Next Article in Journal
Comparative Effectiveness of Apixaban and Rivaroxaban Lead-in Dosing in VTE Treatment: Observational Multicenter Real-World Study
Previous Article in Journal
Krüppel-like Factor 6 Suppresses the Progression of Pancreatic Cancer by Upregulating Activating Transcription Factor 3
 
 
Search for Articles:
Title / Keyword
Author / Affiliation / Email
Journal
Article Type
 
 
Advanced Search
 
Section
Special Issue
Volume
Issue
Number
Page
 
Journals
JCM
Volume 12
Issue 1
10.3390/jcm12010201
jcm-logo
Submit to this Journal Review for this Journal Propose a Special Issue
► Article Menu

Article Menu

share Share announcement Help format_quote Cite question_answer Discuss in SciProfiles thumb_up
...
Endorse textsms
...
Comment
first_page
settings
Order Article Reprints
Font Type:
Arial Georgia Verdana
Font Size:
Aa Aa Aa
Line Spacing:
Column Width:
Background:
Article

Does Frenotomy Modify Upper Airway Collapse in OSA Adult Patients? Case Report and Systematic Review

by
Eduardo J. Correa
1,
Carlos O’Connor-Reina
1,
Laura Rodríguez-Alcalá
1,
Felipe Benjumea
1,
Juan Carlos Casado-Morente
1,
Peter M. Baptista
2,
Manuele Casale
3,
Antonio Moffa
3 and
Guillermo Plaza
4,*
1
Otorhinolaryngology Department, Hospital Quirónsalud Marbella, 29603 Málaga, Spain
2
Otorhinolaryngology Department, Clínica Universidad de Navarra, 31007 Pamplona, Spain
3
School of Medicine, Campus Bio-Medico University, Unit of Integrated Therapies in Otolaryngology, Fondazione Policlinico Universitario Campus Bio-Medico, 00128 Rome, Italy
4
Otorhinolaryngology Department, Hospital Universitario de Fuenlabrada, Universidad Rey Juan Carlos, 28042 Madrid, Spain
*
Author to whom correspondence should be addressed.
J. Clin. Med. 2023, 12(1), 201; https://doi.org/10.3390/jcm12010201
Submission received: 27 November 2022 / Revised: 19 December 2022 / Accepted: 23 December 2022 / Published: 27 December 2022
(This article belongs to the Section Dentistry, Oral Surgery and Oral Medicine)
Browse Figures
Review Reports Versions Notes

Abstract

:
Ankyloglossia (tongue-tie) is a condition of the oral cavity in which an abnormally short lingual frenulum affects the tongue’s mobility. Literature on the correlation between ankyloglossia and obstructive sleep apnea (OSA) is scarce. The main objective of this study was to report our preliminary experience in adult OSA patients before and after ankyloglossia treatment, using drug-induced sleep endoscopy (DISE) to evaluate the upper airway modifications resulting after treatment, and to present a systematic review of the impact of ankyloglossia and its treatment on OSA adults. We found that, after frenotomy, regarding the DISE findings, and according to the VOTE classification, two of the three patients showed an improvement in tongue level, from 2A-P (complete anteroposterior collapse) to 1ap (partial anteroposterior collapse). The third patient showed no changes in his UA after frenotomy, neither worsening nor showing improvement. Thus, the results of this study suggest that frenotomy in OSA patients with ankyloglossia could reduce tongue collapse, probably by allowing the tongue to take into the physiological position in the oral cavity. These patients should undergo speech therapy and oropharyngeal exercises prior to any surgical procedure, in order to avoid glossoptosis and to improve the quality of life and sleep apnea results.

