10.14639/0392-100X-N0830
Objective: Balloon dilation (BD) is a minimally invasive endoscopic treatment for pediatric laryngeal stenosis (LS) with reduced morbidity compared to open surgery. We retrospectively describe our experience in a cohort of children with chronic LS.
Methods: Fourteen children (median age: 28.5; range: 2-81 months) with chronic LS (multilevel in 8) were treated with tubeless total intravenous anesthesia under spontaneous ventilation.
&nbsp;Results: Grade III LS was preoperatively detected in 12 children; the remaining 2 had grade IV stenosis. Six had prior tracheotomy, and one received it during the first intervention. Dilation laryngoplasty was the primary treatment in 11 children and was used as an adjuvant treatment in 3 after open reconstructive surgery. The median number of dilations was 2 (range: 1-6). There were no postoperative complications. At the end of the follow-up (median: 20.5; range: 2-46 months), detectable laryngeal lumen widening and/or respiratory improvement occurred in 12 children. Two out of 7 with tracheostomy were decannulated.
Conclusion: Balloon laryngoplasty is a valuable therapeutic option to improve laryngeal patency in children with chronic multilevel LS, both as a primary and secondary adjuvant treatment after reconstructive surgery.
application/xml
1827-675X
830
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en
Kishore Sandu
https://creativecommons.org/licenses/by-nc-nd/4.0
ACTA Otorhinolaryngologica Italica
children
Outcomes of balloon dilation for paediatric laryngeal stenosis
Text.Serial.Journal
https://www.actaitalica.it/
40
Laryngology
2020-12-03
2020-05-05
2020-12-04
2020-12-04
360-367
1
Kishore Sandu
2020/12/03
10.14639/0392-100X-N0830
360
Airway Sector; Department of Otolaryngology, Lausanne University Hospital CHUV, Switzerland
1827-675X
5
children
367
Outcomes of balloon dilation for paediatric laryngeal stenosis
https://www.actaitalica.it/article/download/830/464
40
ACTA OTORHINOLARYNGOLOGICA ITALICA 2020;40:360-367; doi: 10.14639/0392-100X-N0830




Laryngology


Outcomes of balloon dilation for paediatric laryngeal
stenosis
Il balloon nel trattamento delle stenosi laringee in età pediatrica
Giovanna Cantarella1,2, Michele Gaffuri2, Sara Torretta1,2, Simona Neri3, Maria Teresa Ambrosini3,
Alessandra D’Onghia1,2, Lorenzo Pignataro1,2, Kishore Sandu4
1
  Fondazione IRCCS Ca’ Granda Ospedale Maggiore Policlinico, Department of Otolaryngology and Head and Neck Surgery, Milan,
Italy; 2 Department of Clinical Sciences and Community Health, University of Milan, Milan, Italy; 3 Unit of Pediatric Anesthesiology
and Intensive Care, Fondazione IRCCS Ca’ Granda Ospedale Maggiore Policlinico, Milan, Italy; 4 Airway Sector; Department of
Otolaryngology, Lausanne University Hospital CHUV, Switzerland


SUMMARY
Objective. Balloon dilation (BD) is a minimally invasive endoscopic treatment for paediat-
ric laryngeal stenosis (LS) with reduced morbidity compared to open surgery. We retrospec-
tively describe our experience in a cohort of children with chronic LS.
Methods. Fourteen children (median age: 28.5; range: 2-81 months) with chronic LS (mul-
tilevel in 8) were treated with tubeless total intravenous anaesthesia under spontaneous
ventilation.
Results. Grade III LS was preoperatively detected in 12 children; the remaining 2 had
grade IV stenosis. Six had prior tracheotomy, and one received it during the first interven-
tion. Dilation laryngoplasty was the primary treatment in 11 children and was used as an
                                                                                                Received: May 5, 2020
adjuvant treatment in 3 after open reconstructive surgery. The median number of dilations
                                                                                                Accepted: July 28, 2020
was 2 (range: 1-6). There were no postoperative complications. At the end of the follow-up
(median: 20.5; range: 2-46 months), detectable laryngeal lumen widening and/or respira-
                                                                                                Correspondence
tory improvement occurred in 12 children. Two of 7 patients with tracheostomy were de-
                                                                                                Sara Torretta
cannulated.                                                                                     Department of Clinical Sciences and Community
Conclusions. Balloon laryngoplasty is a valuable therapeutic option to improve laryngeal        Health, Università degli Studi di Milano, Fondazio-
patency in children with chronic multilevel LS, both as a primary and secondary adjuvant        ne IRCCS Cà Granda Ospedale Maggiore Policlini-
treatment after reconstructive surgery.                                                         co, Via F. Sforza 35, 20122 Milano, Italy
                                                                                                Tel. +39 02 50320245. Fax +39 02 50320248
KEY WORDS: balloon dilation, paediatric, laryngeal stenosis, dyspnea, children                  E-mail: sara.torretta@unimi.it

