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International Journal of Pediatric Otorhinolaryngology (2008) 72, 1345—1351

www.elsevier.com/locate/ijporl

Epiglottic suture for treatment of laryngomalacia


Igor Fajdiga a,*, Andreja Borinc Beden b, Uroš Krivec b, Črt Iglič a

a
University Department for Otorhinolaryngology and Cervicofacial Surgery,
Zaloška cesta 2, SI 1525 Ljubljana, Slovenia
b
University Children’s Hospital, Department of Pulmology, Vrazov trg 1,
SI 1525 Ljubljana, Slovenia

Received 25 March 2008; received in revised form 18 May 2008; accepted 19 May 2008
Available online 7 July 2008

KEYWORDS Summary
Laryngomalacia;
Objectives: To present a technique for surgical management of laryngomalacia
Classification;
directed against the basic abnormality of the disease. Considering the cause—con-
Stridor;
sequence relations of the abnormalities, we can distinguish two types of laryngoma-
Respiratory system;
lacia. In the first, the basic abnormality is the pathological shape of the epiglottis: the
Abnormalities;
epiglottis, which normally stands in an upright position, is characteristically exces-
Epiglottis;
sively folded, restricting the supraglottic space directly as well as indirectly due to
Suture techniques
the proximity of the aryepiglottic folds that are attached to its lateral edges. In the
second type the abnormality is the backward displacement ( ptosis) of a normally
shaped epiglottis. All other abnormalities are the consequences of these basic
abnormalities. We present a new procedure, the epiglottic suture, to correct the
pathological shape of the epiglottis. It is a suture placed transversely on the lingual
surface of the epiglottis that unfolds the folded epiglottis and shifts apart the
adjacent aryepiglottic folds.
Patients and methods: Prospective non-randomized study performed on eight
severely distressed patients with laryngomalacia at the University Department for
Otorhinolaryngology and Cervicofacial Surgery, Ljubljana, Slovenia.
Results and conclusion: The epiglottic suture enabled normal breathing in all treated
children without compromising the airway-protection function of the epiglottis. After
an average follow up time of 19.12 months (minimum 7 months and maximum 27
months), we have not noticed any complications or deteriorations of breathing.
# 2008 Elsevier Ireland Ltd. All rights reserved.

1. Introduction
Laryngomalacia is the most common cause of stridor
* Corresponding author. Tel.: +386 41662683;
in children. About 50—60% of congenital laryngeal
fax: +386 15224815. anomalies that occur with stridor are due to laryn-
E-mail address: igor.fajdiga@guest.arnes.si (I. Fajdiga). gomalacia. In most cases, the disease follows

0165-5876/$ — see front matter # 2008 Elsevier Ireland Ltd. All rights reserved.
doi:10.1016/j.ijporl.2008.05.009
1346 I. Fajdiga et al.

