10.14639/0392-100X-N1676
Objective. To review our 5-year experience with a modified version of glossoepiglottopexy for treatment of obstructive sleep apnoea syndrome (OSA) in two hospitals. Methods. A retrospective analysis was carried out on a cohort of adult patients affected by OSA suffering from primary collapse of the epiglottis who underwent a modified glossoepiglottopexy. All patients underwent drug-induced sleep endoscopy, polysomnographic and swallowing evaluation, and assessment with the Epworth Sleepiness Scale (ESS). Results. Forty-nine patients were retrospectively evaluated. Both the apnoea-hypopnoea index (AHI) (median AHIpost-AHIpre = -22.4 events/h; p &lt; 0.001) and oxygen desaturation index (ODI) showed a significant postoperative decrease (median ODIpost-ODIpre = -18 events/h; p &lt; 0.001), as did hypoxaemia index (median T90% post-T90% pre = -5%; p &lt; 0.001). The ESS questionnaire revealed a significant decrease in postoperative scores (median ESSpost-ESSpre =- 9; p &lt; 0.001). None of the patients developed postoperative dysphagia. Conclusions. Our 5-year experience demonstrates that modified glossoepiglottopexy is a safe and reliable surgical technique for treatment of primary epiglottic collapse in OSA patients.
text/xml
1827-675X
1676
en
Marco Barbieri
http://creativecommons.org/licenses/by-nc-nd/4.0
ACTA Otorhinolaryngologica Italica
surgery
Treatment of primary epiglottis collapse in OSA in adults with glossoepiglottopexy: a 5-year experience
Text.Serial.Journal
https://www.actaitalica.it/
42
OSAHS
2022-04-08
2021-05-07
2022-07-26
2022-07-26
265-272
1
Marco Barbieri
2022/04/08
10.14639/0392-100X-N1676
265
Unit of Otolaryngology, Head and Neck Surgery, IRCCS Ospedale Policlinico San Martino, Genoa, Italy; Department of Surgical Sciences and Integrated Diagnostics (DISC), University of Genoa, Genoa, Italy
1827-675X
surgery
272
Treatment of primary epiglottis collapse in OSA in adults with glossoepiglottopexy: a 5-year experience
https://www.actaitalica.it/article/download/1676/754
42
ACTA OTORHINOLARYNGOLOGICA ITALICA 2022;42:265-272; doi: 10.14639/0392-100X-N1676




OSAHS


Treatment of primary epiglottis collapse in OSA
in adults with glossoepiglottopexy: a 5-year experience
Glossoepiglottopessia per il collasso epiglottico primario negli adulti affetti da OSA:
esperienza quinquennale
Marco Fragale1-3, Claudio Sampieri1,2, Gregorio Santori2, Caterina Tripodi4, Francesco Missale1,5, Valeria Roustan6,
Fabiola Incandela7, Marta Filauro1,2,8, Andrea Marzetti4, Giorgio Peretti1,2, Marco Barbieri1,2
1
 Unit of Otolaryngology, Head and Neck Surgery, IRCCS Ospedale Policlinico San Martino, Genoa, Italy; 2 Department of
Surgical Sciences and Integrated Diagnostics (DISC), University of Genoa, Genoa, Italy; 3 Department of Medical and Surgical
Sciences and Advanced Technologies “GF Ingrassia”, ENT section, University of Catania, Catania, Italy; 4 Department of
Otorhinolaryngology, Fabrizio Spaziani Hospital, Frosinone, Italy; 5 Department of Molecular and Translational Medicine, University
of Brescia, Brescia, Italy; 6 Unit of Otorhinolaryngology, ASL 4 Liguria, Ospedale Sestri Levante, Sestri Levante, Italy; 7 Department
of Otorhinolaryngology, Maxillofacial and Thyroid Surgery, Fondazione IRCCS, National Cancer Institute of Milan, University of Milan,
Milan, Italy; 8 Department of Experimental Medicine (DIMES), University of Genoa, Genoa, Italy