RIASSUNTO                                                                                       Funding
Obiettivi. Descrivere retrospettivamente la nostra esperienza con dilatazione laringea con      None.
balloon (DB) per il trattamento della stenosi laringea (SL) cronica in età pediatrica.
Metodi. Quattordici bambini (età mediana: 28,5; range: 2-81 mesi) con SL cronica (multi-        Conflict of interest
livello in 8 casi) sono stati sottoposti a DB in ventilazione spontanea in assenza di intuba-   The Authors declare no conflict of interest.
zione orotracheale con anestesia endovenosa esclusiva.
Risultati. Dodici bambini presentavano pre-operatoriamente una SL di grado III, i rima-           How to cite this article: Cantarella G, Gaffuri
nenti una SL di grado IV. Sei pazienti erano portatori di tracheotomia mentre un paziente         M, Torretta S, et al. Outcomes of balloon dilation
è stato sottoposto a tracheotomia durante la prima DB. La DB è stata eseguita come trat-          for paediatric laryngeal stenosis. Acta Otorhi-
tamento primario in 11 bambini e come adiuvante a chirurgia ricostruttiva in 3 casi. Il           nolaryngol Ital 2020;40:360-367. https://doi.
numero mediano di BD eseguite è 2 (range: 1-6). Non si sono verificate complicanze post-          org/10.14639/0392-100X-N0830
operatorie e, al termine del follow-up (mediana: 20,5; range: 2-46 mesi), in 12 bambini è
stato obiettivato un ampliamento del lume laringeo e/o un miglioramento respiratorio. Due       © Società Italiana di Otorinolaringoiatria
dei 7 pazienti con tracheotomia sono stati decannulati con successo.                            e Chirurgia Cervico-Facciale
Conclusioni. La DB risulta efficace e sicura per migliorare in bambini con SL cronica
multi-livello sia come trattamento primario che come trattamento adiuvante.                                    OPEN ACCESS

                                                                                                This is an open access article distributed in accordance with
PAROLE CHIAVE: dilatazione con balloon, pediatria, stenosi laringea, dispnea, bambini           the CC-BY-NC-ND (Creative Commons Attribution-Non-
                                                                                                Commercial-NoDerivatives 4.0 International) license. The
                                                                                                article can be used by giving appropriate credit and mentio-
                                                                                                ning the license, but only for non-commercial purposes and
                                                                                                only in the original version. For further information: https://
                                                                                                creativecommons.org/licenses/by-nc-nd/4.0/deed.en




360
                                                                                        Balloon dilation for paediatric laryngeal stenosis




Introduction                                                      Study subjects
                                                                  The study involved the charts of children who had been
Laryngeal stenosis (LS) in neonatal and paediatric age
                                                                  treated with BD for chronic LS between February 2016 and
groups is a potentially life-threatening condition and has a
                                                                  October 2019. All patients underwent endoscopic treatment
severe impact on the quality of life of children and families.
                                                                  and subsequent postoperative follow-up in our tertiary out-
It can be congenital or acquired, but the latter is most fre-
                                                                  patient clinic. The authors GC, MG and LP performed all
quent, mainly due to prolonged endotracheal intubation in
                                                                  the procedures with collaborative guidance of KS. All pa-
neonatal intensive care for severe prematurity 1,2. The most
                                                                  tients underwent transnasal flexible high-definition video
commonly involved sites in LS are the subglottis and the
                                                                  laryngoscopies on their initial presentation and during fol-
posterior glottis, as the cricoid, the medial aspects of the
                                                                  low-up visits.
arytenoid cartilage and the posterior commissure are the
                                                                  The diagnosis of LS was confirmed by direct laryngotra-
areas in the airway with a major risk of developing intuba-
                                                                  cheoscopy under general anaesthesia, and patients breathed
tion injuries 1.
                                                                  spontaneously. The degree of LS before and after BD was
Three main types of procedures have been proposed to treat
                                                                  scored according to the Myer-Cotton grading scale 6.
established cicatricial LS: expansion of the airway with
                                                                  LS was defined as chronic if symptoms were present for
costal cartilage, resection of the stenotic part of the airway
                                                                  more than 3 months.
and dilation by means of rigid dilators or by inflatable bal-
loons. Balloon dilation (BD) has been proposed since 1984
                                                                  Interventions
to treat tracheal and bronchial stenoses and is appropri-
                                                                  The retrospective chart review included demographic data
ate only if the cartilaginous skeleton of the airway is pre-
                                                                  and clinical histories. The clinical histories included all
served. A balloon is used under direct endoscopic vision
                                                                  possible conditions predisposing to the development of
and allows the application of radial pressure on the airway
                                                                  LS, including prolonged intubation, intensive-care unit
stricture, thus theoretically reducing the risk of shearing the
                                                                  stay, prematurity, syndromic disease or defined genetic ab-
mucosa with consequent possible restenosis.
                                                                  normalities, previous cardiac surgery and previous airway
Recently, there has been an increase in the number of re-
ports highlighting the important role of balloon laryn-           surgery. History of respiratory distress, stridor, voice im-
goplasty in avoiding tracheotomy in acute and subacute            pairment, failure to thrive, or swallowing impairment were
airway obstruction and in improving airway patency in im-         recorded.
mature or mature scarring with limited extension and thick-       In the children without tracheotomy, BD was performed
ness 4,5. The procedure is minimally invasive and can avoid       under tubeless total intravenous anaesthesia (TIVA) with
the intra- and postoperative risks of major reconstructive        spontaneous ventilation and supplemental oxygen adminis-
surgery.                                                          tration dispensed by an uncuffed silicone endotracheal tube
This paper aims to retrospectively describe our experience        placed through the nasal fossa and left in the hypopharynx
with BD in a cohort of children affected by LS. The pe-           with its bevel facing the airway. A second similar uncuffed
culiarity of the described series is related to the extension     tube was prepared on a 2.7 mm Storz (Germany) rigid 0°
of stenosis - multilevel in most cases - and to the chronic       endoscope ready to be used in case of the need for emer-
nature of the lesion, as the majority of patients were symp-      gency intubation to allow rapid introduction under direct
tomatic for several months or years.                              endoscopic vision. The anaesthetic technique was stand-
                                                                  ardised as reported in Table I. Heart rate, blood pressure,
                                                                  pulse oximetry, Bispectral Index (BIS) level, transcutane-
Materials and methods                                             ous CO2 (TcCO2), respiratory rate and temperature were
                                                                  recorded intraoperatively.
Study design and setting                                          Lidocaine was sprayed into the larynx under direct laryn-
This study is a retrospective chart review of prospectively       goscopic vision at a dose of 2-4 mg/kg. Then, an appropri-
recruited children with LS, and it was carried out at the         ately sized Lindholm or Parson laryngoscope was placed
Otolaryngology Division of Milan University’s Depart-             to expose the larynx under suspension. First, a 0° rigid
ment of Clinical Sciences and Community Health in De-             2.7- or 4-mm telescope was introduced to examine the vo-
cember 2019.                                                      cal cords, anterior and posterior commissures, subglottis,
The protocol was approved by our local Ethics Commit-             trachea, carina and mainstem bronchi. Once the stenosis
tee of Fondazione IRCCS Ca’ Granda Ospedale Maggiore              was assessed, triamcinolone acetate (KenacortR 40 mg/ml,
Policlinico and was conducted in accordance with the prin-        Bristol-Myers Squibb s.r.l.) was injected into the stenotic
ciples of good clinical practice.                                 and scarred areas. If concentric stenosis was found, 3-4 ra-