Table 1 Abnormal anatomical and physiological find- In 2006, the six children treated were selected from
ings in laryngomalacia 15 children registered with laryngomalacia, and in
Tubulated or folded epiglottis 2007, the two treated were chosen from five with
Backward displacement of the epiglottis the same diagnosis. The decision to perform the
Short aryepiglottic folds epiglottic suture in these children was taken in
Excessive mucosa on the aryepiglottic folds cooperation with the Ethics Committee. The
(supraglottis) patients are presented in Table 2.
Bulky arytenoids In all children with typical signs and symptoms of
Inward collapse of the aryepiglottic folds
laryngomalacia, the diagnosis was confirmed with
Inward collapse of the excessive supraglottic mucosa
fiber optic endoscopy during spontaneous breath-
Inward collapse of the epiglottis
ing. The endoscopy revealed that in seven children
the epiglottis was pathologically folded (‘U’ shaped,
a benign course, but the prognosis is less favourable type 1 according to Shah’s classification [4]), while
in 10—15% of cases. These patients may require in one it was tubulated (‘‘C’’ shaped, type 2 accord-
surgical intervention [1]. ing to Shah’s classification [4], Fig. 1, left). All of the
The cause of the disease may result from a varia- epiglottises were standing upright, the aryepiglottic
tion in the anatomy of the supraglottic area, of the folds were short with excessive mucosa, and the
tissue, and/or of the neurological function [2]. arytenoids appeared bulky. In the course of inspira-
Typical findings in severe laryngomalacia are pre- tion, the abundant mucosa of the aryepiglottic folds
sented in Table 1. The leading symptom in any type was fluttering and was sucked into the jar-like space
of laryngomalacia is stridor, which typically between the aryepiglotic folds. This space was in
becomes more pronounced with any kind of activity, fact more or less the only opening available for
emotional agitation, or food intake. In rare cases, inspiration in all the children observed. We esti-
children are exposed to life-threatening situations mated the distance between the epiglottises’ lat-
such as phases of apnea, right-heart failure resulting eral edges (with the insertions of aryepiglotic folds)
from pulmonary hypertension, and a general physi- to be from 2 mm (the tubulated epiglottis with
cal deterioration [2]. overlapping edges) to a maximum of 2 mm in the
Apart from tracheotomy, which cannot be con- least folded epiglottis.
sidered a successful treatment, a number of surgical An epiglottic suture was performed in all the
approaches are described to resolve laryngomala- children. In one child (9 months old with tubulated
cia. Using various tools (laser, microdebrider, clas- epiglottis) the procedure was repeated after 3
sical surgical instruments), most of them are weeks due to recurrent stridor. In this child the
directed against short aryepiglotic folds and excess endotracheal tube was removed immediately after
supraglottic mucosa [3]. both procedures while in the others it was removed
We would like to introduce a new procedure, the after 8 h (one child), 16 h (one child), and 24 h
epiglottic suture that corrects the pathological (three children). In one boy the tube was left for
epiglottis shape, one of the basic problems in lar- 4 days and in one for 6 days due to tracheal infec-
yngomalacia. tions that developed after the procedure. Post-
operative antibiotic prophylaxis was prescribed
for six children (including the two with the infec-
2. Methods tion) while in two cases it was not considered
necessary. Oral feeding with sterile liquid food
In the period from January 2006 to August 2007, the was allowed immediately after the procedure
epiglottic suture was performed at the University (one child, after both procedures) or after the
Department for Otorhinolaryngology and Cervicofa- removal of the tubes (seven children).
cial Surgery, Ljubljana, Slovenia, in eight children Fiber optic endoscopy control examinations were
with laryngomalacia. Prior to the procedure they performed in all children 4—6 weeks after the pro-
were admitted to the University Children’s Hospital cedures (see Section 4).
in Ljubljana, which is a laryngomalacia referral Fig. 1 shows fiber optic view of the epiglottis in
center for the entire country (Slovenia: 2,000,000 laryngomalacia before (left) and immediately after
inhabitants, 9.05 live births per 1000 population and the treatment (right).
fertility rate 1.47 averages for the 2000—2006 per-
iod). The criterion for the epiglottic suture proce- 2.1. Epiglottic suture
dure was laryngomalacia with severe stridor, a
stridor accompanied by suprasternal and sternal The procedure was performed under general endo-
inspiratory retractions while awake and sleeping. tracheal anesthesia using microsurgical instruments.
Epiglottic suture for treatment of laryngomalacia
Table 2 Patients treated with epiglottic suture
Patient’s number
1 2 3 4 5 6 7 8
Gender F F M M F M M M
Gestational 39 40 40 40 40 40 40 39
age (weeks)
Airway obstruction Stridor when Stridor when Stridor when Stridor when Stridor when Stridor when Stridor when Stridor when
awake and awake and awake and awake and awake and awake and awake and awake and
sleeping, life sleeping sleeping sleeping sleeping sleeping sleeping sleeping
threatening
during feeding
Suprasternal and Yes for all Yes Yes Yes Yes Yes Yes Yes
sternal retraction
Oxygen required Yes Yes No No Yes No Yes No
Failure to thrive Yes Yes No No — Yes Yes Yes
Recurrent respiratory Yes No Yes No No No Yes No
infections
Alimentation Aspiration at Aspiration at Occasional No Aspiration No Occasional No
disorders feeding or feeding or aspiration at feeding aspiration
drinking, drinking, at feeding or drinking, at feeding
nasogastric nasogastric or drinking nasogastric or drinking
tube needed tube needed tube needed
Gastroesophageal Confirmed with Confirmed Symptomatic No No No Symptomatic Symptomatic
reflux pH-metry with pH-metry
Diagnosis Symptoms and
fiber optic
endoscopy during
spontaneous
breathing
Age at procedure 6 months 2 weeks 9 months 4 months 3 days 1 month 3 months 2 months
Description of Folded Folded Tubulated Folded Folded Folded Folded Folded
epiglottis shape (‘‘U’’ shape) (‘‘U’’ shape) (‘‘C’’ shape) (‘‘U’’ shape) (‘‘U’’ shape) (‘‘U’’ shape) (‘‘U’’ shape) (‘‘U’’ shape)
Aryepiglottic folds Close and short Close and short Close and Close Close and Close and Close and Close and
with prolapsing with prolapsing short and short short with short short with short with
mucosa mucosa prolapsing prolapsing prolapsing

