Endoscopic anterior cricoid split and balloon dilation in pediatric subglottic stenosis.

Abstract

To analyze the outcome of a new endoscopic approach for the treatment of pediatric subglottic stenosis.
Case series.
Tertiary care center.
Eighteen pediatric cases of grade II to IV subglottic stenosis (8 congenital and 10 acquired) consecutively treated at our institutions by Endoscopic Anterior Cricoid Split (EACS) and balloon dilation between 2006 and 2010. Treatment protocol encompassed systematic postoperative laryngeal stenting (7 days of intubation or 1 month of Montgomery T-tube in previously tracheotomized patients) and endoscopic controls with possible additional balloon dilation every 15 days for at least 2 months.
Patients' ages ranged from 1 to 101 months. Postoperative follow-up ranged from 4 to 45 months (median value±SD: 15.3±11.9). The mean duration of the endoscopic procedure was 35.2±13.2 min. The number of days spent in PICU during the perioperative period varied between 2 and 15. Four patients (22.2%) needed one and 14 patients (77.7%) required several (from 4 to 7) additional balloon dilations during the postoperative endoscopic controls. No incident was observed during or immediately after EACS. Treatment was efficient in 83% of cases (n=15), with no residual respiratory symptoms and grade 0 to 1 SGS at the end of follow-up.
EACS is a safe and efficient technique to treat pediatric subglottic stenosis, regardless of their grade and length, provided to associate it with postoperative laryngeal stenting and regular endoscopic follow-up with possible additional balloon dilations. In our teams, it has become the first line treatment for most grades II to IV SGS. Its indications can be extended to congenital stenosis with cartilaginous involvement and to long-lasting acquired stenosis with firm fibrosis.

Full text

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Endoscopic anterior cricoid split and balloon dilation in pediatric
subglottic stenosis
Lorenzo Mirabilea, P. Paola Serioa, R. Roberto Baggia, V. Vincent Couloignerb,*
aPediatric Anesthesiology and Intensive Care Department, Azienda Ospedaliera Anna Meyer, Firenze, Italy
bPediatric ENT Department, Ho ˆpital Necker – Enfants Malades, AP-HP, Paris, France
1. Introduction
Since the 1980s, the classical management of symptomatic SGS
consists of controlling associated risk factors and performing
laryngotracheal surgery via an external approach. Three main
surgical techniques have been proposed: cricoid split, single- or
two-stage laryngotracheal reconstruction, and partial cricotra-
cheal resection. These open airway procedures represented a great
improvement in the management of pediatric laryngeal stenosis.
They proved to be effective in about 90% of cases, limiting the need
for long-term tracheotomy and its associated morbidities such as
cannula obstruction or inadvertent decannulation. Some of them,
such as cricoid split [1] or partial cricotracheal resection [2], can be
successfully proposed during infancy. However, they often require
several surgical procedures, prolonged intensive care admission,
prolonged intubation, or tracheotomy, and they have the potential
for serious complications [2–5]. Their vocal outcome is often not
very satisfactory [6] and they create unappealing cervical scars. In
order to reduce or to avoid some of these drawbacks, several
techniques of endoscopic treatments have been developed during
recent years. In the present study, we present a series of 18 cases of
congenital or acquired SGS treated with anterior cricoid split
(EACS) associated with balloon dilation(s) and postoperative
laryngeal stenting.
2. Material and methods
2.1. Population
Eighteen pediatric cases of SGS (8 congenital and 10 acquired)
consecutively treated at our institution by EACS and balloon
International Journal of Pediatric Otorhinolaryngology 74 (2010) 1409–1414
A R T I C L EI N F O
Article history:
Received 19 July 2010
Received in revised form 14 September 2010
Accepted 19 September 2010
Keywords:
Subglottic stenosis
Laryngotracheal surgery
Suspension microlaryngoscopy
Gastroesophageal reflux disease
A B S T R A C T
Objectives: To analyze the outcome of a new endoscopic approach for the treatment of pediatric
subglottic stenosis.
Study design: Case series.
Setting: Tertiary care center.