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G. Cantarella et al.




Table I. Description of the anaesthetic protocol for balloon dilation.                         Superpulse). Next, an appropriately selected balloon was
 Anaesthetic procedures                                                                        placed in the stenotic tract of the airway under direct tel-
 Premedication: intranasal dexmedetomidine (3 mcg/kg), 30 minutes before                       escopic or microscopic vision. An angioplasty balloon
 the procedure                                                                                 (Boston Scientific PTCA Dilatation Catheter) was utilised
 Mask induction with sevoflurane to for intravenous access placement                           for the first 26 dilations, while the TRACOE Aeris® Bal-
 Anaesthetic maintenance with propofol (target-controlled infusion model;                      loon Dilation Catheter was available for the 4 dilations per-
 3-4 mcg/ml) and remifentanil (0.07-0.15 mcg/kg/min)
                                                                                               formed in cases 13 and 14. The balloon size was selected
 Propofol infusion titrated to a clinically adequate level of anaesthesia,
 guided by the BIS (target 40-60) and remifentanil infusion titrated to                        by adding 1 mm for the larynx and 2 mm for tracheal ste-
 respiratory rate (target >10 breaths per min) to avoid apnoea and                             nosis to the outer diameter of the age-appropriate endotra-
 desaturation                                                                                  cheal tube size for the patient 7,8.
 Careful laryngoscopy and 2% lidocaine spray to the vocal cords (2-4 mg/kg)                    Balloon inflation was performed and maintained for 2 min
 Supplemental oxygen administered through a Portex Blue Line tube                              or until oxygen saturation decreased to less than 90%. It
 positioned through a nostril into the hypopharynx                                             was repeated 2-3 times for one min in patients who desatu-
 Propofol bolus to induce apnoea during balloon inflation, followed by hand-                   rated early. In cases of severe stenosis or fragile mucosa,
 mask ventilation or endotracheal intubation until spontaneous breathing
 was resumed                                                                                   the duration of each dilation was 20-30 sec to avoid mu-
                                                                                               cosal shearing and/or development of an ischaemic lesion.
                                                                                               In syndromic children, we used a balloon smaller than the
dial incisions were performed using a microlaryngoscopy                                        age-appropriate balloon and reduced the dilation time to
disposable sickle knife (Medtronic) mounted on a handle                                        20-30 sec for each inflation to minimise the risk of collat-
(Microfrance). In the case of posterior glottic stenosis, a                                    eral mucosal damage under the assumption that a congeni-
single median sagittal incision was performed either by a                                      tally small cricoid might not safely tolerate an excessive or
sickle knife or by the digital AcuBlade CO2 laser (Lumenis,                                    prolonged pressure that might cause cartilage damage.


Table II. Demographic and clinical characteristics of patients (Part I).
 Case         Age (months)              Gender              Comorbidities                         Previous surgery               Site of stenosis             Cause of stenosis
 No.