1347
mucosa mucosa mucosa
1348
Table 2 (Continued )
Patient’s number
1 2 3 4 5 6 7 8

Epiglottis position Upward (normal)


Shah’s classification Type 1 Type 1 Type 2 Type 1 Type 1 Type 1 Type 1 Type 1
Distance between 1 2 2 (overlapping) 3 1 2 2 2
lateral edges
of epiglottis (mm)
Therapeutic procedure Epiglottic suture Epiglottic Epiglottic suture Epiglottic Epiglottic Epiglottic Epiglottic Epiglottic
suture (2 in 3 weeks suture suture suture suture suture
time)
Removal of the After 16 h After 8 h Immediately After 24 h After 4 days After 24 h After 6 days After 24 h
breathing tube after both (respiratory (respiratory
(reason) procedures infection) infection)
Immediate results Effortless breathing, Normal Normal breathing Normal Normal Normal Effortless Normal
normal feeding, breathing and feeding breathing breathing breathing breathing, breathing
occasional breathing and feeding (both procedures) and feeding and feeding and feeding normal and feeding
sound at effort/sleep occasional feeding,
first 3 months breathing sound occasional
at effort breathing
sound
at effort/
sleep first
3 months
Distance between 4 4 2 5 3 4 4 4
epiglottis’ lateral
edges at the control
endoscopy (mm)
Follow—up period 27 26 23 22 19 17 12 7
(months)
Status at last visit Normal breathing, Normal Normal breathing, Normal Normal Normal Normal Normal
feeding, growth breathing, feeding, growth breathing, breathing, breathing, breathing, breathing,

I. Fajdiga et al.
feeding, light stridor feeding, feeding, feeding, feeding, feeding,
growth occasionally growth growth growth growth growth
during effort
Epiglottic suture for treatment of laryngomalacia 1349

Fig. 1 Fiber optic endoscopy view of the epiglottis in laryngomalacia patient before (left) and after the placing of
epiglottic suture (right).

After the epiglottis was exposed by retracting the tying, unfolded it further to the desired shape (Fig. 2,
tongue root with a flat retractor attached to an right).
exterior holder, a narrow saddle-like strip (5 10—
15 mm) of mucosa was removed transversely from
the epiglottic lingual convexity. A resorbable suture 3. Results
(0—4, braided) was then placed over the exposed
site. At both ends the suture was driven through the In all cases, the final effect of the laryngomalacia
lateral edges of excised mucosa and attached to the treatment with the epiglottic suture was smooth
cartilage without touching its laryngeal lining (Fig. 2, breathing and normal feeding. Although in some of
left). As the epiglottis was already spread by the the children a light stridor (without any breathing
inserted endotracheal tube, the tying of the suture effort) was still heard sporadically during physical
fixed the epiglottis in that position or, with tighter exertion or respiratory infection, we consider them

Fig. 2 Left: the placing of epiglottic suture. Right: epiglottic suture tied. Note: epiglottic suture opens up the
obstructed laryngeal entrance; unfolds the excessively folded epiglottis, and spreads apart and lengthens the adjacent
and short aryepiglottic folds.
1350 I. Fajdiga et al.

