Jurgita Borodičienė¹, Eglė Žukauskaitė², Matas Kalinauskas², Marius Kašėta³, Andrius Macas¹
¹ Department of Anesthesiology, Lithuanian University of Health Sciences, Eiveniu str. 2, LT-50009, Kaunas, Lithuania
² Faculty of Medicine, Academy of Medicine, Lithuanian University of Health Sciences, Eiveniu str. 2, LT-50009 Kaunas, Lithuania
³ Department of Otorhinolaryngology, Lithuanian University of Health Sciences, Eiveniu str. 2, LT-50009, Kaunas, Lithuania
Abstract
Background. Preterm birth is one of the most significant risk factors for infants’ laryngeal stenosis (LS), which can occur as laryngomalacia (LM). LS can be defined as a partial or circumferential narrowing of the endolaryngeal airway and may be congenital or acquired. This condition cause various symptoms such as stridor, dyspnea and respiratory distress, therefore early intubation is required. Most of the patients with LS or LM outgrow their disease, but the rest of them need surgical help.
Objectives. The aim of this article is to present a successful surgical treatment for preterm infant with LS.
Methods: case presentation and an analysis of literature. Research of articles in “PubMed”, “Google Scholar” databases with keywords used as follows: “Laryngeal stenosis”, “Laryngomalacia”, “Pediatric stent”, “Premature delivery”, “Endolaryngeal microsurgery”.
Case presentation. 3-year-old male presented the hospital with diagnosed LM and for laryngeal stent insertion surgery. The patient was born at 26 gestational weeks and was treated for sepsis during the neonatal period, was intubated at birth because of severe respiratory distress, had 6 endolaryngeal microsurgeries (EM) and received Kenalog injections 5 times. Laryngeal stent was inserted during the last EM procedure. Patient undergone general anesthesia, the American Society of Anesthesiologists classification (ASA) was evaluated as III. The operation was successful without any complications. After three months the stent was removed and the patient’s difficulty of breathing regressed.
Conclusion. In respiratory distress caused by LM and LS an insertion of a stent can be as equally or more effective than laryngoplasty. Regression of the respiratory distress symptoms after removal of the stent is considered a successful outcome.
Keywords: laryngeal stenosis, laryngomalacia, pediatric stent, premature delivery, endolaryngeal microsurgery.
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216
Medical Sciences 2021 Vol. 9 (4), p. 216-220
Laryngeal stenosis and laryngomalacia treated with a silicone stent in a
prematurely delivered child: a case report
Jurgita Borodičienė¹, Eglė Žukauskaitė², Matas Kalinauskas², Marius Kašėta³, Andrius Macas¹
¹ Department of Anesthesiology, Lithuanian University of Health Sciences, Eiveniu str. 2, LT-50009, Kaunas,
Lithuania
² Faculty of Medicine, Academy of Medicine, Lithuanian University of Health Sciences, Eiveniu str. 2, LT-50009
Kaunas, Lithuania
³ Department of Otorhinolaryngology, Lithuanian University of Health Sciences, Eiveniu str. 2, LT-50009, Kaunas,
Lithuania
Abstract
Background. Preterm birth is one of the most significant risk factors for infants’ laryngeal stenosis (LS), which can
occur as laryngomalacia (LM). LS can be defined as a partial or circumferential narrowing of the endolaryngeal airway
and may be congenital or acquired. This condition cause various symptoms such as stridor, dyspnea and respiratory
distress, therefore early intubation is required. Most of the patients with LS or LM outgrow their disease, but the rest
of them need surgical help.
Objectives. The aim of this article is to present a successful surgical treatment for preterm infant with LS.
Methods: case presentation and an analysis of literature. Research of articles in “PubMed”, “Google Scholar”
databases with keywords used as follows: “Laryngeal stenosis”, “Laryngomalacia”, “Pediatric stent”, “Premature
delivery”, “Endolaryngeal microsurgery”.