 1                        48                      Male             Severe prematurity                      No                 Posterior glottis and                 Acquired
                                                                                                                                    subglottis
 2                        76                      Male             Severe prematurity                      No                Subglottis and posterior               Acquired
                                                                                                                                      glottis
 3                        20                     Male           Head, neck, chest burns                  No                         Subglottis                      Acquired
 4                        30                     Male             Down’s syndrome                   PCTR and LTR               Subglottis, glottis.                  Mixed
 5                        27                     Male             Severe prematurity                     LTR                    Glottis, subglottis                 Acquired
 6                        81                     Male                 Prematurity                        No                         Subglottis                      Acquired
 7                        14                     Male                 Prematurity                 Endoscopic surgery            Glottis, subglottis                  Mixed
 8                         7                    Female           Genetic disease with                    No                         Subglottis                      Acquired
                                                                multiple malformations
 9                        51                      Male           Severe prematurity -                      LTR                Glottis, subglottis, and              Acquired
                                                                                                                                       trachea
 10                        6                      Male           Down’s syndrome and                       No                        Subglottis                       Mixed
                                                                previous cardiac surgery
 11                       20                      Male             Severe prematurity                      No                 Glottis and subglottis                  Mixed

 12                       64                    Female           Down’s syndrome. and                      No                        Subglottis                       Mixed
                                                                previous cardiac surgery
 13                        2                      Male            DiGeorge syndrome                        No                         Glottis                        Mixed
 14                       43                      Male             Severe prematurity                      No                 Glottis and subglottis                Acquired
PCTR: partial cricotracheal resection; LTR: laryngotracheal reconstruction; EPCTR: extended partial cricotracheal resection. * according to the Myer-Cotton classification 6;
++
   no longer dependent on noninvasive ventilation.




362
                                                                                                                                Balloon dilation for paediatric laryngeal stenosis




Noninvasive ventilation (CPAP) was given in the immedi-                                         are given as absolute numbers and percentages or median
ate postoperative period along with temporary adrenaline                                        values with ranges.
and budesonide aerosols. Dexamethasone (1 mg/kg), cefa-
zoline (25 mg/kg) and proton pump inhibitors were admin-                                        Results
istered in the operating room and were continued during
the postoperative period. Children were admitted to the                                         This case series comprised 14 children – median age: 28.5
                                                                                                months, range: 2-81 months; 12 boys and 2 girls; and 8
paediatric intensive care unit for at least one night after the
                                                                                                (57.1%) patients with severe prematurity and 5 (35.7%) with
surgery. If additional dilations were needed, repeated pro-
                                                                                                syndromic or genetically-defined disease. Patients had a his-
cedures were performed at 2-6-week intervals.
                                                                                                tory of prolonged intubation for severe prematurity (8/14,
The effectiveness of the procedure was defined by three
                                                                                                57.1%), respiratory distress at birth (3/14, 21.4%), previous
main outcome measures:                                                                          cardiac surgery (2/14, 14.3%) and severe burns extending to
• Stable improvement of airway patency with a reduction                                         the lower face, neck and chest (1/14, 7.1%). The detailed de-
   in the grade of stenosis according to the Myer-Cotton                                        mographic and clinical characteristics are reported in Table II.
   grading scale                                                                                Dilation laryngoplasty was the primary treatment in 11
• Clinical improvement and use of a phonatory valve that                                        children; among them, acquired LS was documented in 8,
   was not possible prior to dilation                                                           while mixed-type stenosis was found in 3 syndromic chil-
• Decannulation or tolerating a fully capped tracheostomy                                       dren (cases 10, 12, 13). A congenital membranous glot-
   cannula                                                                                      tic web (Cohen type 2) 9 affected case 13, but his stenosis
                                                                                                became symptomatic after intubation for cardiac surgery
Statistical analysis                                                                            (ventricular septal defect correction), adding mild acquired
Descriptive statistics were used to summarise the demo-                                         stenosis to the existing congenital stenosis. He was treated
graphic and clinical characteristics of patients. The results                                   with sickle knife lysis of the web, followed by dilation. In 3


Table II. Demographic and clinical characteristics of patients (Part II).
    Type of     Pre-op. grade           No. of           Post-op.                               Tracheotomy                 Clinical condition after         Follow-up (months)
  treatment      of stenosis*       endoscopic           grade of                                                                  treatment
                                     treatments          stenosis*
                                                                                      Predilation         Post dilation
     Primary                 III                  2                     I                Yes                   No                 Asymptomatic                          25

     Primary                 III                  2                     I                  No                  No            Mild fatigue at maximal                    25
                                                                                                                                       effort
     Primary                 III                  1                     I                 No                   No                 Asymptomatic                          16
     Adjuvant                III                  2                    III                Yes                  Yes               Phonatory valve                        19
     Adjuvant                III                  3                     II                Yes                  No                 Asymptomatic                          46
     Primary                 III                  1                     I                 No                   No                 Asymptomatic                          45
     Primary                 III                  1                    III                Yes                  Yes              Requires EPCTR                          42
     Primary                 III                  1                     I                 No                   No                 Asymptomatic                          25