SUMMARY
Objective. To review our 5-year experience with a modified version of glossoepiglottopexy
for treatment of obstructive sleep apnoea syndrome (OSA) in two hospitals.
Methods. A retrospective analysis was carried out on a cohort of adult patients affected by
OSA suffering from primary collapse of the epiglottis who underwent a modified glossoe-
piglottopexy. All patients underwent drug-induced sleep endoscopy, polysomnographic and
swallowing evaluation, and assessment with the Epworth Sleepiness Scale (ESS).
Results. Forty-nine patients were retrospectively evaluated. Both the apnoea-hypopnoea
index (AHI) (median AHIpost-AHIpre = -22.4 events/h; p < 0.001) and oxygen desaturation
index (ODI) showed a significant postoperative decrease (median ODIpost-ODIpre = -18
events/h; p < 0.001), as did hypoxaemia index (median T90% post-T90% pre = -5%; p < 0.001).
                                                                                                 Received: May 7, 2021
The ESS questionnaire revealed a significant decrease in postoperative scores (median
                                                                                                 Accepted: November 9, 2021
ESSpost-ESSpre =- 9; p < 0.001). None of the patients developed postoperative dysphagia.         Published online: April 8, 2022
Conclusions. Our 5-year experience demonstrates that modified glossoepiglottopexy is a
safe and reliable surgical technique for treatment of primary epiglottic collapse in OSA         Correspondence
patients.                                                                                        Claudio Sampieri
KEY WORDS: OSA, glossoepiglottopexy, epiglottis collapse, surgery                                Unit of Otolaryngology, Head and Neck Surgery,
                                                                                                 IRCCS Ospedale Policlinico San Martino, largo
                                                                                                 Rosanna Benzi 10, 16132 Genova, Italy
RIASSUNTO                                                                                        E-mail: claudio.sampieri@outlook.com
Obiettivo. Analisi della nostra esperienza a 5 anni con una versione modificata di
glossoepiglottopessia su una coorte di pazienti affetti da sindrome delle apnee ostruttive del     How to cite this article: Fragale M, Sampieri
sonno (OSA) e collasso primario dell’epiglottide in due diversi centri ospedalieri.                C, Santori G, et al. Treatment of primary epi-
Metodi. Un’analisi retrospettiva è stata effettuata su una coorte di pazienti affetti da OSA       glottis collapse in OSA in adults with glossoe-
e collasso primario dell’epiglottide trattati con glossoepiglottopessia modificata. Tutti i        piglottopexy: a 5-year experience. Acta Otorhi-
pazienti sono stati valutati con endoscopia in sedazione, valutazione polisonnografica, con        nolaryngol Ital 2022;42:265-272. https://doi.
questionario Epworth Sleepiness Scale (ESS).                                                       org/10.14639/0392-100X-N1676
Risultati. Quarantanove pazienti sono stati inclusi nell’analisi. I seguenti valori hanno
mostrato una significativa diminuzione postoperatoria: l’indice di apnea ed ipoapnea             © Società Italiana di Otorinolaringoiatria
                                                                                                 e Chirurgia Cervico-Facciale
(mediana AHIpost-AHIpre = -22,4 eventi/h; p < 0,001), l’indice di desaturazione (mediana
ODIpost-ODIpre = -18 eventi/h; p < 0,001), l’indice di ipossiemia (mediana T90% post -T90%
                                                                                                                OPEN ACCESS
pre
    = -5%; p < 0,001). Il questionario ESS (mediana ESSpost-ESSpre = -9; p < 0,001). Nessuno
ha sviluppato disfagia postoperatoria.                                                           This is an open access article distributed in accordance with
                                                                                                 the CC-BY-NC-ND (Creative Commons Attribution-Non-
Conclusioni. La nostra esperienza dimostra che la glossoepiglottopessia modificata è una         Commercial-NoDerivatives 4.0 International) license. The
tecnica chirurgica sicura e affidabile per il trattamento dei collassi epiglottici primari nei   article can be used by giving appropriate credit and mentio-
pazienti affetti da OSA.                                                                         ning the license, but only for non-commercial purposes and
                                                                                                 only in the original version. For further information: https://
PAROLE CHIAVE: OSA, glossoepiglottopessia, collasso epiglottide, chirurgia                       creativecommons.org/licenses/by-nc-nd/4.0/deed.en




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M. Fragale et al.




