolaryngol. 43 (1998) 11–20

Table 3
Severity of laryngomalacia versus other patient factors

Laryngomalacia Uncomplicated/mild Complicated/severe Totals (n = 33)

%

Smokers in household
No 39 (13/33) 36 (12/33) 76 (25/33)
Yes 0 24 (8/33) 24 (8/33) P = 0.013
Other medical problems
No 39 (13/33) 46 (15/33) 73 (24/33)
Yes 0 (0/33) 15 (5/33) 27 (9/33) P = 0.065
Fhx laryngomalacia
No 61 (20/33) 15 (5/33) 76 (25/33)
Yes 15 (5/33) 9 (3/33) 24 (8/33) P= 0.130
Prematurity
No 1.36 (12/33) 42 (14/33) 79 (26/33)
Yes 3 (1/33) 18 (6/33) 21 (7/33) P =0.131

Evaluation for differences between laryngomalacia categories were performed

using the Mann-Whitney rank sum test or the Fisher exact test when appropriate.
Evaluation of the differences between the GER groups were evaluated using the
Mann-Whitney test and the Kruskal-Wallis 1-way ANOVA on ranks, each when
appropriate. Statistics were carried out using SPSS for Windows release 6.0.

4. Discussion

The classic presentation of laryngomalacia consists of onset of inspiratory stridor

early after birth which worsens with activity and in the supine position, a normal

Table 4
Degree of GER versus other patient factors

GER None 33% (9/27) Borderline 18% (5/27) High grade 48% (13/27)

DL&B
No 26% (7/27) 7% (2/27) 19% (5/27)
Yes 7% (2/27) 11% (3/27) 30% (8/27) P = 0.054
Surgical intervention
No 30% (8/27) 11% (3/27) 37% (10/27)
Yes 4% (1/27) 7% (2/27) 11% (3/27) P =not significant
Other medical problems
No 33% (9/27) 15% (4/27) 33% (9/27)
Yes 0% (0/27) 4% (1/27) 15% (4/27) P = 0.086
 C. Giannoni et al. / Int. J. Pediatr. Otorhinolaryngol. 43 (1998) 11–20 17

Table 5
Parent-reported symptoms (sx) associated with degree of GER

GER None 33% (9/27) Borderline 18% (5/27) High grade 48% (13/27)

Apnea
Rare 24% (8/33) 9% (3/33) 27% (9/33)
Occasional 3% (1/33) 0% (0/33) 12% (4/33)
Frequent 0% (0/33) 6% (2/33) 0% (0/33)
Sx when supine
Rare 9% (3/33) 3% (1/33) 6% (2/33)
Occasional 15% (5/33) 6% (2/33) 6% (2/33)
Frequent 3% (1/33) 6% (2/33) 27% (9/33)
Birth weight
Average (lbs) 5.87 8.01 4.16
St deviation 3.33 1.79 2.96

cry and may have more difficulty with feeding [5]. It is generally considered to be
a congenital lesion of infants, however a mechanistically similar entity has been
described numerous times in adults following head injury, laryngeal trauma or
surgery and patients with neuromuscular disorders [2,8]. In addition, it has been
described following tonsillectomy and adenoidectomy for obstructive sleep apnea
and it has been reported to be inherited in rare cases in a dominant fashion [6].
The natural history in most patients is for spontaneous recovery; however,
because of frank airway obstruction or other complications 10–15% of babies
require surgical intervention [14]. Tracheotomy has been the standard treatment for
upper airway obstruction for over a century. Supraglottoplasty techniques utilizing
the CO2 laser have been recently popularized for the treatment of this disorder
[11,12,14]. The indications for surgical intervention were patients previously consid-
ered tracheotomy candidates or those who had other severe symptoms of their
airway obstruction, such as subcostal retractions with minimal activity, intermittent
airway distress, oxygen desaturation with crying and failure to thrive secondary to
respiratory compromise.
Stridor results from rapid, turbulent air flow through a narrowed airway. In
laryngomalacia, the major mechanisms of obstruction result from epiglottic and
arytenoid tissue prolapse into the rima glottidis with inspiration, thus, one sees
normal expiration and prolapse of tissues with inspiration [5].
The specific etiology of laryngomalacia is not known, but it is believed to be
either due to a structural anatomic abnormality or due to a neurogenic flaccidity of
the supraglottic larynx [14]. The anatomic theory is supported by histologic exams
which demonstrate normal cartilage in autopsy specimens [3]. Current evidence
suggests that hypoactive neuromuscular control secondary to immaturity, dysfunc-
tion, or incoordination causing flaccidity of the supraglottic structures may also
play a role in the etiology of laryngomalacia [1,14]. Studies show an association of
other neurologic disorders including central apnea, hypothermia and GER in some
of these patients [1,2].
18 C. Giannoni et al. / Int. J. Pediatr. Otorhinolaryngol. 43 (1998) 11–20