     Adjuvant                IV                   6                    III                Yes                  Yes            Phonatory valve 24 h                      22

     Primary                 IV                   3                    III                Yes                  Yes               Phonatory valve                        12
                                                                                                                                    (daytime)
     Primary                 III                  2                    III                 No                  Yes               Phonatory valve                        13
                                                                                                                                    (daytime)
     Primary                 III                  2                     I                  No                  No                 Asymptomatic                           6

     Primary                 III                  3                     I                  No                  No                   Improved++                           6
     Primary                 III                  1                    II                  No                  No                   Improved++                           2
PCTR: partial cricotracheal resection; LTR: laryngotracheal reconstruction; EPCTR: extended partial cricotracheal resection. * according to the Myer-Cotton classification 6;
++
   no longer dependent on noninvasive ventilation.




                                                                                                                                                                                363
G. Cantarella et al.




children, balloon laryngoplasty was an adjuvant treatment                                       er grade of LS in 11 patients (78.6%). In 2 of the remain-
following reconstructive surgery (Tab. II).                                                     ing 3 children, subjective improvement occurred, and these
Grade III LS was preoperatively detected in 12 of 14 (85.7%)                                    children currently tolerate the use of a phonatory valve over
children; the remaining 2 patients presented with grade IV                                      a down-sized cannula.
stenosis. In one case of grade IV stenosis, a Down’s syn-                                       Six children had an initial tracheotomy, and one was tra-
drome patient had a cranio-caudal length of 3 mm and was                                        cheotomised at the time of the first dilation (Tab. III).
affected by mixed-type stenosis; in the other patient, recur-                                   Two were decannulated (cases No. 1 and 5) after BD,
rence of grade IV stenosis was observed with a size of ap-                                      and 5 remained tracheotomised. The benefit achieved by
proximately 5 mm after a second attempt at airway expansion                                     BD procedures in cases No. 4, 9, 10 and 11 did not al-
by cartilage grafts. Two patients with grade III stenosis and                                   low decannulation but the use of a phonatory valve was
1 patient with grade IV stenosis had previously undergone                                       tolerated all day long. Case 7 had no benefit at all: he
major surgical treatment (laryngotracheal reconstruction in                                     was an extremely premature child with severe multilevel
2 cases and partial cricotracheal resection followed by laryn-                                  fibrotic stenosis resulting from several endoscopic laser
gotracheal reconstruction in 1 case). In most treated patients                                  procedures performed elsewhere. After failing BD, he
(8/14, 57.1%), multiple sites of obstruction were detected.                                     underwent a successful reconstructive procedure with
A total of 30 dilation procedures were performed; the me-                                       subsequent decannulation. A detailed description of the
dian number of dilation procedures was 2 (range: 1-6).                                          clinical characteristics and outcomes in the subset of tra-
Only in one child (No. 11), affected by severe grade III                                        cheotomised patients is reported in Table III.
glotto-subglottic stenosis, was a tracheotomy performed on                                      BD laryngoplasty established a clearly detectable widening
the day of his first dilation to establish a secure distal air-                                 of the laryngeal lumen and respiratory improvement in 12
way. All the other dilation procedures were well tolerated                                      (85.7%) of the 14 children.
without any immediate or late complications. We continued                                       Figures 1-3 show the results achieved in 3 patients.
to use BD only if we found that there was clinical and en-
doscopic improvement compared to prior the endoscopy.
Postoperatively, the patients had a median follow-up of
                                                                                                Discussion
20.5 months, ranging from 2-46 months. Residual grade I                                         The use of BD is becoming progressively widespread in
LS was documented in 8 of 14 (57.1%) patients; grade II                                         several surgical fields and is considered a valid and mini-
and grade III LS were found in 1 (7.1%) and 5 (45.4%) pa-                                       mally invasive alternative to major open surgical proce-
tients, respectively. Follow-up laryngoscopy showed a low-                                      dures. The morbidity associated with airway dilation is low


Table III. Detailed description of clinical characteristics and outcomes in the subset of tracheotomised patients.
 Case No.               Comorbidities                       Previous procedures                   Clinical staging*                                            Outcome
                                                                 (elsewhere)                  (before balloon dilation)
 1                        Severe prematurity                       Multiple unsuccessful                      Multisite grade III LS                         Decannulated
                                                                  decannulation attempts
                                                                        (elsewhere)
 4                         Down’s syndrome                               PCTR; LTR                            Multisite grade III LS                   Non-decannulated but using
                                                                                                                                                            phonatory valve
 5                        Severe prematurity                  Approximately 18 endoscopic                     Multisite grade III LS                         Decannulated
                                                            laser procedures (elsewhere); LTS
 7                        Severe prematurity                    Multiple endoscopic laser                     Multisite grade III LS             Decannulated after reconstructive
                                                                  procedures (elsewhere)                                                                   procedure
 9                        Severe prematurity                       2 LTRs (1 elsewhere)                       Multisite grade IV LS                Non-decannulated but using
                                                                                                                                                        phonatory valve
 10                 Down’s syndrome and cardiac              Multiple unsuccessful extubation                Subglottic grade IV LS                Non-decannulated but using
                              surgery                                     attempts                                                                      phonatory valve
                                                                        (elsewhere)
 11               Severe prematurity and prolonged           Multiple unsuccessful extubation                 Multisite grade III LS              Tracheotomy at the first dilation;
                       endotracheal intubation                            attempts                                                                  non-decannulated but using
                                                                        (elsewhere)                                                                 phonatory valve. Waiting for
                                                                                                                                                       reconstructive surgery
LS: laryngeal stenosis; PCTR: partial cricotracheal resection; LTR: laryngotracheal reconstruction; * according to the Myer-Cotton classification 6.