1. Introduction

Ankyloglossia—commonly named “tongue-tie”—is a condition of the oral cavity in which an abnormally short lingual frenulum affects the tongue’s mobility by tethering it to the floor of the mouth. Its incidence is estimated to be between 3–11% of the pediatric population [1,2,3,4], being slightly more common in male subjects.
Its most recent definition, by the American Academy of Otolaryngology–Head and Neck Surgery (AAO-HNS) consensus, describes it as a “condition of limited tongue mobility caused by a restrictive lingual frenulum” [5]. During fetal development, the frenulum balances the tongue, lip muscles, and growing facial bones, before its retraction after birth, while in ankyloglossia there is a failure to recede [6,7].
The clinical presentation of ankyloglossia varies from asymptomatic to adverse consequences, such as impairing the sucking, chewing, deglutition, speaking functions, and orthodontic anomalies [8,9,10]. Moreover, this condition may contribute to oromyofacial dysfunction [6] and obstructive sleep apnea (OSA) [11].
Several methods for the clinical assessment of tongue mobility and ankyloglossia have been published by Kotlow [12], Ruffoli [9], Hazelbaker [13], Marchesan [14], or Ingram [15]. During the office examination of OSA patients, we routinely follow the “Tongue+ Protocol” [16] using both the Marchesan and Hazelbaker tests [13,14].
Although the literature on the correlation between ankyloglossia and OSA is scarce, as shown in a systematic review conducted by Chinnadurai et al. in 2015 [17], different authors have described the incidence of palatal and maxillomandibular disorders in patients with tongue-tie [10,11]. Speech therapy and surgical procedures, such as frenotomy, are currently the treatment options for patients with ankyloglossia and clinical consequences. A recent study by Yuen et al. found an independent association between low tongue mobility and a higher risk of OSA, concluding that reduced tongue mobility is associated with the occurrence and severity of OSA [18].
OSA is treated mainly with continuous positive airway pressure (CPAP) therapy, mandibular advancement devices, or surgical procedures [19,20]. Since Guimaraes et al. [21] published their results of myofunctional therapy (MFT), this approach has been presented as a promising complementary treatment modality, supported by increasing published evidence. MFT has shown a reduction in snoring, the apnea-hypopnea index (AHI), oxygen desaturation, and daytime sleepiness [22,23,24,25]. As it can be difficult for our patients to access a professional speech therapist, we recommend them to carry out a home-based therapy with oropharyngeal exercises, using a mHealth app (Airway Gym®) which has demonstrated acceptable rates of adherence and results [26,27,28,29].
Given that MFT is based on oral exercises, patients with ankyloglossia are not good candidates for this modality, as the condition presents with impaired tongue mobility that will limit its success. Thus, we suggest lingual frenulum surgery (frenotomy) as an option for these patients to improve their results. Furthermore, if the tongue can relevantly participate in the upper airway (UA) collapse in OSA, when ankyloglossia is reversed, we might expect an OSA improvement. A drug-induced sleep endoscopy (DISE) is a diagnostic tool to assess the UA of snorers and OSA patients in conditions that mimic natural sleep [30]. DISE may be used to assess such improvements.
The main objective of this study was to report our preliminary experience in adult OSA patients before and after ankyloglossia treatment using DISE to evaluate the UA modifications resulting after treatment, and to present a systematic review of the impact of ankyloglossia and its treatment on OSA adults.

2. Materials and Methods

2.1. Systematic Review

As lingual frenulum surgery has been given different names in the literature, such as frenectomy, frenotomy, frenulectomy, frenuloplasty, miofrenuloplasty, lingual frenum release, and tongue-tie surgery, we designed a systematic review using all these medical subject readings (MESH) terms and sleep apnea or snoring in adults (PRISMA-Search is provided as supplementary material).
The review followed the PRISMA guidelines [31], and the protocol was registered in PROSPERO (CRD42022360552). Databases PubMed, Cochrane, Scopus, Web of Science, Google Scholar, and ProQuest were searched (search strategy and results can be found as complementary material). Deduplication was fulfilled using SR-Accelerator® [32] and the studies selection with Rayyan® [33].
The inclusion criteria were: adult patients (16 years old or more); diagnosis of snoring and/or OSA; clinical diagnosis of ankyloglossia following a published and reproductible assessment tool/protocol; lingual frenulum surgery (no distinction of the surgical technique).
Exclusion criteria were: OSA diagnosis not confirmed by a standardized sleep test (as by questionnaires); syndromic patients with craniofacial malformations; neurological comorbidities with swallowing or speech impairment; other/s upper airway surgical procedure/s performed concurrently.