Material and methods: Eighteen pediatric cases of grade II to IV subglottic stenosis (8 congenital and 10
acquired) consecutively treated at our institutions by Endoscopic Anterior Cricoid Split (EACS) and
balloon dilation between 2006 and 2010. Treatment protocol encompassed systematic postoperative
laryngeal stenting (7 days of intubation or 1 month of Montgomery T-tube in previously
tracheotomized patients) and endoscopic controls with possible additional balloon dilation every
15 days for at least 2 months.
Results: Patients’ ages ranged from 1 to 101 months. Postoperative follow-up ranged from 4 to 45
months (median value ? SD: 15.3 ? 11.9). The mean duration of the endoscopic procedure was
35.2 ? 13.2 min. The number of days spent in PICU during the perioperative period varied between 2
and 15. Four patients (22.2%) needed one and 14 patients (77.7%) required several (from 4 to 7) additional
balloon dilations during the postoperative endoscopic controls. No incident was observed during or
immediately after EACS. Treatment was efficient in 83% of cases (n = 15), with no residual respiratory
symptoms and grade 0 to 1 SGS at the end of follow-up.
Conclusion: EACS is asafe and efficienttechnique totreat pediatricsubglottic stenosis, regardlessof their
grade and length, provided to associate it with postoperative laryngeal stenting and regular endoscopic
follow-up with possible additional balloon dilations. In our teams, it has become the first line treatment
for most grades II to IV SGS. Its indications can be extended to congenital stenosis with cartilaginous
involvement and to long-lasting acquired stenosis with firm fibrosis.
? 2010 Elsevier Ireland Ltd. All rights reserved.
* Corresponding author at: Service d’ORL Pe ´diatrique, Ho ˆpital Necker – Enfants
Malades, 149, rue de Se `vres, 75015 Paris, France. Tel.: +33 1 44 49 46 82;
fax: +33 1 44 49 46 90.
E-mail address: vincent.couloigner@nck.aphp.fr (V.V. Couloigner).
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology
journal homepage: www.elsevier.com/locate/ijporl
0165-5876/$ – see front matter ? 2010 Elsevier Ireland Ltd. All rights reserved.
doi:10.1016/j.ijporl.2010.09.020

Page 2

dilation between 2006 and 2009 were analyzed. Patients’ clinical
characteristics and endoscopic findings are detailed in Table 1.
Based on endoscopic assessment, stenosis were graded according
to the Cotton–Myer classification [7].
2.2. Inclusion criteria
We performed EACS only if the following conditions were
present:
? Grade II, grade III or grade IV SGS;
? No sign of systemic infection;
? Satisfactory pulmonary function;
? GERD absent or correctly controlled by specific medical or
surgical treatment;
? Absence of associated neurological pathology or swallowing
disorder;
? No history of failed surgical attempts.
2.3. Preoperative assessment
Preoperative evaluation always began with UNA VALUTA-
ZIONE CLINICA FINALIZZATA ALLA VALUTAZIONE DELLA QUA-
LITa ` DELLA VOCE PRIMA DELL’INTERVENTO, CUI SEGUIVA UNA
VALUTAZIONE ENDOSCOPICA DELLA FUNZIONALITa ` LARINGEA.
Dynamic assessment was performed with a fiberoptic endo-
scope (Karl Storz GmbH & Co. KG, Tuttlingen, Germany) under
local anesthesia. Then, a complete airway assessment was done
under general anesthesia using rigid telescopes and broncho-
scopes (KARL STORZ). The diagnosis of congenital stenosis was
based on the absence of risk factors for acquired stenosis and of
severe laryngeal inflammation. When the stenosis was too
narrow to go through it with an endoscope, a CT-imaging of the
airway was done (n = 2; patients 8 and 9). In case of laryngeal or
esophageal inflammation, a 24ph-esophageal probe was per-
formed. If it was positive, medical therapy with proton pump
inhibitors was prescribed for at least 1 month. If the latter
treatment failed, patients underwent Nissen fundoplication
before EACS.
2.4. Description of the procedure and postoperative management
EACS was performed under general anesthesia obtained by
delivering inhalation anesthetics through a nasopharyngeal tube
(n = 9) or through a tracheotomy cannula if the child was
tracheostomized before the procedure (n = 9). Patients with a
nasopharyngeal tube breathed spontaneously, and the airflow
delivered through it equaled three to four times the tidal volume.