Introduction                                                      period from January 1, 2015 to December 31, 2019 on a
                                                                  cohort of patients affected by OSA who underwent glos-
Obstructive sleep apnoea syndrome (OSA) is a prevalent dis-
                                                                  soepiglottopexy at the Department of Otorhinolaryngology
ease affecting around 20% of the population (reaching up to
                                                                  and Head and Neck Surgery of the University of Genoa and
60% in those over 65 years) 1 with potentially life-threaten-
                                                                  the Department of Otorhinolaryngology - Fabrizio Spazi-
ing consequences as it is associated with other comorbidi-
                                                                  ani Hospital in Frosinone.
ties such as cardiovascular events, neurocognitive impair-
                                                                  For patients in whom multilevel surgery was performed,
ment and stroke. The gold standard in the management of
                                                                  all procedures were considered in the analysis.
upper airway collapse in OSA is continuous positive airway
                                                                  Tonsillectomy was carried out by preserving the
pressure (CPAP). Despite its proven efficacy, a significant
                                                                  palatoglossus and palatopharyngeus muscles. Septoplasty
number of patients cannot tolerate this form of treatment and
                                                                  and turbinoplasty were performed under rhinoscopy 9.
seek other alternatives. In this regard, surgery has a role in
                                                                  Non-resective pharyngoplasty was performed using the
non-compliant patients who are not willing to receive CPAP
                                                                  barbed suspension pharyngoplasty technique (BSP) 9,
therapy and in cases where CPAP therapy fails to restore nor-
                                                                  barbed reposition pharyngoplasty (BRP) 10, functional
mal breathing 2. In this setting, the effectiveness of surgical
                                                                  expansion pharyngoplasty (FEP) 11, modified expansion
treatment in reducing OSA-related cardiovascular morbidity
                                                                  sphincter pharyngoplasty (MESP) 12, or barbed anterior
and mortality has been demonstrated 3.
                                                                  pharyngoplasty (BAPh) 13.
In recent years, the role of surgery in the management of
                                                                  Inclusion criteria were: OSA confirmed by polysomnography
OSA has been evolving: the development of new tech-
                                                                  study with an apnoea-hypopnoea index (AHI) > 15 episodes/h;
niques, together with a better understanding of their indica-
                                                                  body mass index (BMI) < 35 kg/m2; primary epiglottic collapse
tions, has allowed for more precise selection of patients in
                                                                  diagnosed by DISE; and ease of laryngeal and oropharyngeal
order to tailor surgery to the multitude of anatomical con-
                                                                  exposure (Laryngoscore < 6). The latter was evaluated with
ditions that characterise OSA 4. When choosing the right
technique, structured pre-operative work-up is essential:         the Laryngoscore instrument, which is commonly employed
drug-induced sleep endoscopy (DISE) is crucial, as it is the      in our hospitals to select patients for transoral procedures 14.
only exam that can recreate sleeping conditions and allow         As is standard policy at our clinics, we strongly suggest CPAP
surgeons to identify the site and mechanism of obstruc-           therapy, especially to patients with an AHI > 30, and reserve
tion 5. Moreover, DISE facilitates the recognition of ob-         surgical treatment only for those who are not compliant or
structive patterns that are not evident during awake flexible     who do not respond to non-surgical therapy. Major comorbidi-
transnasal video endoscopy, such as epiglottic collapse 6.        ties, severe tongue base hypertrophy, cranio-facial malforma-
Epiglottic collapse is well known in the literature as one of     tions, laryngeal dysfunction (swallowing, motility disorders,
the possible conditions implicated in paediatric laryngoma-       and laryngeal stenosis) and other sleep-related disorders were
lacia. In adults, while laryngomalacia is more of an anec-        considered exclusion criteria.
dotic entity that has not been clearly described, primary         All patients underwent a standard preoperative evaluation
collapse of the epiglottis can be related to treatment failure    protocol consisting of awake flexible transnasal video en-
of CPAP 3. This happens as CPAP-generated airflow pushes          doscopy, polysomnographic assessment and DISE evalua-
down the epiglottis, closing the laryngeal aditus, and con-       tion. Postoperative evaluation was performed at 6 months
sequently worsening the airway obstruction 7. Non-surgi-          after surgery by clinical, polysomnographic and endoscopic
cal treatment has been demonstrated to be inadequate to           assessment. All data were extracted from a single database.
treat this condition, with mandibular advancement devices
(MADs) leading to disappointing results 8. In this respect,       Clinical evaluation
a surgical modification of the Monnier’s glossoepiglot-           Preoperatively, all patients underwent thorough otolaryn-
topexy was developed and published with preliminary data          gologic physical examination. Clinical history was collect-
in 2017, demonstrating encouraging results 7. Herein, we          ed focusing on sleep habits and sleep disturbances. BMI
describe our experience with this technique together with         was also reported. The Epworth Sleepiness Scale (ESS)
the results obtained in a cohort of patients suffering from       was used to rate daytime sleepiness 15, while swallowing
primary collapse of the epiglottis treated at two hospitals.      function was assessed by the Eating Assessment Tool (EAT
                                                                  10) 16 and the penetration aspiration scale 17.
Materials and methods                                             Respiratory polygraphic study
Study design and population                                       All patients underwent a sleep study with cardiorespiratory
A retrospective multicentric analysis was carried out on a        monitoring (Vital night, Vital aire, Milan Italy). The



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                                                                  Glossoepiglottopexy for primary epiglottis collapse in adult OSA patients