2.2. Case Report

We analyzed three cases of adult patients with a clinical diagnosis of ankyloglossia following the “Tongue+Protocol” [16] and severe OSA based on a home sleep apnea test (HSAT). Following the Marchesan protocol, patients were asked to place the tongue behind the maxillary incisors, and then open their mouth while still in contact. The first measurement of mouth opening was taken. After that, the patient was asked to open his mouth with the tongue resting down, and a second measurement was taken. A difference of 50% or more was considered a pathological lingual frenulum [14,16]. All patients were male, with no adverse nasal or maxillomandibular findings, and who had acceptable strength parameters for the tongue and buccinator measured by the Iowa Oral Performance Instrument (IOPI®) [34,35]. They performed MFT with the mHealth app Airway Gym® and presential speech therapy for the tongue-tie, with the objective being to improve tongue mobility. After showing no improvements on MFT, patients were offered a frenotomy to provide the tongue with adequate mobility. Patients agreed and signed the informed consent regarding the surgical procedure and the permission to publish an anonymized clinical record, recordings, and results.
On the day of the scheduled frenotomy, we performed a three-step procedure. First, we applied DISE (see below), then waited for the patient to wake up spontaneously and proceeded with the second step—frenotomy under local anesthesia with a collaborative patient, using cold dissection and bipolar coagulation, if required. In the third step, we performed an immediate postoperative DISE to assess any changes in the upper airway.
The DISE protocol was performed under sedation with propofol and bispectral index scale (BIS) monitoring, and the results were registered according to the VOTE classification [36] as recommended on the European position on DISE [37,38].
Three doctors trained in OSA and DISE reviewed the anonymized DISE recordings separately, without knowing which was preoperative or postoperative, and the results were recorded.

3. Results

3.1. Systematic Review Results

After removing 95 duplicates, 240 articles were screened, of which 201 were excluded by the reading of the title or abstract. We could not retrieve three of them, resulting in 36 eligible studies. Of the 36 eligible studies (Figure 1), 29 were discarded after a full-text reading for not fulfilling the inclusion and exclusion criteria: population = 20 studies (pediatric or syndromic patients); intervention = seven (other intervention, techniques comparison, medical intervention); and outcome = two (not related to sleep apnea). Finally, we selected the seven studies included in this systematic review (Table 1).

3.2. Case Report Results

Baseline data were as follows:
  • Patient number 1 was male and 54 years old, with a body mass index (BMI) of 28 kg/m2, an apnea/hypopnea index (AHI) of 31, maximum oxygen desaturation of 81%, presenting with a Friedman tongue position (FTP) [44] grade 2, and IOPI® scores: tongue (67 kPa) and buccinator (34 kPa).
  • Patient number 2 was male and 48 years old, with a BMI of 33 kg/m2, AHI 37, maximum oxygen desaturation 72%, FTP 2, and IOPI® scores: tongue (59 kPa) and buccinator (28 kPa).
  • Patient number 3 was male and 42 years old, with a BMI of 27 kg/m2, AHI 62, maximum oxygen desaturation of 85%, FTP 2, and IOPI® scores: tongue (49 kPa) and buccinator (32 kPa).
After the frenotomy, regarding the DISE findings, there were no changes in the velum, oropharynx, or epiglottis in any case, when comparing the preoperative and immediate postoperative records. According to the VOTE classification, two of the three patients showed an improvement in their tongue level, from 2A-P (complete anteroposterior collapse) to 1ap (partial anteroposterior collapse). The third patient showed no changes in his UA after frenotomy, neither worsening nor showing improvement.
Video recordings of the DISE procedures are provided as supplementary material.