Case presentation. 3-year-old male presented the hospital with diagnosed LM and for laryngeal stent insertion
surgery. The patient was born at 26 gestational weeks and was treated for sepsis during the neonatal period, was
intubated at birth because of severe respiratory distress, had 6 endolaryngeal microsurgeries (EM) and received
Kenalog injections 5 times. Laryngeal stent was inserted during the last EM procedure. Patient undergone general
anesthesia, the American Society of Anesthesiologists classification (ASA) was evaluated as III. The operation was
successful without any complications. After three months the stent was removed and the patient’s difficulty of
breathing regressed.
Conclusion. In respiratory distress caused by LM and LS an insertion of a stent can be as equally or more effective
than laryngoplasty. Regression of the respiratory distress symptoms after removal of the stent is considered a
successful outcome.
Keywords: laryngeal stenosis, laryngomalacia, pediatric stent, premature delivery, endolaryngeal microsurgery.
Journal of Medical Sciences. May 3, 2021 - Volume 9 | Issue 4. Electronic - ISSN: 2345-0592
217
Abbreviations
LS: Laryngeal stenosis
LM: Laryngomalacia
EM: Endolaryngeal microsurgery
ASA: American Society of Anesthesiologists
classification
ENT: Ear, nose and throat
MAC: Minimal alveolar concentration
LTP: Laryngotracheoplasty
Background
Preterm infants often face a variety of organ
developmental failures such as respiratory
abnormalities. One of the most frequent conditions is
laryngomalacia. Respiratory issues caused by
laryngeal abnormalities in neonates may be life-
threatening, therefore we should found the best
treatment to avoid complications [1]. Bronchoscopy is
the best method to diagnose respiratory abnormalities
including LM and LS [2].
Treatment of airway stenosis in pediatric patients is an
indication for stent insertion surgery [3]. Today there
are two types of stents: metallic stent and silicon stent.
It has been proved that treatment of airway stenosis
with silicon stent implantation is as effective as the
metallic stent implantation [2]. The Montgomery
Laryngeal Stent is a molded silicone prosthesis
designed to conform to the normal endolaryngeal
surface. It is used for the prevention and treatment of
LS when the glottic stenosis involves any of glottic
area singularly or in combination. This article reviews
several treatment methods of airway stenosis
encountered during the neonatal period and solutions
in a later age.
Case report
We present a case of a 3-year-old male, who was born
at 26 gestational weeks and was treated for sepsis
during the neonatal period. The patient was quickly
intubated at birth because of severe respiratory
distress, although the mother antenatally received
dexamethasone injections for fetal lung maturity. At
two months of age a bronchoscopy was performed,
because of persisting dyspnea. During the procedure
LM and LS were diagnosed and the patient was
referred to an ear, nose and throat (ENT) surgeon for
tracheostomy. A tracheostomy was performed, no
complications of anesthesia or surgery were noted.
The patient had no complaints after general anesthesia
or operation and was discharged from the hospital one
day after the procedure. At 1 year and 2 months of age
he again was admitted to ENT department, because of
persisting dyspnea. The anesthesiologist evaluated the
patient condition as II ASA. The child then underwent
general anesthesia and was mechanically ventilated
through tracheostomy tube. EM was performed to
remove the granulation tissue from the larynx and a
biopsy was taken of the granulation tissue from the
larynx to specify the changes. Also, the patient
received his first Triamcinolone 40ml (Kenalog)
injection. The biopsy came back as chronic
inflammation with signs of fibrosis. After surgery and
Kenalog injection the patient for a short time was
having no complaints of shortness of breath. However,
three months later dyspnea reoccurred and the boy was
again admitted to ENT department. The patient again
underwent general anesthesia (ASA class was
evaluated as III), EM and Kenalog injection and after
one day at the hospital was discharged with no
complaints. But persistent dyspnea still was present
after some time and after that multiple EM procedures
(6 in total) were done at: 1 year and 11 months, 2 years,
2 years and 1 month and 2 years and 3 months of age.
Multiple Kenalog injections (5 in total) were also
given at: 1 year and 11 months, 2 years, 2 years and 1
month. All times the patient underwent general
anesthesia and was mechanically ventilated through
tracheostomy tube without any complications.