cardiorespiratory analysis comprised nocturnal snoring          Outcome evaluation
sound, arterial oxygen saturation, body position, nasal         Surgical success was evaluated at least 6 months after sur-
and mouth airflow, thoracic and abdominal respiratory           gery, performing a respiratory polysomnographic study
movements and heart rate. To determine the severity of          and repeating the ESS questionnaire 15. Criteria for evalua-
sleep apnoea, we considered the AHI, oxygen desaturation        tion of the outcomes are in agreement with Montevecchi et
index (ODI) and T < 90% (percent of total time with oxygen      al. 20. Cured: AHI < 5 and ESS < 10 and reduction of both
saturation less than 90%).                                      >50%. Success: AHI < 20 and ESS < 10 and reduction of
                                                                both > 50%. Failure: AHI > 20 and any ESS value and re-
Drug-induced sleep endoscopy                                    duction of both < 50%.
At both centres, DISE was performed with the patient
in a supine position. Transnasal flexible endoscopy             Statistical analysis
was performed using a high definition fibreoptic video-         The results are expressed as mean ± standard deviation,
endoscope connected to an Evis Exera II CLV-180B light          median, or percentage. Sample size was calculated by
source (Olympus Medical Systems Corporation, Tokyo,             assuming effect size = 0.45, α = 0.05, power (1-β error
Japan).                                                         probability) = 0.80 for a two-tailed paired test. With these
During the examination, head rotation and positioning of        parameters, a minimum sample size of 43 patients was re-
the patient in lateral decubitus were performed to assess       quired. The Shapiro-Wilk test was used to assess normal
the positional component of the collapse. Moreover, chin        distributions of continuous variables. Categorical variables
lift and mandibular pull up manoeuvres were routinely           were analysed with χ2 test or Fisher’s exact test as appropri-
performed to evaluate the possible benefit in applying          ate. Comparisons between continuous variables were per-
a MAD. Midazolam was administered with intravenous              formed with the Mann-Whitney-Wilcoxon rank sum test.
repeated bolus in a range of 1-3 mg, while propofol was         The evaluation of the continuous variables before and after
administered intravenously via target-controlled infusion.      treatment was carried out using the Wilcoxon signed-rank
At the end of the procedure, flumazenil was used to             test, plotting them with paired boxplot and adding lines to
antagonise the effects of midazolam. The use of a low           points for each patient to show the trend change. Consider-
dose of midazolam and propofol allows sedation to be            ing the binary successful outcome, pre-treatment clinical
as physiologic as possible, with snoring, apnoea events,        and polysomnographic covariates were investigated with
controlled desaturations and rapid recovery 18. The NOHL        univariable and multivariable logistic regression models.
classification was used to assess the obstruction severity      Statistical significance was assumed in each test with a
at multiple levels 19.                                          two-tailed p value < 0.05. Statistical analysis was carried
                                                                out using the R software/environment (version 3.6.3; R
Surgical glossoepyglottopexy                                    Foundation for Statistical Computing. Vienna, Austria).
The glossoepyglottopexy procedure has been previously
described 7; the main steps are briefly summarised as fol-
                                                                Results
lows. The procedure is carried out under microlaryngos-
copy with the patient lying in Boyce-Jackson’s position;        From January 2015 to December 2019, 49 patients affected
firstly, the surgeon with a Sataloff laryngoscope (Microf-      by OSA with primary epiglottic collapse underwent
rance Sataloff Laryngoscopes 124, Medtronic ENT, Jack-          glossoepiglottopexy. By DISE, the most frequent pattern of
sonville FL USA) exposes the base of the tongue, the entire     epiglottic collapse was complete anteroposterior collapse,
valleculae and the epiglottis. Secondly, with a CO2 laser       followed by partial anteroposterior collapse; in our series,
(Ultrapulse Dualpro Laser CO2, Lumenis, Yokneam, Israel)        no patients showed a lateral collapse of the epiglottis. In
coupled with a microscope, the operator vaporises the mu-       37 patients (75.5%), concomitant to glossoepiglottopexy, a
cosa overlying the valleculae and the base of the tongue.       non-resective pharyngoplasty was also performed according
Finally, from outside of the neck, two 16-gauge needles are     to the palatal collapse. Furthermore, tonsillectomy was
inserted projecting out of the valleculae, serving as a guide   performed in 26 patients (53.1%), and septoplasty and
to apply, through a loop, number 1 Premilene® sutures (Pre-     turbinoplasty in 9 patients (18.4%). The main patient
milene, Braun, Melsungen Germany) that embrace the hy-          characteristics are reported in Table I. During the
oid bone and stitch the lingual surface of the epiglottis to    postoperative period, concomitant with polysomnographic
the base of the tongue. Both wires are then fixed outside of    assessment, no significant change in BMI was observed
the neck, anteriorly to the larynx, using a Silastic sheet to   [mean ΔBMI = 0.035 (95% CI -0.123, 0.193), p = 0.66].
protect the skin from local trauma.                             Complications occurred in 2 of 49 procedures: one patient



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M. Fragale et al.