4. Discussion

In 2020, the AAO-HNS published the Clinical Consensus Statement on Ankyloglossia in Children [5], hypothesizing that the “anterior tethering of the tongue serves, to some degree, to prevent posterior collapse of the tongue and that if the frenulum is released, it could lead to worsening OSA”. However, when reviewing the references in the consensus, this concept is based on two case reports [45,46].
The first case report referred was published in 1995, describing that, after induction with anesthesia and frenotomy, the patient presented with signs of UA obstruction [45]. The authors did not specify whether the muscular strength was evaluated or if the patient performed speech therapy before the frenotomy. Moreover, they did not determine which signs of UA obstruction were identified (possibly oxygen desaturation, a known complication of general anesthesia). In the same report, the authors reported that “…after surgical release, the genioglossus may not be able to generate sufficient force to prevent airway collapse…” a concept that supports our theory that muscular tone needs to be improved prior to the surgical procedure. We also consider the use of local anesthetics relevant, as general anesthesia may lead to impaired genioglossus function, as described in this referred case report. We must also consider that this procedure was performed before the standardization of DISE and knowledge of the patterns of airway collapse.
The other publication referred to by the AAO-HNS was a two-case report by Genther et al. in 2015 [46], regarding patients with a Pierre Robin sequence speech defect. These patients present with severe micrognathia and glossoptosis. We agree with the authors that the high risk of UA obstruction in this syndrome is caused by glossoptosis secondary to micrognathia. This is a specific population of a rare disease, with an incidence of 1/8500 to 1/30,000 newborns [47], and we cannot extrapolate this situation to the general population with OSA.
Our systematic review has found that there are recent publications regarding the association between ankyloglossia and OSA. The recent literature review published by Bussi et al. [41], showed that an untreated shortened lingual frenulum is associated with OSA, and also alters the craniofacial growth with respiratory consequences. The authors retrieved four articles for inclusion, in accordance with the difficulties that we found in collecting studies addressing our concern. Regarding ankyloglossia surgery, they concluded that this procedure in association with MFT can improve quality of life and snoring.
In 2016, Guilleminault et al. published a retrospective study on 150 pediatric patients [11], addressing to correlate ankyloglossia with consequent OSA, by clinical investigation and polysomnographic evaluation, concluding that ankyloglossia left untreated is associated with OSA at a later stage in life. Although it encompasses a pediatric population, the study comes to value as the authors used an objective sleep study to diagnose OSA.
The retrospective cohort published in 2019 by Zaghi et al. [39] concluded that surgical treatment with MFT is a safe procedure, and potentially effective for the treatment of snoring. Although it is a promising outcome, we must take into consideration that the study was carried out in a pediatric population, and also the evaluation of snoring was subjectively assessed by quality-of-life questionnaires, subject to the parent’s evaluation.
Baxter et al. [40] published, in 2020, a prospective study on the consequences of frenotomy in combination with MFT, finding functional improvements in speech, feeding, and sleep. Even though the results were promising, there is the need to consider that the assessment relied on parental reports. Although it can be considered reliable and feasible, we believe that objective sleep studies are necessary at the moment of comparison between different interventions and investigations. As with nasal breathing, the tongue position is important for a patent airway, and ankyloglossia modifies the UA anatomy and breathing.
In the lecture given by Professor Dr. Eric Kezirian in 2020, available on his Youtube® Channel [48], this referent in OSA noticed that ankyloglossia surgery was not an OSA treatment, and also has never been shown to affect tongue position, as no objective studies were addressing the consequences of this procedure. Despite the fact that frenotomy is not considered as an isolated treatment for OSA, it may have a role as an adjuvant for MFT, whose benefits have been demonstrated [27], to improve compliance and outcomes of oropharyngeal exercises. Furthermore, this is the first study to evaluate the effects of a frenotomy in the upper airway of patients with OSA objectively.
A case report published by Jaikumar et al. [43] in two patients, 14 and 20 years old, showed the benefits of a combination of MFT and frenotomy. Immediately after surgery, the tongue mobility was improved, but limited. It was with the practice of tongue exercises that a significant improvement was recorded.
This concept is also supported by Bargiel et al. [42]. They performed frenotomy in six patients, who were performing speech therapy before the procedure, and continued with tongue exercises in the immediate postoperative period, followed by MFT. Even though they published a step-by-step surgical technique, a so-called miofrenuloplasty, they also concluded that a predictable result requires the cooperation of a speech therapist.
However, to this date, there are no studies objectively comparing the preoperative and postoperative outcomes of a frenotomy in OSA patients. Our cases have shown that a frenotomy is able to modify the tongue’s position and thus help to prevent its collapse into the airway in two of three patients. The third one showed no changes after the procedure. These findings are the opposite of what was expected, that untethering the tongue would produce glossoptosis with a worsening of the UA collapse. As far as we are aware, this is the first study to use DISE to monitor the UA pattern of collapse in patients with OSA and ankyloglossia before and after frenotomy.
In our cases, we have identified a change in the tongue level (T level in the VOTE classification [36]) with an improvement in the pattern of airway collapse after frenotomy in two of the three patients. How this improvement occurs is not clear, but we demonstrated that untethering the tongue, in a patient with a standard or adequate muscular strength, allows it to take its place in the oral cavity, thus avoiding airway collapse at this level. To explain this effect, we must consider that the tongue is not a rigid structure, but a muscular one with certain adaptative characteristics. In patients with a normal lingual frenulum and its mobility, the tongue contacts the hard palate, adopting in its dynamic form a configuration where the retroglossal space is not invaded (Figure 2). In patients with ankyloglossia, the tongue is tied anteriorly to the floor of the mouth, and it cannot reach the hard palate, so it adapts its form by increasing its posterior volume, invading the space of the oropharyngeal airway (Figure 3).
It was hypothesized originally that untethering the tongue would provoke glossoptosis with an immediate airway collapse. Following this line of thought, if just the tethering of the tongue would be able to prevent its collapse, then we should use a simple suture technique to prevent a tongue base collapse in OSA, avoiding the increased morbidity of current techniques such as Coblator® [49,50,51] or TransOral Robotic Surgery [52,53,54].
On the contrary, in these selected patients, undertaking MFT (i.e., the lingual muscular strength was within normal parameters), releasing the tongue did not result in glossoptosis. We believe this was because of the prior efforts at improving muscular strength, so releasing the tongue helped it to take its optimal position in the oral cavity. Without restraint, the exercised tongue can work in the usual way (Figure 4).
The referred above study by Bussi et al. [41] described that, after frenotomy, there was an improvement in sleep quality due to the tongue “…being able to rest on the palate instead of resting in the floor of the mandible”. This conclusion supports our explanation of the improvement we observed here, as it was also expressed by Baxter et al. [40] that “a tongue-tie prevents the tongue from resting on the palate”.
Although the relationship between ankyloglossia and OSA is still under discussion, more evidence has been published supporting this association. In 2016, Guilleminault et al. [11] showed that a short lingual frenulum is associated with OSA. The higher risk for OSA in patients with ankyloglossia has been supported by the findings of Pia Villa et al. in 2020 [55] and the above cited literature review conducted by Bussi [41]. According to the literature correlating ankyloglossia and OSA, we currently perform a routine evaluation of the lingual frenulum and mobility in every patient [16,26,34], despite most sleep society airway evaluation guidelines do not recommend it [56].
In our experience, every patient with ankyloglossia requires mandatory MFT and speech therapy, with the objective of improving the musculature of the tongue; otherwise, it will fall into the UA when a frenotomy is performed. This recommendation is supported by several authors such as Murias et al. [57], who published a systematic review regarding the laser technique concluding that it “does not eliminate the need for myofunctional exercises and work with a speech therapist”, and Pia Villa et al. [58], who showed that their patients with OSA had poor tongue strength and benefited from MFT.
Following the current concept of performing nasal procedures (e.g., turbinoplasty and septoplasty), not for nasal benefit directly but to improve adaptation and the adherence to CPAP therapy, we consider that a frenotomy may be an effective and necessary procedure for those patients performing MFT, to improve compliance, as we have demonstrated previously [27], and the outcomes of oropharyngeal exercises.
Thus, it is important to evaluate lingual frenulum and tongue motility in the clinical practice, especially in the OSA field. As sleep surgeons, we are not presented with many patients with ankyloglossia, so it is difficult to perform a more extensive population study. However, we hope to encourage other groups to perform the described three-step procedure, as further research in larger populations will confirm or invalidate our findings.