This anesthetic technique permitted the surgical field to be free of
any ventilation material. After spraying the vocal folds with 2%
Lidocaine (3–5 mg/kg), the subglottic region was exposed by
suspension microlaryngoscopy. Visualization was improved by
retracting the false vocal folds with a Lindholm laryngeal
distending forceps (#8654B, KARL STORZ GmbH & Co., Tuttlingen,
Germany). A vertical midline anterior incision was made with a
cold knife (#8655, KARL STORZ) through the cricoid anterior ring.
In case of long or high grade stenosis, this incision was extended
superiorly to the infraglottic of the thyroid cartilage, and inferiorly
AIPRIMI2 ANELLITRACHEALItrachealrings. Boththe cartilageand
its external perichondrium were incised (Fig.
incision margins could be laterally displaced a few millimeters
away from each other in a stable way by balloon dilatation. This
dilatation was performed using an angioplasty balloon (CORDIS
CORPORATION, Bridgewater, NJ, USA, or Boston Scientific – France,
Nanterre, France) (Fig. 2). The diameter of the inflated balloon was
approximately 2 mm larger that the normal size of the subglottic
lumen at the age of the patient. The laryngeal dilatation was
maintained until patient’s oxygen saturation dropped under 90%.
At the end of the procedure, in non-tracheotomized patients, a
nasotracheal intubation was performed using a tube with a half-
size larger diameter than the one usually used at the age of the
patient. Patients were kept under sedation and mechanical
ventilation until extubation at day 7. In previously tracheotomized
children, the tracheotomy cannula was replaced by a Montgomery
T-tube in order to stent the subglottic lumen. The T-tube was
removed at day 30, except in two preterm patients (patients 1 and
13) in which it was maintained during 60 days. Post-operative
endoscopic controls were systematically performed at the time of
extubation or at the time of Montgomery tube removal, then once
every 2 weeks during 2 months. Additional endoscopies were
1), so that the
Table 1
Patients’ clinical characteristics and endoscopic findings.
Patient
number
Body weight
(kg)
Age
(months)
Type of stenosisSGS
gradea
Associated laryngeal
anomalies
GERDb(type
of treatment)
Airway stenting
before surgery
1
2
3
4
5
6
7
8
9
1.8
4.3
14.0
8.0
4.1
12.0
5.4
8.1
4.0
22.0
8.1
7
12
9
6
Acquired
Congenital
Congenital
Acquired
Congenital
Acquired
Acquired
Acquired
Acquired
Acquired
Congenital
Congenital
Acquired
III
II
III
II
III
III
II
IV
IV
III
II
III
III
None
None
None
Subglottic hemangioma
None
None
None
None
None
None
None
None
Ectopic gastric and pancreatic
tissue in the left aryepiglottic
fold and false vocal fold
None
None
None
Anterior glottis web
Anterior glottis web
Yes (PPI)
Yes (PPI)
Yes (PPI)
Yes (Nissen)
Yes (Nissen)
No
No
Yes (PPI)
Yes (Nissen)
No
No
No
Yes (Nissen)
Tracheostomy
Endotracheal tube
Tracheostomy
Tracheostomy
Endotracheal tube
None
None
Tracheostomy
Tracheostomy
None
None
Tracheostomy
Tracheostomy
20
9
8
20
8
6
1
10
11
12
13
101
8
4
27
14
15
16
17
18
7
8
4
6 Congenital
Congenital
Acquired
Acquired
Congenital
III
III
II
II
II
Yes (PPI)
Yes (Nissen)
No
Yes (PPI)
Yes (Nissen)
Tracheostomy
Tracheostomy
None
None
None
32
5.3
48
6
16
5
No patient had any previous open air laryngotracheal surgery before undergoing EACS.
aGrading according to Cotton and Myer’s classification.
bGERD: gastro-esophageal reflux disease; PPI: proton pump inhibitors.
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performedasafunctionofpatients’clinicalevolution.Duringthese
endoscopic sessions, balloon dilatations were performed in case of
persistent narrowing of the subglottic lumen.