364
                                                                                                               Balloon dilation for paediatric laryngeal stenosis




A                                        B                                        A                                         B




C                                        D                                       C                                          D




Figure 1. A: Grade III subglottic stenosis in a 20-month-old male intubated      Figure 2. Multilevel obstruction in a 76-month-old male, severe prematurity,
with several failed attempts of extubation elsewhere, due to diffuse burns to    with a history of prolonged invasive ventilation. A: Posterior glottic stenosis due
head, neck and chest. B: Radial incisions of the concentric stenosis. Note the   to inter-arytenoid scarring and associated subglottic stenosis. B: View of the
naso-oropharyngeal tube. C: Laryngoscopic view soon after dilation. D: La-       subglottic stenosis. C: Subglottic result achieved after a single dilation, a sec-
ryngoscopy performed 4 months later shows a stable scar and a sub-optimal        ond dilation is performed together with median incision of the posterior glottic
airway.                                                                          scar, and intralesional injection of triamcinolone; D: final result.


if the procedure is cautiously performed and if the indica-                       A                                     B
tions and contraindications are correctly respected 4,5,9-11.
In our opinion, BD is mainly indicated for grade I-III ste-
nosis (Cotton-Myer classification) and in rare cases of
thin diaphragm-like grade IV stenosis. Conversely, long
and thick stenoses, narrow congenital cricoid malforma-
tions, associated airway malacia, inflammation and multi-
site dense stenoses require open operations such as airway
expansion (laryngotracheal reconstruction with cartilage
                                                                                 C                                      D
grafts) or airway resection and anastomosis (cricotracheal
resection-CTR or an extended CTR); BD can be used as a
complementary procedure to enhance open surgical results.
Reconstructed multi-planar 3-D CT scans can be useful in
multi-site airway stenosis. However, precise interpretation
of laryngotracheal stenosis on CT scan in small children
can be challenging.
A dedicated dynamic and rigid endoscopy is more valuable                         Figure 3. Multilevel obstruction in a 64-month-old female, affected by Down
in detecting the exact site(s), length of the stenosis and ex-                   syndrome, with prior history of cardiac surgery. A: Subglottic mixed type con-
tent of remaining normal trachea 17. The quality of airway                       centric stenosis and posterior glottic stenosis. B: Immediate result after radial
mucosa is critical and a CT scan cannot detect this informa-                     incisions of the stenosis and dilation by 6 mm balloon. C: Laryngoscopic view 4
                                                                                 weeks later showing that the improvement in patency is maintained; a second
tion. Additionally, stagnated secretions at the stenotic sites                   incision and dilation treatment with 8 mm balloon is performed; D: final result.
will exaggerate the stenosis. We prefer doing CT/MRI scan
in cervico-mediastinal vascular anomalies, though virtual
bronchoscopy could be useful in complete airway stenosis                         Most patients with correct selection criteria can undergo
that will not allow passing of endoscopes.                                       laryngeal BD without needing tracheotomy. In our series,