Gastroesophageal reflux can cause a variety of gastrointestinal symptoms in-

cluding emesis, dysphagia, choking, and failure to thrive. It has also been well
documented to cause a number of respiratory symptoms: stridor, recurrent croup,
exacerbation of subglottic stenosis, chronic cough, laryngeal irritation, laryn-
gospasm and obstructive apnea [4]. It has even been implicated in sudden infant
death syndrome. Belmont and Grundfast found GER in 80% of their laryngoma-
lacia patients (n = 30) [3]. Polonovski et al. note an ‘‘association of GER with
50% of [their] cases’’ [11]. GER was found by Zalzal to be associated with failure
of supraglottoplasty [14].
In our study, the incidence of complications (severe laryngomalacia) was found
to be significantly associated with the degree of GER (P= 0.0163). In patients
with severe laryngomalacia 65% (11/17) had high grade GER and an additional
18% of them (3/17) had borderline GER; only 18% of patients (3/17) with
complications had no GER at all. Patients with uncomplicated, mild laryngomala-
cia exhibited high grade GER in only 20% of cases (2/10). This does not necessar-
ily imply that GER causes or worsens laryngomalacia, but this association of
GER with complications of laryngomalacia may have important clinical implica-
tions for the evaluation and subsequent management of patients with laryngoma-
lacia.
The observation was interesting that the presence of smokers in the immediate
family was significantly associated with severe laryngomalacia (P= 0.009). Second
hand smoke has been implicated in a number of respiratory disorders, especially
recalcitrant asthma and this is a notable finding [13]. Parents should obviously be
encouraged to quit smoking and informed that smoke in a child’s environment
may worsen the medical condition.
All five patients with significant, other medical problems (cardiac, pulmonary
and CNS) had severe laryngomalacia and four of five had high grade GER
(P = 0.065). Notably, all three patients with CNS disorders had high grade GER
and complications of their laryngomalacia, two of whom required surgical inter-
vention. The rate of CNS disorders in our study patients correlates with findings
in the literature [1]. It appears, as might be expected, that patients with CNS
disorders and other major medical conditions form a small, but significant group
of laryngomalacia patients with a high risk for GER and severe laryngomalacia.
Parent reported symptoms, even vomiting, however, did not correlate signifi-
cantly with the incidence of respiratory complications or gradations of GER.
Historical and physical examinations may not reveal which children have concur-
rent GER which may be compromising their respiratory situation. Prematurity, a
family history of laryngomalacia and low birth weight showed a trend towards an
association with severity of laryngomalacia. A trend between the decision to
perform DL&B and the degree of GER was also noted, suggesting that GER may
worsen or prolong the symptoms of GER in patients with laryngomalacia. There
were also trends between low birth weight, the severity of symptoms in the supine
position and apnea in cases of high grade GER. Larger population studies are
necessary to determine the affect of these factors on a patient’s clinical condition.
 C. Giannoni et al. / Int. J. Pediatr. Otorhinolaryngol. 43 (1998) 11–20 19