In the present study, successful treatment was defined as an
open stable adequate airway usually consisting of a grade 0 or 1
SGS, without clinical respiratory compromise.
3. Results
Patients’ follow-up ranged from 4 to 45 months (median
value ? SD: 15.3 ? 11.9). Their clinical characteristics and endoscop-
ic findings are detailed in Table 1. Their age ranged from 1 to 101
months(medianvalue ? SD: 18.1 ? 23.9)andtheirbodyweightfrom
1.8 to 22 kg (median value ? SD: 8.37 ? 5.1). The 10 acquired cases
resulted from intubation trauma. Gastroesophageal reflux was
detected by 24h-pH probe in 12 patients and 6 of them had severe
reflux resistant to medical treatment necessitating a Nissen
fundoplication. At the time of EACS, nine patients were tracheoto-
mized. In four cases, there were some associated laryngeal
comorbidities (Table 1). Patient 4 had a subglottic hemangioma
previously removed by endoscopic diode laser resection (CERALAS
Bare fiber BFF, CERAMOPTEC GmbH, Bonn, Germany). Iatrogenic
symptomatic grade II SGS developed within a few months after this
resection. Patient 13 initially presented with a mass implanted in the
left aryepiglottic fold, false vocal cord and hypopharynx and which
consisted of ectopic gastric and pancreatic tissue. This benign tumor
was endoscopically removed with diode laser. During the postopera-
tiveperiod,bothaSGSandashort(about5 mminlength)butstraight
upper esophageal stenosis were observed. The SGS was treated with
[()TD$FIG]
EACS and balloon dilation whereas the upper esophageal stenosis
required surgical resection of a 25 mm long segment of upper
esophagus. Patients 17 and 18 had both a SGS and an anterior glottic
web. The latter was treated during the EASC procedure with cold
instruments.
The duration of the EACS procedure and characteristics of the
postoperative management are detailed in Table 2. The mean
duration of the endoscopic procedure was 35.2 ? 13.2 min. No
incident was observed during or immediately after EACS. In 83.3% of
patients (15/18), the endotracheal or the Montgomery tubes were
removed according to the planned protocol. The number of days
spent in PICU varied between 2 and 15 (median value ? SD:
7.72 ? 4.19).
During the postoperative endoscopic procedures, four patients
(22.2%) needed one and 14 patients (77.7%) required several
balloon dilations (from 4 to 7) (Table 3). In two patients, topical
applications of mitomycin-C were performed according to the
protocol described in 2001 by Hartnick et al. [8] (mitomycin
concentration: 0.4 mg/ml; application duration: 2 min).
[()TD$FIG]
Fig. 1. Endoscopic view of the section of the subglottic lumen during the anterior midline section of the laryngeal cartilages (A) and just after this section (B).
Fig. 2. Balloon dilation of the subglottic stenosis.
Table 2
Duration of the EACS procedure and postoperative management.
Patient
number
Procedure
duration
(min)
Duration of
hospitalization
in PICUa/total
(days)
Duration of
postoperative
intubation or
tracheostomy
(days)
Number of
laryngeal
endoscopic
dilatations
during the
postoperative
endoscopic
controls
1
2
3
4
5
6
7
8
9
35
27
20
40
22
20
25
38
35
55
38
25
55
65
45
20
30
40
2/9
10/14
2/7
2/7
8/13
9/13
8/11
14/20
15/25
9/15
7/12
7/10
3/15
15/20
3/10
8/10
9/12
8/10
60
7
30
30
7
7
7
7
7
7
7
7
60
12
30
7
7
7
1
6
4
1
7
4
4
4
4
4
0
0
2
3
2
1
1
3
10
11
12
13
14
15
16
17
18
aPICU: pediatric intensive care unit.
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Three EACS failures (17%) were observed (patients 4, 5, and 11).
In patient 4, the supraglottic region was obstructed by granulomas
in the area of contact between the laryngeal mucosa and the upper
extremity of the Montgomey T-tube. These granulomas were
probably caused by the associated GERD, and maybe also by the
laryngealinflammationinducedbythepreviouslaserresectionofa
subglottic hemangioma. This child was found dead by her parents
at home in the morning 18 months after the EACS procedure. The
cause of decease remains unknown. The possibility of a cannula
plugging cannot be discarded.