Table I. Main characteristics of the patients enrolled.                                   as did ODI [median ODIpost-ODIpre = -18 events/h (95%
                                               Overall (n = 49)                           CI -13.0, -20.0); p < 0.001] and T90% [median T90% post-T90%
 Age                                                                                      pre
                                                                                              = -5% (95% CI -5.0, -7.0); p < 0.001]. Daytime sleepiness,
   Mean (SD)                                          50.7 (11.7)                         assessed by the ESS questionnaire, also showed a significant
   Median (min, max)                               50.0 [24.0, 74.0]                      decrease in postoperative scores evaluated at 6 months after
 Sex                                                                                      surgery [median ESSpost-ESSpre = -9.63 (95% CI -7, -11);
   F                                                   9 (18.4%)                          p < 0.001].
   M                                                  40 (81.6%)                          Clinical swallow evaluation was negative and EAT-10
 Tonsillectomy                                                                            scored 0 in all patients after surgery. All patients received
   No                                                 23 (46.9%)
                                                                                          a score of 1 at the penetration-aspiration scale evaluation
   Yes                                                26 (53.1%)
                                                                                          postoperatively. Post-treatment AHI was < 5 in 40.8% of
                                                                                          patients (n = 20) and postoperative ESS scored less than 7
 Septo-turbinoplasty
                                                                                          points in 89.8% of cases (n = 44).
   No                                                 40 (81.6%)
                                                                                          Considering the criteria for outcome evaluation, a successful
   Yes                                                 9 (18.4%)
                                                                                          procedure was obtained in 34 cases (69%) and failure in 15
 Pharyngoplasty
                                                                                          (31%); notably, 8 of these failures were due to ESSpost > 10
   BAPh                                                 1 (2.0%)
                                                                                          (1 patient) or its improvement < 50% (7 patients); on the
   BRB                                                  4 (8.2%)
                                                                                          other hand, only 4 failures were due to an AHIpost > 20, as
   BRP                                                12 (24.5%)                          shown in Figure 2. The analysis of pre-treatment clinical
   BSP                                                11 (22.4%)                          and polysomnographic covariates showed that higher ESSpre
   FEP                                                 6 (12.2%)                          values were associated with a higher chance of successful
   MESP                                                 3 (6.1%)                          surgery at both univariable (OR = 1.24, 95% CI 1.08-1.47,
   No                                                 12 (24.5%)                          p = 0.005) and multivariable analysis (OR = 1.22, 95% CI
F: female; M: male; BAPh: barbed anterior pharyngoplasty; BRB: barbed Roman blinds;       1.06-1.46, p = 0.013), whereas older age was related to a
BRP: barbed reposition pharyngoplasty; BSP: barbed suspension pharyngoplasty;
FEP: functional expansion pharyngoplasty; MESP: modified expansion sphincter              lesser chance of success (OR = 0.94, 95% CI 0.88-0.99,
pharyngoplasty.                                                                           p = 0.035), which was not confirmed after adjustment for
                                                                                          the ESS effect (p = 0.152; Tab. III, Fig. 3). None of the
                                                                                          other clinical variables were associated with post-treatment
had suture breakage at 7 days post-operatively, but flexible                              outcomes.
transnasal video endoscopic control revealed the stability of
the glossoepiglottopexy, while the other had the epiglottis
lacerated by the sutures that were probably placed too high.                              Discussion
Neither bleeding, dysphagia, nor aspiration occurred in any                               In adult patients suffering from OSA, collapse of the
case, and postoperative AHI improved from 66 to 10 and                                    epiglottis may be primary or secondary: the latter occurs
from 37 to 9, respectively.                                                               when a bulky tongue base pushes the epiglottis backward.
Comparisons between paired parameters measured before                                     On the other hand, primary collapse of the epiglottis
and after surgery are reported in Table II, whereas the                                   may result from an altered conformation of the epiglottis
trends for each patient are shown in Figure 1. AHI showed                                 (cartilage deformation due to pharyngeal wall compression
a significant postoperative decrease [median AHIpost-                                     during sleep or laxity of the glossoepiglottic ligament) in
AHIpre = -22.4 events/h (95% CI -25.7, -19.0; p < 0.001)],                                combination with the high negative intrathoracic pressure


Table II. Comparison between variables measured before and after surgery (mean values and SD).
 Variable                                    Before surgery                           After surgery                                                  P value
 AHI                                                    29.37 ± 1.81                                 7.02 ± 6.51                                    < 0.001
 ESS                                                    13.65 ± 5.41                                 4.02 ± 3.15                                    < 0.001
 ODI                                                   27.87 ± 13.79                                 7.75 ± 9.92                                    < 0.001
 T90(%)                                                  7.96 ± 6.55                                 1.35 ± 1.79                                    < 0.001
 SpO2(%)                                                86.80 ± 2.86                                95.04 ± 2.14                                    < 0.001
AHI: apnoea-hypopnea index; ESS: Epworth sleepiness scale; ODI: oxygen desaturation index; T90: percent of the total time passed with an oxygen saturation level lower than 90%;
SpO2: mean peripheral capillary oxygen saturation.




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                                                                                         Glossoepiglottopexy for primary epiglottis collapse in adult OSA patients




Figure 1. Box-plots for comparisons between variables measured before (Pre) and after surgery (Post), with trends in each patient. (A) apnoea-hypopnea index
(AHI); (B) Epworth sleepiness scale (ESS); (C) oxygen desaturation index (ODI); (D) percent of the total time passed with oxygen saturation level less than 90% (T90);
(E) mean peripheral capillary oxygen saturation (SpO2).


generated during obstructive events 7. This deformity of                             awake investigations. In awake findings, the laryngeal
the epiglottis can be congenital or created by the pressure                          obstruction may be only hypothesised as being due to the
of parapharyngeal fat pads and chronic collapse of the                               deformed epiglottic shape, while the direct visualisation
retroglottal airway.                                                                 of the laryngeal collapse in many cases is possible only
In OSA patients, both medical and surgical treatment                                 during the sedated state 21. The introduction of DISE in
must be tailored to the physiopathology of the individual                            the diagnostic routine has revealed the high incidence
case. New tools have been developed to improve patient                               of laryngeal obstruction due to primary and secondary
selection and the therapeutic solutions that can be offered.                         collapse of the epiglottis, as widely documented in the
One such tool is DISE, which allows to accurately identify                           literature, reaching up to 73% of cases 3,6.
patterns of collapse that cannot otherwise be seen with                              OSA is a disease with multiple treatment modalities



                                                                                                                                                                269
M. Fragale et al.




  ESS: Epworth sleepiness scale; AHI: apnoea-hypopnea index.