Positive identification of these factors may aid in the decision to perform GER
testing.
The large number of patients in our study with complications of their laryn-
gomalacia suggests that many patients with minimally symptomatic laryngoma-
lacia are not referred for formal otolaryngologic examination. Our center is a
large tertiary referral center and a larger proportion of our patients have com-
plicated medical histories and conditions than are seen in the population at
large. None the less, these patients represent the clinically significant sector of
patients, those who exhibit the most severe symptoms and therefore require
intervention more frequently. Because of this, our analysis of the association of
GER and complicated laryngomalacia is most likely representative of the pop-
ulation at large, especially in the group of patients with complicated laryngo-
malacia.
We propose that GER testing be considered in all children with moderate to
severe respiratory symptoms associated with laryngomalacia and those with
other major medical problems. pH probe is a more sensitive test for detecting
GER, but barium esophagogram has the advantage of potentially identifying
vascular rings or other aerodigestive tract abnormalities which may be seen
concurrently with laryngomalacia. Additional evaluation is required to further
evaluate the relationship between laryngomalacia and GER.

References

[1] J. Appley, The infant with stridor: a follow-up survey of 80 cases, Arch. Dis. Child. 28 (1953)
423–435.
[2] S.M. Archer, Acquired flaccid larynx: a case report supporting the neurologic theory of laryn-
gomalacia, Arch. Otolaryngol. Head Neck Surg. 118 (1992) 654 – 657.
[3] J. Belmont, K. Grundfast, Congenital laryngeal stridor (laryngomalacia): etiologic factors and
associated disorder, Ann. Otol. Rhinol. Laryngol. 93 (1984) 430 – 437.
[4] D.M. Burton, S.M. Pransky, R.M. Katz, D.B. Kearns, A.B. Seid, Pediatric airway manifesta-
tions of gastroesophageal reflux, Ann. Otol. Rhinol. Laryngol. 101 (1992) 742 – 749.
[5] R.T. Cotton, Congenital laryngeal anomalies, Otol. Clin. North Am. 14 (1) (1981) 203 – 218.
[6] M.J. Cunningham, C.K. Anonsen, B. Kinane, Acquired laryngomalacia secondary to obstruc-
tive adenotonsillar hypertrophy, Am. J. Otolaryngol. 14 (1993) 132 – 136.
[7] E.M. Friedman, A.P. Vastola, T.J. McGill, G.B. Healy, Chronic pediatric stridor: etiology and
outcome, Laryngoscope 100 (1990) 277 – 280.
[8] C.A. Galliani, B.H. Matt, Laryngomalacia and intra-neural striated muscle in an infant with
the Freeman-Sheldon syndrome, Int. J. Pediatr. Otorhinolaryngol. 25 (1993) 243 – 248.
[9] C. Gonzalaz, J.S. Reilly, C.D. Bluestone, Synchronous airway lesions in infancy, Ann. Otol.
Rhinol. Laryngol. 96 (1987) 77–80.
[10] L.D. Holinger, Etiology of stridor in the neonate, infant and child, Ann. Otol. Rhinol. Laryn-
gol. 89 (1980) 397–400.
[11] J.M. Polonovski, P. Contencin, M. Francois, P. Viala, P. Narcy, Aryepiglottic fold excision
for the treatment of severe laryngomalacia, Ann. Otol. Rhinol. Laryngol. 99 (1990) 625 – 627.
[12] A.B. Seid, S.M. Park, M.J. Kearns, S. Gugenheim, Laser division of the aryepiglottic folds
for severe laryngomalacia, Int. J. Pediatr. Otorhinolaryngol. 10 (1985) 153 – 158.
20 C. Giannoni et al. / Int. J. Pediatr. Otorhinolaryngol. 43 (1998) 11–20

[13] I.B. Tager, S.T. Weiss, A. Munoz, B. Rosner, F.E. Speizer, Longitudinal study of the effects of
maternal smoking on pulmonary function in children, New Engl. J. Med. 309 (1983) 699.
[14] G. Zalzal, J. Anon, R.T. Cotton, Epiglottoplasty for the treatment of laryngomalacia, Ann. Otol.
Rhinol. Laryngol. 96 (1987) 72–76.