Patient 5 was not tracheotomized prior to EACS and had no
associated laryngeal comorbidities. He had a severe GERD treated
by Nissen fundoplication. This treatment failed with persistent
hypotonic inferior esophageal sphincter and GERD after surgery.
This might have contributed to EACS failure.
In patient 11, failure was probably due to the fact that after the
EACS procedure, he was followed in another pediatric center and
was not planned for any postoperative endoscopic control. In this
patient, the SGS eventually became more severe than before the
EACS, requiring an open surgical procedure (Table 3).
4. Discussion
As stated in the introduction, the classical treatment of SGS is
open surgery. Endoscopic techniques, in comparison, offer fewer
operative risks, a shorter hospital stay, and possibly less emotional
and financial burden on the family [9,10].
The two main endoscopic techniques described so far are laser
resectionandlaryngealdilation.AccordingtoMonnieretal.[9],laser
resection should be reserved for grade I to mild grade III SGS with a
cranio-caudal extension of less than 15 mm and in the absence of
lossofcartilagesupport(Table4).Itshouldnotberepeatedincaseof
recurrence to the initial grade of stenosis. Triglia et al. [11] advised
laser resection only for grade I stenosis less than 5 mm in length.
Laryngeal dilation can be performed with rigid dilators or with
inflatable balloons. In a recent Brazilian study including 64 cases,
dilatation with rigid Chevalier Jackson dilators was used in grades I
to III stenosis with a success yield of 100% [12]. However, repeated
procedures were necessary, especially for high grade stenosis. For
instance, the mean number of dilations required to treat grade III
stenosis was 12 (Table 4). As compared to the use of rigid dilators,
the main advantages of balloon dilatation are the absence of
longitudinal shearing forces, and the possibility to control both the
balloon diameter and the dilation pressure. In a small series of 10
SGS treated with this technique, the success rate was 100% for the
two grade II stenosis and 62.5% for the eight grade III stenosis [13]
(Table 4). According to the authors, balloon laryngoplasty should
be avoided in cases in which the SGS is long standing, congenital or
has cartilaginous involvement.
The benefit of combining several endoscopic techniques has
been described previously. Thus, in a series of 29 pediatric cases of
SGS, Bakthavachalam and McClay used various combinations of
rigid manual dilation, carbon dioxide laser and microdebridement
[10]. Their percentageof successrangedfrom100% (3/3) for grade I
stenosis to 76% (9/17) for grade III stenosis. However, the authors
state in their discussion that these good results might be
attributable to the fact that they mainly reserved endoscopic
treatment for low grade, short-segment (less than 1.5 cm) stenosis
as well as thin, membranous, granular and spiral ones.
Ascomparedtoallpreviouslydescribedendoscopicprocedures,
the new technique of EACS has several potential advantages:
? By utilizing a full depth incision of the subglottic laryngeal
cartilages and first tracheal rings, the indications can be
extended to congenital stenosis with cartilaginous involvement
and to long-lasting acquired stenosis with firm fibrosis;
? The anterior opening of the larynx and of the first tracheal rings
also enables to obtain major enlargement of the subglottic
lumen, so that EACS can be used in high-grade stenosis (Fig. 3);
? As compared to laser resection, the use of cold instruments
precludes thermal damage.
The comparison between the clinical outcomes obtained with
EACS and with other endoscopic techniques suggests that EACS is
as safe and probably more efficient in high-grade and in congenital
forms of SGS (Table 4). Its efficacy is probably not far from that
obtained with open procedures such as anterior cricoid split or
laryngotracheal reconstruction with cartilaginous grafts [14–19].
The major drawback of EACS is the necessity of post-operative
intubation or 1-month indwelling with a Montgomery T-tube, and
Table 3
Clinical outcome.