5. Conclusions

The results of this study suggest that a frenotomy in OSA patients with ankyloglossia could reduce tongue collapse, probably by allowing the tongue to take its optimal position in the oral cavity.
These patients should undergo speech therapy and oropharyngeal exercises prior to any surgical procedure, in order to avoid glossoptosis and improve their quality of life and sleep apnea results.

Supplementary Materials

The following supporting information can be downloaded at: https://www.mdpi.com/article/10.3390/jcm12010201/s1, File S1.—Video Patient #1. File S2.—Video Patient #2. File S3.—Video Patient #3. File S4.—PRISMA-Search strategy. File S5.—PRISMA-Checklist. File S6.—Prospero protocol.

Author Contributions

Conceptualization, E.J.C. and C.O.-R.; methodology, E.J.C., C.O.-R. and G.P.; software, E.J.C. and C.O.-R.; validation, E.J.C., L.R.-A., F.B., J.C.C.-M. and C.O.-R.; formal analysis, E.J.C., C.O.-R. and G.P.; investigation, E.J.C., C.O.-R. and G.P.; resources, E.J.C. and C.O.-R.; data curation, E.J.C., C.O.-R., P.M.B. and G.P.; writing—original draft preparation, E.J.C.; writing—review and editing, E.J.C., C.O.-R., A.M., M.C. and G.P.; visualization, E.J.C., C.O.-R. and G.P.; supervision, E.J.C., C.O.-R. and G.P.; project administration, E.J.C., C.O.-R. and G.P. All authors have read and agreed to the published version of the manuscript.

Funding

This research received no external funding.

Institutional Review Board Statement

The study was conducted in accordance with the Declaration of Helsinki, and approved by our local Ethics Committee (AWGAPN-2019-01).

Informed Consent Statement

Informed consent was obtained from all subjects involved in the study.

Data Availability Statement

Not applicable.

Conflicts of Interest

Carlos O’Connor-Reina is the creator of Airway Gym app. Rest of the authors presents no conflict of interest.