                                                                                                                                                              365
G. Cantarella et al.




only one of the 8 non-tracheotomised patients (a child with      proving airway patency. A small hypoplastic cricoid and
a history of severe prematurity, affected by severe stridor      severe hypertrophy of subglottic submucosal glands with
and chest retractions at rest) needed a tracheotomy on the       an added intubation insult respond best to cricotracheal
day of his first dilation. All the remaining 7 patients under-   resection and anastomosis 17. Similarly, an elliptic cri-
went dilation procedures without tracheotomy, obtaining          coid malformation with minor intubation sequelae is best
immediate and persistent improvements in airway patency;         treated with cartilage graft(s) expansion cricoplasty 17.
these patients are currently asymptomatic despite their co-      In syndromic children, we used a balloon smaller in size
morbidities.                                                     than the age-appropriate balloon and reduced the dilation
The type of anaesthesia adopted is of crucial importance         time to 20-30 seconds for each inflation to minimise the
for both diagnostic assessment and the endoscopic proce-         risk of mucosal ulceration and ischaemia, as a congeni-
dure, as spontaneous breathing under intravenous anaes-          tally smaller cricoid might not safely tolerate excessive or
thesia provides an unobstructed surgical field and excellent     prolonged pressure.
evaluation of the dynamic airway function and the grades         Our results, attesting to the effectiveness of BD in approx-
of obstruction. We strongly advocate the use of noninva-         imately 86% of children with chronic LS as both primary
sive ventilation in the immediate postoperative period. The      and adjuvant treatment, corroborate the results of previous
continuous airway pressure acts like a pneumatic stent and       studies. In particular, Wentzel et al. 18 in 2014 reported a
promotes satisfactory airway epithelisation.                     success rate of 77% after BD in a cohort of 60 children,
Eight of our patients had a history of severe prematurity.       including 44 with subglottic stenosis; most of the cases
Recent advances in neonatal care have greatly improved           were acquired after prolonged intubation. This result was
the survival rates of premature infants who often require        further confirmed by a systematic literature review per-
intubation and mechanical ventilation. Currently, only a         formed by the same authors 18. The overall success rate,
small percentage of intubated babies develop LS, although        defined as symptomatic improvement, a decrease in the
the majority of paediatric LS cases are acquired because of      Myer-Cotton LS grade, decannulation, or the avoidance
prolonged intubation (Figs. 1,2).                                of reconstructive surgery, was slightly lower in their study
LS can be associated with some syndromes, and its treat-         (64%) than in ours.
ment may be challenging if comorbidities are severe. One         More recently, Wenzel and coworkers 4 evaluated the im-
of the 8 patients without tracheotomy in our series was af-      pact of balloon laryngoplasty on the management of acute
fected by 22q11 microdeletion syndrome. He needed non-           subglottic stenosis in 23 children and reported a success
invasive ventilation without oxygen supplementation for          rate of approximately 83%. Tracheotomy was required in
several months after dilation, as two previous thoracoto-        3 patients, and open laryngotracheal reconstruction was
mies for cardiac surgery subsequently weakened his chest         required in one case.
wall.                                                            The effectiveness of BD in mature severe laryngo (tra-
Most of the current literature regarding airway BD claims        cheal) stenosis has been previously documented in a small
the efficacy of BD mainly in cases of acute/subacute             case series of 8 children and adolescents by Guarisco and
stenosis 12-15 or membranous chronic stenosis with lim-          Yang 5, as an open surgical procedure was required in only
ited vertical extension. In both cases, a prerequisite for a     one patient. They simultaneously applied laryngeal stent-
successful procedure is the integrity of the cartilaginous       ing in the 4 tracheotomised patients, who were all sub-
laryngotracheal skeleton. Our small series suggests that         sequently decannulated. None of the remaining patients
the dilation procedure can be safely attempted in care-          required tracheotomy. In our opinion, an important fac-
fully selected cases of acquired or congenital LS and that       tor that determines success after BD is the cranio-caudal
a minor congenital subglottic malformation might not be          length of LS, which has not been well elucidated in many
an absolute contraindication. It is known that Down’s syn-       publications. The vertical extent of the stenosis is calcu-
drome might be associated with underdevelopment and/or           lated using a rigid, long and thin telescope that can go
malformation of the cricoid cartilage 16 with consequent         beyond the stenotic segment. The distal and proximal
airway narrowing. The frequent association of heart de-          points of the stenosis are ink-marked on the telescope at
fects in these patients will prompt early cardiac surgery,       the level of the incisor. The cranio-caudal length is then
and the subsequent prolonged intubation will add oedema          measured on a caliper ruler. In our opinion, any grade of
and/or scar tissue, thus worsening congenital stenosis.          thin diaphragm-like stenosis that is less than 5 mm in a
This was the case in 2 of our patients affected by Down’s        non-collapsing airway could respond to BD, although this
syndrome. Despite congenital stenosis, cautious dilation         needs to be confirmed with prospective and larger multi-
might solve the acquired component, significantly im-            centric studies. Open surgery must be considered when