However, because of difficulty of breathing and
persistent dyspnea it was decided that the patient
would undergo one more operation and a laryngeal
stent would be inserted. At the age of three a silicone
Montgomery laryngeal stent was placed during EM
procedure. The patient underwent general anesthesia
(ASA class was evaluated as III), induction reached
with Sevoflurane (Sevoflurane minimal alveolar
concentration (MAC) was kept at 1.0), 25µg of
Fentanyl and 3mg of Mivacurium. The operation was
completed successfully and no complications were
observed. Post operation analgesia was given in form
of Ketoprofen 30mg and Acetaminophen 300mg
intravenously. The patient was discharged after one
day without any dyspnea or difficulty of breathing.
After three months he was admitted to ENT
department for removal of the stent. During that time
there were no complaints of dyspnea or difficulty of
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218
breathing noted. The stent was removed under general
anesthesia without any complications. In three-month
follow-up the patient’s difficulty of breathing has
regressed, no complications of stent extraction were
observed.
Discussion
LM is caused by floppiness of the laryngeal tissues
above the vocal cords (the supraglottic larynx). In
children born with LM the aryepiglottic folds are
shorter than normal so that it resembles the Greek
letter omega (Ω). Studies have identified three types of
LM and each of them have special surgical treatment
[1,4]. During inspiration the tissues above the vocal
cords fall in towards the airway and cause partial
obstruction. This creates stridor for newborns and
infants, which occurs in 45–75% of cases [4]. In most
cases, children with LM outgrow their airway
problems. However, part of them encounter some
difficulties, for example feeding problems,
regurgitation, coughing/choking, failure to thrive, and
significant respiratory distress [4].
LM can be suspected or presumptively diagnosed by
anamnesis and physical exam. It is necessary to know
a full birth history, information about any surgical
procedures or intubations the patient has underwent
[5]. Diagnosis must be confirmed with laryngoscopy
while the child is conscious. The procedure is
preformed using a flexible nasopharyngoscope [1].
Most cases can be treated with conservative methods.
If no severe condition is present and the infant has only
mild or moderate stridor without feeding problems,
after the diagnosis of LM patient must be monitored
for proper development and growth. However, 10 to
20 % of cases with LM are faced with severe
symptoms that result in poor feeding or poor weight
gain, pauses in the breathing (apnea) or cyanosis.
These patients often require surgical treatment [5].
Surgery for this condition is called supraglottoplasty
and it can be performed using several techniques,
counting the use of a laser, cold steel, laryngeal
microdebrider or coblator [6]. In our case during the
bronchoscopy both LM and LS were diagnosed and
the patient was directed for tracheostomy because of
his life-threatening condition.
LS is the second most common cause of respiratory
distress in newborns. It is also the leading
laryngotracheal anomaly that requires tracheostomy in
patients younger than 1 year. Classification of LS can
be provided as congenital or acquired and these can
further be characterized by clinical, anatomical and
histopathological features [7]. Clinically LS depends
on the grade of the obstruction of airway [8]. The
symptoms may present from mild stridor to respiratory
failure: dyspnea, hyperventilation, retractions of
suprasternal and subcostal muscles, apnea, severe
hypoxia, cyanosis. The most crucial risk factor for LS
is preterm delivery [7]. Bronchoscopy is the preferred
procedure to diagnose LS or any congenital airway
malformation [2]. In severe cases of LS tracheostomy
is the first-choice method of symptom relief, but in
some rare cases a stent can be used as a temporary
solution of symptom control for children who have a
high risk of intraoperative fatality and have
contraindications for surgery and general anesthesia
[8-13]. Children who were diagnosed with LS,
whether they had to undergo tracheostomy procedure
or not, should receive systemic steroid injection during
surgery [8]. In our case the patient was born
prematurely at 26 gestational weeks and was quickly
intubated. At two months of age LM with LS was
diagnosed using bronchoscopy and the patient
underwent tracheostomy operation. Kenalog
injections were used alongside EM procedures. The
stent was inserted after failure to control the symptoms
with EM and Kenalog injections.