Figure 2. Venn diagram showing causes of failure among the 15 patients
experiencing a non-successful procedure.


including positive airway pressure devices, surgery, behav-
ioural treatment and oral appliances 3, but when approach-
ing epiglottic collapse the therapeutic choices are limited.
CPAP has demonstrated suboptimal results with these pa-                                              ESS: Epworth sleepiness scale.
tients who often require higher pressures to be effective, with
possible consequences on patient compliance 3. Vonk et al.                                  Figure 3. Estimates of success probability with 95% CI bands according to
analysed the presence of primary epiglottis collapse in non-                                univariable or multivariable logistic regression models presented in Table III.
apnoeic snoring patients, non-positional OSA patients and
position-dependent OSA patients, while evaluating the im-                                   non-positional OSA and position-dependent OSA patients,
pact of head-turning, chin-lift, mandibular pull-up manoeu-                                 while suggesting the need for new trials to evaluate other
vres and body position during DISE in 324 patients. Their                                   therapeutic solutions that possibly combine position-therapy
results indicate that in the vast majority of cases, primary ep-                            with surgery 22.
iglottis collapse was position-related. Nevertheless, they did                              Surgery remains the treatment of choice in many cases of
not explain the pathophysiological mechanism underlying                                     epiglottic collapse, and different surgical approaches have


Table III. Univariable and multivariable logistic regression models with a successful surgical procedure as the dependent variable, considering AHI and ESS and
applying Montevecchi’s criteria 20.
 Variable                                            Failure           Success or             OR (95% CI, p value)                 OR (95% CI, p value)
                                                                          cure                     (univariable)                      (multivariable)
 Age                                Mean (SD)          56.2 (13.0)            48.3 (10.3)            0.94 (0.88-0.99, p = 0.035)              0.96 (0.90-1.01, p = 0.152)
 Sex                                   F               2 (22.2%)              7 (77.8%)                           -                                        -
                                       M               13 (32.5%)             27 (67.5%)             0.59 (0.08-2.88, p = 0.549)                           -
 Tonsillectomy                         No              5 (21.7%)              18 (78.3%)                          -                                        -
                                      Yes              10 (38.5%)             16 (61.5%)             0.44 (0.12-1.53, p = 0.210)                           -
 Septoplasty / Turbinoplasty           No              12 (30.0%)             28 (70.0%)                          -                                        -
                                      Yes              3 (33.3%)              6 (66.7%)              0.86 (0.19-4.59, p = 0.845)                           -
 AHIpre                             Mean (SD)          29.3 (10.7)            29.4 (12.4)            1.00 (0.95-1.06, p = 0.989)                           -
 ESS pre                            Mean (SD)           10.1 (5.0)             15.2 (4.9)            1.24 (1.08-1.47, p = 0.005)              1.22 (1.06-1.46, p = 0.013)
 ODI pre                            Mean (SD)          29.6 (12.7)            27.1 (14.4)            0.99 (0.94-1.03, p = 0.556)                           -
 T90 pre                            Mean (SD)           5.5 (3.9)               9.0 (7.2)            1.19 (1.02-1.46, p = 0.061)                           -
 SpO2 pre                           Mean (SD)           86.9 (3.1)             86.7 (2.8)            0.98 (0.78-1.21, p = 0.822)                           -
AHI: apnoea-hypopnoea index; ESS: Epworth sleepiness scale; F: female; M: male; PRE: before surgery; ODI: oxygen desaturation index; T90: percent of the total time passed with oxy-
gen saturation level less than 90%; SpO2: peripheral capillary oxygen saturation.



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                                                                   Glossoepiglottopexy for primary epiglottis collapse in adult OSA patients