all to be successfully treated. The average follow up toward the arytenoids, which makes them short
time was 19.12 months (minimum 7 months and and the mucosa on them excessive.
maximum 27 months). In the first group the narrowing lies in a sagittal
plane, while in the second it is transverse. The two
groups could be referred as types 1 and 2 (first
4. Discussion group) and type 3 (second group) according to Shah’s
classification [4] or types 1—3 (first group) and types
In general, an effective therapeutic approach 4 and 6 (second group) according to Holinger [6].
should be directed against the cause of a disease. Understanding laryngomalacia as described, we
As the etiology of laryngomalacia is not clear [5], can conclude that its basic problems are the abnor-
treatment should be directed against the problem mal shape and/or position of the epiglottis while all
that is most responsible for its occurrence. In the other abnormal manifestations are simply their con-
literature, the anatomical abnormalities found with sequences. This conclusion offers a clear direction
laryngomalacia are presented in a descriptive man- for laryngomalacia treatment. In the first group it is
ner (Table 1) with no mention of any possible causal necessary to correct the shape of the epiglottis,
relation between them. Presuming that they are while in the second the epiglottis should be ele-
related, we tried to identify the primary abnorm- vated.
ality that could be the cause of all the others. All our patients were from the first group and
With this cause—effect relationship approach, we therefore the procedure we are introducing applies
can distinguish two groups of laryngomalacia. In the only to this type of abnormality. To correct the
first, the basic abnormality is the pathological shape pathological shape of epiglottis we chose the sim-
of the epiglottis. The epiglottis, which (most often) plest surgical tool, a suture.
stands in a normal upright position, is characteris- The epiglottic suture is a suture placed transver-
tically excessively folded and holds the (probably sely on the lingual surface of the epiglottis. The
normal) aryepiglottic folds that are attached to its tension of the suture spreads the epiglottis, pulls its
lateral edges in close proximity and moves them lateral edges apart and consequently the aryepiglot-
backward against the arytenoids. Consequently, the tic folds away from each other, and opens up the
folds become flaccid, they seem shorter, and the laryngeal entrance for normal breathing. In this new
mucosa on them becomes excessive and makes the (normal) position, the aryepiglottic folds gain ten-
arytenoids appear bulky. With the resulting constric- sion so they become longer and the excess mucosa
tion of the laryngeal entrance, the negative pres- on them stretches to cover the folds normally.
sure of streaming inspiratory air (according to The tension applied on a child’s soft epiglottic
Bernoulli’s principle) can cause further retraction cartilage by the suture is sufficient to remold its
and an inward collapse of the aryepiglottic folds and improper shape. The excision of mucosa prior to
their mucosa as well as of the epiglottis itself in placing the suture is important to prevent the devel-
severe cases. opment of edema and for the formation of the scar
The pathological shapes of the epiglottises in this tissue that preserves the shape of the epiglottis
group of laryngomalacia are difficult to describe for after the resorption of the suture.
a useful classification. They are usually illustrated As the suture is placed only on the lingual side of
by the letters ‘‘U’’ (folded) or ‘‘C’’ and ‘‘V’’ (tubu- the epiglottis, its movements are not impaired and
lated) or as posterior and postero-lateral collapses its airway protection function is not affected. Post-
[4], respectively. operatively there were no signs in the children
However, there is a characteristic feature in all treated of aspiration during feeding or drinking that
these epiglottises that defines the degree of con- could present a postoperative complication.
striction and that can be measured: the distance In seven children the treatment resulted in
between the lateral epiglottic edges (to which the immediate and lasting success. Aside from the occa-
aryepiglottic folds are attached) that determines sional light breathing sound at effort or/and sleeping
the space at the laryngeal entrance confined by the up to 3 months after the procedure in two children,
epiglottis and the aryepiglotic folds. breathing became silent and effortless. Further-
In the second group of laryngomalacia, the more, the accompanying problems——aspiration of
breathing is impaired by the pathological position food and liquids, aspiration pneumonias, alimenta-
of the epiglottis. The epiglottis, which is (most tion disorders, gastroesophageal reflux, and thriving
often) normally shaped, leans backwards ( ptosis) problems——disappeared promptly after the treat-
and closes the laryngeal entrance. Although the ment.
attachments of the aryepiglottic folds in this case In one case, however, it was necessary to repeat
are normally apart, they are shifted backward the procedure. In this case the initial relief from
Epiglottic suture for treatment of laryngomalacia 1351