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there is no change or when there is progression of stenosis       References
after 1-3 trials of BD, as well as in cases of severe carti-      1
                                                                       Smith MM, Cotton RT. Diagnosis and management of laryngotrache-
lage malformations It is difficult to decide the appropriate           al stenosis. Exp Rev Resp Med 2018;12:709-17. https://doi.org/10.10
period when adjuvant BD can be performed safely after                  80/17476348.2018.1495564
an open neck surgery. At the senior authors’ institution,
                                                                  2
                                                                       Choi SS, Zalzal GH. Changing trends in neonatal subglottic stenosis.
                                                                       Otolaryngol Head Neck Surg 2000;122:61-3. https://doi.org/10.1016/
BD is done only after 6 weeks following laryngotracheal                S0194-5998(00)70145-8
reconstruction or cricotracheal resection and anastomosis.        3
                                                                       Cohen MD, Weber TR, Rao CC. Balloon dilatation of tracheal and
We did not find any difference between the angioplasty                 bronchial stenosis. Am J Roentgenol 1984;142:477-8. https://doi.
                                                                       org/10.2214/ajr.142.3.477
balloons and the Tracoe balloon. However, our experience          4
                                                                       Wenzel AM, Schweiger C, Manica D, et al. Impact of balloon lar-
with the Tracoe Aeris balloon is not sufficient to make any            yngoplasty on management of acute subglottic stenosis. Eur Arch
definite conclusions.                                                  Otorhinolaryngol 2018;275:2325-31. https://doi.org/10.1007/s00405-
The importance of choosing patients wisely, of adequate                018-5064-7
training in endoscopy and all types of open airway surger-
                                                                  5
                                                                       Guarisco JL, Yang CJ. Balloon dilation in the management of severe air-
                                                                       way stenosis in children and adolescents. J Pediatr Surg 2013;48:1676-
ies, of anesthesia strategies and of postoperative manage-             81. https://doi.org/10.1016/j.jpedsurg.2012.12.035
ment cannot be overemphasised. The first choice of treat-         6
                                                                       Myer CM 3rd, O’Connor DM, Cotton RT. Proposed grad-
ment, whether endoscopic or open, is the best chance to                ing system for subglottic stenosis based on endotracheal tube
                                                                       sizes. Ann Otol Rhinol Laryngol 1994;103:319-23. https://doi.
cure patients with LS. Additional collateral damage due to             org/10.1177/000348949410300410
a wrongly chosen intervention might worsen the original           7
                                                                       Avelino M, Maunsell R, Jube Wastowski I. Predicting outcomes
stenosis and should be avoided. Surgical treatment of LS               of balloon laryngoplasty in children with subglottic stenosis. Int J
must be tailored depending on the patient’s clinical condi-            Pediatr Otorhinolaryngol 2015;79:532-6. https://doi.org/10.1016/j.
                                                                       ijporl.2015.01.022
tion, including the grade and type of stenosis and the pres-      8
                                                                       Sharma SD, Gupta SL, Wyatt M, et al. Safe balloon sizing for endo-
ence of concomitant disease. Complex, multi-site stenosis              scopic dilatation of subglottic stenosis in children. J Laryngol Otol
with additional cricoarytenoid ankyloses is best treated by            2017;131:268-72. https://doi.org/10.1017/S0022215117000081
open approaches 17.                                               9
                                                                       Cohen SR: Congenital glottic webs in children. A retrospective re-
                                                                       view of 51 patients. Ann Otol Rhinol Laryngol Suppl 1985;121:2-16.
An important limitation of our study is the relatively            10
                                                                       Hulstein S, Hoffman H. Technique for improved safety in the en-
small number of patients. Nevertheless, to the best of our             doscopic management of subglottic stenosis. Am J Otolaryngol
knowledge, this is the first Italian report on the system-             2016;37:490-2. https://doi.org/10.1016/j.amjoto.2015.10.009
atic use of BD laryngoplasty for treating paediatric LS.          11
                                                                       Maresh A, Preciado DA, O’Connell AP, et al. A comparative analysis
The present results encourage the use of BD as a primary               of open surgery vs endoscopic balloon dilation for pediatric subglot-
                                                                       tic stenosis. JAMA Otolaryngol Head Neck Surg 2014;140:901-5.
or adjuvant treatment to obtain adequate patency of the                https://doi.org/10.1001/jamaoto.2014.1742
airway with the aim of avoiding tracheotomy when fea-             12
                                                                       Axon PR, Hartley C, Rothera MP. Endoscopic balloon dilatation
sible and reducing the necessity of major reconstructive               of subglottic stenosis. J Laryngol Otol 1995;109:876-9. https://doi.
                                                                       org/10.1017/s0022215100131561
surgery.
                                                                  13
                                                                       Durden F, Sobol SE. Balloon laryngoplasty as a primary treatment for
                                                                       subglottic stenosis. Arch Otolaryngol Head Neck Surg 2007;133:772-
Conclusions                                                            5. https://doi.org/10.1001/archotol.133.8.772
                                                                  14
                                                                       Bakthavachalam S, McClay JE. Endoscopic management of subglot-
Our results confirm the efficacy of BD as a minimally inva-            tic stenosis. Otolaryngol Head Neck Surg 2008;139:551-9. https://
sive technique and as the first therapeutic option in selected         doi.org/10.1016/j.otohns.2008.07.024
cases of chronic and multilevel LS with an intact laryn-
                                                                  15
                                                                       Hebra A, Powell DD, Smith CD, et al. Balloon tracheoplasty in chil-
                                                                       dren: results of a 15-year experience. J Pediatr Surg 1991;26:957-61.
gotracheal cartilaginous framework. Failure of this first-             https://doi.org/10.1016/0022-3468(91)90843-i
line upfront endoscopic procedure does not compromise             16
                                                                       Hamilton J, Yaneza MM, Clement WA, et al. The prevalence of airway
the chances of success of subsequent major reconstructive              problems in children with Down’s syndrome. Int J Pediatr Otorhi-
                                                                       nolaryngol 2016;81:1-4. https://doi.org/10.1016/j.ijporl.2015.11.027
surgery.                                                          17
                                                                       Monnier P. Pediatric airway surgery. Berlin, Germany: Springer; 2011.
This limited series also confirms the validity of dilation lar-   18
                                                                       Wentzel JL, Ahmad SM, Discolo CM, et al. Balloon laryngoplasty for
yngoplasty as an adjuvant treatment to improve and stabi-              pediatric laryngeal stenosis: case series and systematic review. Laryn-
lise the results of previous reconstructive surgeries.                 goscope 2014;124:1707-12. https://doi.org/10.1002/lary.24524




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