been described. Tongue base advancement or hyoid sus-            the role of ESS reduction in defining successful treatment
pension are theoretical surgical options in OSA, but to date     in the applied classification 20. In 7 (47%) of our patients,
there are no studies that have demonstrated their direct ef-     the definition of failure was due only to not achieving ESS
fect on epiglottis collapse 23. Partial epiglotectomy has been   reduction < 50%, despite having an ESSpost < 10 and ob-
the most widely described technique; however, it should be       taining recovery of AHI parameters. In fact, low values of
noted that excessive resection of the epiglottis can lead to     baseline ESS in non-symptomatic patients are less likely
aspiration, whereas insufficient resection might not cure the    to change, despite recovery by polygraphy. These findings
OSA. Moreover, there is no standard method to assess the         may help to improve this classification in defining surgical
volume of the epiglottis to excise without causing postop-       outcomes.
erative sequelae. Subsequently, more cautious approaches         Finally, we acknowledge that the present study has intrinsic
have been developed. Bourolias et al. proposed a transoral       limitations considering its retrospective nature. Moreover,
conservative technique to treat epiglottic collapse. A laser     the cohort of patients analysed is limited, even though to
beam was delivered on the lingual surface of the epiglottis      our knowledge it is one of the largest case series present in
in order to model it in a new curved shape, warping towards      the literature on this specific category of patients3. In fact,
the direction of laser beam application thus freeing the la-     OSA is a complex disease, and many factors can contribute
ryngeal inlet. Nevertheless, the technique has been studied      to upper airway collapse. A clear understanding of such a
only on anatomical ex-vivo models 24.                            complex condition, by identifying the key factors that play
In order to treat primary epiglottic collapse without ablat-     a role in the different upper airway obstruction patterns can
ing it, a surgical technique called epiglottis stiffening was    aid in the definition of better therapeutic protocols and tar-
recently developed: it consists of cauterising the lower half    geted compound surgical strategies.
of the lingual side of the epiglottis in the area between the
lateral glossoepiglottic folds, and avoiding reaching the
                                                                 Conclusions
free margin of the epiglottis itself 25. By maintaining the
free edge unharmed, the risk of dysphagia and aspiration is      Our 5-year experience demonstrates that glossoepiglot-
overcome. However, in our view, adding two nylon sutures         topexy is a safe and reliable surgical technique for the
to embrace the hyoid bone ensures safer and long-lasting         treatment of primary epiglottic collapse in OSA patients.
results, while maintaining the important epiglottic function     By not producing major anatomical alterations, it maintains
of glottic plane protection.                                     oropharyngeal and laryngeal functions. In addition, it is ca-
In the present work, this technique was demonstrated to be       pable of improving the main polysomnographic parameters
a safe and reliable choice to treat primary collapse of the      and has the potential to increase compliance and efficacy
epiglottis in OSA patients. In our series, the complication      of positive airway pressure devices in this category of pa-
rate was very low (4%) and none of these cases experienced       tients.
dysphagia or inhalation. Regarding outcomes, our results
showed significant (p < 0.001) post-operative improvement        Conflict of interest statement
of AHI, ODI, T90% and ESS after surgical treatment with          The authors declare no conflict of interest.
mean values approaching the normal population. In our
opinion, these results are consistently achievable if reason-    Funding
able selection criteria are respected. As a general rule, pa-    This research did not receive any specific grant from fund-
tients should not be affected by severe OSA with > 30 AHI        ing agencies in the public, commercial, or not-for-profit
and should also not be obese: otherwise, CPAP should be          sectors.
always advised in the first instance and surgical treatment
should be reserved only for those who do not respond to or       Authors’ contributions
who refuse treatment with positive airway pressure.              Conceptualisation: MF, MB. Data curation: MF, CS, FM.
Further research needs to be carried to determine if glos-       Formal analysis: FM, GS. CS. Investigation: CT, RV, FI,
soepiglottopexy can enhance compliance and increase the          MF. Methodology: MF, FI, AM, CT, GS, VR. Supervision:
effectiveness of CPAP in this category of patients. In our       GP, MB, AM. Writing – original draft: CS, MF. Writing –
cohort, considering pre-treatment clinical and polysomno-        review and editing: FM, MB, GP.
graphic variables, ESS score was the only covariate that
independently associated with surgical outcomes as pre-          Ethical consideration
viously observed in patients treated with palatal or lateral     This study was approved by the Institutional Ethics
oropharyngeal collapse 4. This finding may be explained by       Committee (CER Liguria) (approval number/protocol



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M. Fragale et al.