Airway stents have first been described in the year
1965 by W.W. Montgomery, where he presented his
signature T-Tube silicone stent [14]. In the years to
follow J.F. Dumon introduced his own dedicated
comprised of molded silicone tracheobronchial stent
[15]. There were different variations of silicone and
metallic based stents used throughout the years and in
the late 1990s self-expandable metallic stents were
presented [16]. Although and ideal airway stent is still
not made, there are quite a few diverse choices
available today, but each of them need to be carefully
and individually selected. A few indications for airway
stenting can be found in the literature: palliative care
for patients with advanced cancer; benign airway
stenosis relief; fistula of the respiratory tract therapy;
post lung-transplant anastomosis treatment and
treatment of airway stenosis in pediatric patients [16,
17].
Laryngeal and tracheal stents in pediatric patients are
most commonly used as postoperative tools. Their
main cause is to aid in the healing of surgically
reconstructive tissue or added grafts in the trachea and
larynx, prevent aspiration and keep granulation tissue
formation as low as possible [9, 18]. Furthermore,
stents should not cause any difficulties while being
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219
examined or removed and their care should not
interfere with tracheotomy tube daily cleaning routine
[18]. Stents as the stand-alone treatment of congenital
malformations of the respiratory tract are very rarely
used. Most of them are used after a successful
laryngotracheoplasty (LTP) [9, 10, 19]. Kumar et al.
published a study of 39 pediatric patients with
laryngotracheal stenosis, who underwent airway
stenting with Montgomery T-tube. Out of those
patients 32 were successfully decannulated and after
6-month follow-up are doing well, 3 patients are still
on stenting and 3 patients died to comorbid diseases
unrelated to stenting [10]. In the case described above
a Montgomery laryngeal stent was used molded to
easily adapt to endolaryngeal mucosa and consisting
of radio-opaque silicone. The stent is made atraumatic
and flexible as possible in order to minimize damage
to soft tissue.
Stent insertion can be associated with short and long-
term complications [19, 13]. There are no clear
guidelines for stent management after its placement,
thus every case should be handled individually. The
most commonly reported complications include
granulation tissue formation and trachea obstruction,
stent migration, difficulty of removing the stent and
once in a while erosion of the respiratory tract [13, 16,
19, 20]. A study of 100 pediatric patients, who were
diagnosed with serious airway obstruction received
stent insertion, found that silicone-based stents were
more likely to migrate than metallic stents (39.2% vs
4.1%) and are more liable to granulation tissue
formation (11.6% vs 0.8%). Furthermore, after stent
insertion 80 patients reported to have clinical
improvement, for 17 mechanical ventilation was no
longer needed and 3 had no significant clinical
improvement [21]. There is also a reported case of
tracheal stent erosion, which formed an arterio-
tracheal fistula in an 18 months old female that ended
fatally [20]. A few case reports suggest Pseudomonas
aeruginosa and Staphylococcus aureus colonization
were found on the stent after a successful LTP. The
authors recommended antibiotic prophylaxis that
cover these two microorganisms, their choice was
oxacillin and ciprofloxacin. In both cases the stent was
removed without any adverse effects [19, 22]. Other
complications of stent placement include the
following: stent fracture, airway rupture and halitosis;
all of which are rarely reported, especially in pediatric
patients [17]. Good patient management and follow up
are needed in order to evaluate stents clinical effect.
Recommendations in the literature state that the stent
should be in place for at least 2 – 3 months and it
should eliminate dyspnea or need for mechanical
ventilation. Furthermore, fiber bronchoscopy or
computer tomography scan should be used to confirm
the airway is not obstructed and the stent should be
easily removed by using flexible bronchoscopy [16].
In our reported case there were no complication
reported of stent insertion and removal. The stent was
in place for 3 months and has completely disposed of
any difficulty of breathing or dyspnea. Granulation
tissue formation was a problem before stent
placement, hence the multiple EM.
Conclusion
In conclusion, LM and LS can both be fatally
associated conditions that can cause severe respiratory
distress in a prematurely born child. Although the
primary treatment is LTP, but as an alternative a stent
placement may have outcomes that are as equally or
even more effective. Potential risk factors that need to
be taken into account are: granulation tissue
formation, obstruction of the trachea, migration of the
stent and erosion. In addition, the patient must be
closely monitored during follow-up examination in
order to early detect adverse events. A stent should not
be kept in place longer than 3 months. Regression of
the respiratory distress symptoms after removal of the
stent is considered a successful outcome.
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