stridor lasted for 6 days but then the stridor gradu- 5. Conclusion
ally returned over the following 2 weeks. During the
fiber optic endoscopy examination 6 weeks after the The epiglottic suture is a simple and well-tolerated
operation, the epiglottis shape was found to be procedure that relieves stridor in laryngomalacia
similar to that before the procedure. The resorbable patients immediately without compromising the air-
suture had disappeared, and there was a thin scar on way protection function of the epiglottis. No com-
the lingual side of the epiglottis that was evidently plications have been observed in eight treated
not strong enough to preserve its new shape. We children, and no recurrent breathing problems have
repeated the procedure and subsequently the been noticed after 153 months (total for all chil-
breathing became silent and effortless. Although dren) of follow-up.
a light stridor can be still heard occasionally during
physical exertion, we consider the child cured.
The reason for the failure of the first procedure in Appendix A. Supplementary data
the third child was most probably the tubular ‘‘C’’-
shaped epiglottis with overlapping lateral edges.
Supplementary data associated with this article
Compared to the less folded ‘‘U’’-shaped epiglot-
can be found, in the online version, at doi:10.1016/
tises in the other children, it required considerably
j.ijporl.2008.05.009.
stronger traction to successfully maintain the new
shape.
The fiber optic endoscopy control examinations Conflict of interest
performed 4—6 weeks after the procedures (the
second for the third child) confirmed that the arye- All authors declare that no financial or other pot-
piglottic folds were spread apart and straightened. ential conflicts of interest exist. No funds were
The distances between the insertions of aryepiglot- received.
tic folds were increased from 2 mm to 4 mm
(Table 1). Though the increase may not seem impor-
tant, it was sufficient to enable normal breathing in
References
all cases. The scars holding the epiglottis in the new
shape were smooth and confined to its lingual side.
[1] O. Merrot, P. Fayoux, F. Vachin, D. Chevalier, A. Desaulty,
The epiglottic suture is a causal treatment of Severe laryngomalacia: surgical indications and results in
laryngomalacia as it treats one of its basic causes, 33 patients, Ann. Otolaryngol. Chir. Cervicofac. 121 (2004)
the pathological shape of the epiglottis. In this sense 14—21.
it can be compared (but not equated) with epiglot- [2] J.A. Werner, B.M. Lippert, A.A. Dunne, T. Ankermann, B.
Folz, H. Seyberth, Epiglottopexy for the treatment of severe
topexy [2], which corrects a pathological position of
laryngomalacia, Eur. Arch. Otorhinolaryngol. 259 (2002)
the epiglottis, another possible cause of laryngoma- 459—464.
lacia. [3] G.H. Zalzal, W.O. Collins, Microdebrider-assisted supraglot-
The causal approach makes the epiglottic suture toplasty, Int. J. Pediatr. Otorhinolaryngol. 69 (2005) 305—309.
different from established laryngomalacia treat- [4] U.K. Shah, R.F. Wetmore, Laryngomalacia: a proposed clas-
sification form, Int. J. Pediatr. Otorhinolaryngol. 46 (1998)
ments such as the excision of hyperplastic supra-
21—26.
glottic mucosal formations [1,6], epiglottoplasty [5] R.K. Chandra, M.E. Gerber, L.D. Holinger, Histological insight
[6,7], supraglottoplasty [3,6], and supraglottic trim- into the pathogenesis of severe laryngomalacia, Int. J.
ming [2]. These procedures could be described as Pediatr. Otorhinolaryngol. 61 (2001) 31—38.
‘‘symptomatic’’ as they are all directed against the [6] L.D. Holinger, R.J. Konior, Surgical management of severe
laryngomalacia, Laryngoscope 99 (1989) 136—142.
short aryepiglottic folds and their excessive mucosa, [7] G.H. Zazal, J.B. Anon, R.T. Cotton, Epiglottoplasty for the
which in our opinion are clearly the consequences of treatment of laryngomalacia, Ann. Otol. Rhinol. Laryngol. 96
the basic causes for the disease. (1987) 72—76.

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