Patient numberFollow-up duration (months) Respiratory outcomea
Voice qualityb
General clinical conditions
1
2
3
4
39
27
42
24
Good
Good
Good
Obstructive supraglottic granulomas
impeding decannulation
Residual stenosis requiring reconstruction
laryngotracheoplasty
Patient still tracheotomized
Good
Good
Good
Good
Good
Residual stenosis requiring reconstruction
laryngotracheoplasty
Patient still tracheotomized
Good
Good
Good
Good
Good
Good
Good
Normal
Normal
Normal
Hoarse
Good
Good
Good
Patient deceased 18 months
after EACS
Good
5 22
Soft Voice
6
7
8
9
14
13
10
10
Normal
Normal
Hoarse
Hoarse
Normal
Hoarse
Good
Good
Good
Good
Good
GERD
10
11
7
12
12
13
14
15
16
17
18
2.5
17
4
13
5
7
4
Normal
Normal
Normal
Normal
Normal
Normal
Normal
Good
Good
Good
Good
Good
Good
Good
aDefinition of good respiratory outcome: no dyspnea, and no residual laryngeal stenosis during the last endoscopic control.
bThe voice was assessed clinically and classified as normal, soft or hoarse. In patient 4, the voice quality was tested after brief obstruction of the tracheotomy cannula.
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Table 4
Outcome of endoscopic treatment of pediatric subglottic stenosis. Comparison between literature data and the present series.
ReferenceTechniqueNumber of
patients
Mean age (range)Stenosis
severity
Type of
stenosis
Mean follow-up Mean number
of procedure
Restenosing
Requiring
tracheotomy
or open surgery
Complications
Triglia etal. [11]Laser (CO2, KTP) 25 6 yearsI: 80%
II: 4%
III: 8%
IV: 8%
Not specifiedNot specified1.5 4%Not specified
Monnier etal. [9]a
Radial CO2laser incision
with or without
dilatation with
Savary-Gilliard dilators
10022 years
(6 months–73 years)
I: 15% Acquired 85%18 months48%: n=1 13% of patients
still tracheotomized
at the end of the
study
0
II: 44%
III: 41%
Congenital 7%
Mixed 8%
47% n=2
5% n=3–5
Maksoud-Filho
et al. [12]
Dilatation with Chevalier
Jackson Dilators
64 18.9 months
(1 month–7 years)
I: 70%AcquiredNot specified From 6.3 (grade I)
to 11.9 (grade III)
00
II: 19%
III: 11%
SGS
Bakthavachalam
and McClay [10]
Manual rigid dilation;
CO2laser:
microdebridement
2926.6 months
(0–168 months)
I: 10% Not specified Not specifiedFrom 1.3 (grade I)
to 3.5 (grade III)
12% grade IINot specified
II: 31%
III: 59%
24% grade III
Durden and Sobol [13] Balloon dilatation104.8 months (2–12) II: 20%
III: 80%
Acquired SGS3.5 months1.430%0
Present seriesEndoscopic Anterior
Cricoid SPLIT
11 17.8 months
(1–101 months)
II: 36.4%Acquired and
congenital SGS
16.4 months 3. 918. 2%0
III: 45.4%
IV: 18.2%
aThis series encompassed 45% of adult patients.
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of several planned postoperative endoscopic controls with balloon
dilations. Thus, in short, low-grade and mainly membranous
stenosis, a more straight forward approach such as simple balloon
dilations or laser treatment, might be preferable.
5. Conclusion
This study suggests that EACS is probably a safe and efficient
treatmentofSGS.Inourteam,ithasbecomethefirstlinetreatment
for most grades II to IV SGS, given that the glottic and supraglottic
structures are free of any associated morphological or dynamic
anomaly, and that there is no uncontrolled GERD-induced
inflammation of the larynx. Our remaining indications for open
air procedures are the following cases: (1) laryngeal stenosis that
concern both the glottic and subglottic levels; (2) failures of EACS;
(3) impossibility to correctly expose the larynx under suspension
laryngoscopy. Further prospective studies encompassing higher
numbers of patients are required to confirm these results and to
better specify the indications of this technique.
Conflict of interest
None.
Acknowledgement
We would like to thank Dr Seth Pransky (Pediatric ENT
Department, Rady Children’s Specialist Medical Foundation, San
Diego, USA) for his helpful comments on this manuscript.
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[()TD$FIG]
Fig. 3. Endoscopic view of the laryngeal lumen before (A) and three months (B) after EACS in patient 6.
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