number 230/2019).                                                               11
                                                                                     Sorrenti G, Piccin O. Functional expansion pharyngoplasty in the
                                                                                     treatment of obstructive sleep apnea. Laryngoscope 2013;123:2905-
The research was conducted as a retrospective study on                               2908. https://doi.org/10.1002/lary.23911
patient records in accordance with the ethical standards                        12
                                                                                     Lorusso F, Dispenza F, Modica DM, et al. The role of modified
of the institutional and/or national research committees                             expansion sphincter pharyngoplasty in multilevel obstructive sleep
and with the 1964 Helsinki Declaration and its later                                 apnea syndrome surgery. Int Arch Otorhinolaryngol 2018;22:432-
                                                                                     436. https://doi.org/10.1055/s-0038-1648248
amendments or comparable ethical standards. Informed                            13
                                                                                     Salamanca F, Costantini F, Mantovani M, et al. Barbed anterior
consent for disclosure of privacy in managing personal data                          pharyngoplasty: an evolution of anterior palatoplasty. Acta
for scientific purposes was obtained from all participants                           Otorhinolaryngol Ital 2014;34:434-438.
included in the study.
                                                                                14
                                                                                     Piazza C, Mangili S, Bon F Del, et al. Preoperative clinical
                                                                                     predictors of difficult laryngeal exposure for microlaryngoscopy:
                                                                                     the laryngoscore. Laryngoscope 2014;124:2561-2567. https://doi.
References                                                                           org/10.1002/lary.24803
                                                                                15
                                                                                     Vignatelli L, Plazzi G, Barbato A, et al. Italian version of the Epworth
1
     Iannella G, Maniaci A, Magliulo G, et al. Current challenges in the             sleepiness scale: external validity. Neurol Sci 2003;23:295-300.
     diagnosis and treatment of obstructive sleep apnea syndrome in                  https://doi.org/10.1007/s100720300004
     the elderly. Polish Arch Intern Med 2020;130:649-654. https://doi.         16
                                                                                     Belafsky PC, Mouadeb DA, Rees CJ, et al. Validity and reliability
     org/10.20452/pamw.15283                                                         of the eating assessment tool (EAT-10). Ann Otol Rhinol Laryngol
2
     Brietzke SE, Ishman SL, Cohen S, et al. National database analysis of           2008;117:919-924. https://doi.org/10.1177/000348940811701210
     single-level versus multilevel sleep surgery. Otolaryngol - Head Neck      17
                                                                                     Robbins JA, Coyle J, Rosenbek J, et al. Differentiation of normal
     Surg 2017;156:955-961. https://doi.org/10.1177/0194599817696503                 and abnormal airway protection during swallowing using the
3
     Torre C, Camacho M, Liu SYC, et al. Epiglottis collapse in adult                Penetration-Aspiration Scale. Dysphagia 1999;14:228-232. https://
     obstructive sleep apnea: a systematic review. Laryngoscope                      doi.org/10.1007/PL00009610
     2016;126:515-523. https://doi.org/10.1002/lary.25589                       18
                                                                                     Llatas MC, Porras GA, Gonzalez MTC, et al. Drug-induced
                                                                                     sleep endoscopy: a two drug comparison and simultaneous
4
     Missale F, Fragale M, Incandela F, et al. Outcome predictors for
                                                                                     polysomnography. Eur Arch Otorhinolaryngol 2014;271:181-187.
     non-resective pharyngoplasty alone or as a part of multilevel surgery,
                                                                                     https://doi.org/10.1007/s00405-013-2548-3
     in obstructive sleep apnea-hypopnea syndrome. Sleep Breath
     2020;24:1397-1406. https://doi.org/10.1007/s11325-019-01985-2.
                                                                                19
                                                                                     Vicini C, De Vito A, Benazzo M, et al. The nose oropharynx
                                                                                     hypopharynx and larynx (NOHL) classification: a new system of
5
     Kezirian EJ, Hohenhorst W, De Vries N. Drug-induced sleep                       diagnostic standardized examination for OSAHS patients. Eur Arch
     endoscopy: the VOTE classification. Eur Arch Otorhinolaryngol                   Otorhinolaryngol 2012;269:1297-1300. https://doi.org/10.1007/
     2011;268:1233-1236. https://doi.org/10.1007/s00405-011-1633-8                   s00405-012-1965-z
6
     Cavaliere M, Russo F, Iemma M. Awake versus drug-induced sleep             20
                                                                                     Montevecchi F, Meccariello G, Firinu E, et al. Prospective multicentre
     endoscopy: evaluation of airway obstruction in obstructive sleep                study on barbed reposition pharyngoplasty standing alone or as a part
     apnea/hypopnoea syndrome. Laryngoscope 2013;123:2315-2318.                      of multilevel surgery for sleep apnoea. Clin Otolaryngol 2018;43:483-
     https://doi.org/10.1002/lary.23881                                              488. https://doi.org/10.1111/coa.13001
7
     Roustan V, Barbieri M, Incandela F, et al. Transoral glossoepiglottopexy
                                                                                21
                                                                                     Gazayerli M, Bleibel W, Elhorr A, et al. A correlation between the
     in the treatment of adult obstructive sleep apnoea: a surgical                  shape of the epiglottis and obstructive sleep apnea. Surg Endosc
     approach. Acta Otorhinolaryngol Ital 2018;38:38-44. https://doi.                Other Interv Tech 2006;20:836-837. https://doi.org/10.1007/
     org/10.14639/0392-100X-1857                                                     s00464-005-0641-4
                                                                                22
                                                                                     Vonk PE, Ravesloot MJL, Kasius KM, et al. Floppy epiglottis during
8
     Kent DT, Rogers R, Soose RJ. Drug-induced sedation endoscopy in
                                                                                     drug-induced sleep endoscopy: an almost complete resolution by
     the evaluation of osa patients with incomplete oral appliance therapy           adopting the lateral posture. Sleep Breath 2020;24:103-109. https://
     response. Otolaryngol Head Neck Surg 2015;153:302-307. https://                 doi.org/10.1007/s11325-019-01847-x
     doi.org/10.1177/0194599815586978                                           23
                                                                                     Bae MR, Chung YS. Epiglottic collapse in obstructive sleep
9
     Barbieri M, Missale F, Incandela F, et al. Barbed suspension                    apnea. Sleep Med Res 2021;12:15-19. https://doi.org/10.17241/
     pharyngoplasty for treatment of lateral pharyngeal wall and palatal             SMR.2021.00948
     collapse in patients affected by OSAHS. Eur Arch Otorhinolaryngol          24
                                                                                     Bourolias C, Hajiioannou J, Sobol E, et al. Epiglottis reshaping using
     2019;276:1829-1835. https://doi.org/10.1007/s00405-019-05426-4                  CO2 laser: a minimally invasive technique and its potent applications.
10
     Vicini C, Hendawy E, Campanini A, et al. Barbed reposition                      Head Face Med 2008;4:5-8. https://doi.org/10.1186/1746-160X-4-15
     pharyngoplasty (BRP) for OSAHS: a feasibility, safety, efficacy            25
                                                                                     Salamanca F, Leone F, Bianchi A, et al. Surgical treatment of epiglottis
     and teachability pilot study. “We are on the giant’s shoulders.” Eur            collapse in obstructive sleep apnoea syndrome: epiglottis stiffening
     Arch Otorhinolaryngol 2015;272:3065-3070. https://doi.org/10.1007/              operation. Acta Otorhinolaryngol Ital 2019;39:404-408. https://doi.
     s00405-015-3628-3                                                               org/10.14639/0392-100X-N0287




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