References

  1. Messner, A.H.; Lalakea, M.L.; Aby, J.; Macmahon, J.; Bair, E. Ankyloglossia: Incidence and Associated Feeding Difficulties. Arch. Otolaryngol. Head Neck Surg. 2000, 126, 36–39. [Google Scholar] [CrossRef] [PubMed] [Green Version]
  2. Ballard, J.L.; Auer, C.E.; Khoury, J.C. Ankyloglossia: Assessment, Incidence, and Effect of Frenuloplasty on the Breastfeeding Dyad. Pediatrics 2002, 110, e63. [Google Scholar] [CrossRef] [PubMed] [Green Version]
  3. Klockars, T. Familial Ankyloglossia (Tongue-Tie). Int. J. Pediatr. Otorhinolaryngol. 2007, 71, 1321–1324. [Google Scholar] [CrossRef] [PubMed]
  4. O’Shea, J.E.; Foster, J.P.; O’Donnell, C.P.; Breathnach, D.; Jacobs, S.E.; Todd, D.A.; Davis, P.G. Frenotomy for Tongue-Tie in Newborn Infants. Cochrane Database Syst. Rev. 2017, 3, CD011065. [Google Scholar] [CrossRef] [PubMed]
  5. Messner, A.H.; Walsh, J.; Rosenfeld, R.M.; Schwartz, S.R.; Ishman, S.L.; Baldassari, C.; Brietzke, S.E.; Darrow, D.H.; Goldstein, N.; Levi, J.; et al. Clinical Consensus Statement: Ankyloglossia in Children. Otolaryngol. Neck Surg. Off. J. Am. Acad. Otolaryngol. Neck Surg. 2020, 162, 597–611. [Google Scholar] [CrossRef] [Green Version]
  6. Yoon, A.; Zaghi, S.; Weitzman, R.; Ha, S.; Law, C.S.; Guilleminault, C.; Liu, S.Y.C. Toward a Functional Definition of Ankyloglossia: Validating Current Grading Scales for Lingual Frenulum Length and Tongue Mobility in 1052 Subjects. Sleep Breath. 2017, 21, 767–775. [Google Scholar] [CrossRef]
  7. Walsh, J.; Tunkel, D. Diagnosis and Treatment of Ankyloglossia in Newborns and Infants: A Review. JAMA Otolaryngol. Head Neck Surg. 2017, 143, 1032–1039. [Google Scholar] [CrossRef]
  8. Wang, J.; Yang, X.; Hao, S.; Wang, Y. The Effect of Ankyloglossia and Tongue-Tie Division on Speech Articulation: A Systematic Review. Int. J. Paediatr. Dent. 2022, 32, 144–156. [Google Scholar] [CrossRef]
  9. Ruffoli, R.; Giambelluca, M.A.; Scavuzzo, M.C.; Bonfigli, D.; Cristofani, R.; Gabriele, M.; Giuca, M.R.; Giannessi, F. Ankyloglossia: A Morphofunctional Investigation in Children. Oral Dis. 2005, 11, 170–174. [Google Scholar] [CrossRef]
  10. Brożek-Mądry, E.; Burska, Z.; Steć, Z.; Burghard, M.; Krzeski, A. Short Lingual Frenulum and Head-Forward Posture in Children with the Risk of Obstructive Sleep Apnea. Int. J. Pediatr. Otorhinolaryngol. 2021, 144, 110699. [Google Scholar] [CrossRef]
  11. Guilleminault, C.; Huseni, S.; Lo, L. A Frequent Phenotype for Pediatric Sleep Apnea: Short Lingual Frenulum. ERJ Open Res. 2016, 2, 00043-2016. [Google Scholar] [CrossRef] [PubMed] [Green Version]
  12. Kotlow, L.A. Ankyloglossia (Tongue-Tie): A Diagnostic and Treatment Quandary. Quintessence Int. 1999, 30, 259–262. [Google Scholar] [PubMed]
  13. Amir, L.H.; James, J.P.; Donath, S.M. Reliability of the Hazelbaker Assessment Tool for Lingual Frenulum Function. Int. Breastfeed. J. 2006, 1, 3. [Google Scholar] [CrossRef] [Green Version]
  14. Marchesan, I.Q. Lingual Frenulum Protocol. Int. J. Orofac. Myol. Myofunctional Ther. 2012, 38, 89–103. [Google Scholar] [CrossRef]
  15. Ingram, J.; Johnson, D.; Copeland, M.; Churchill, C.; Taylor, H.; Emond, A. The Development of a Tongue Assessment Tool to Assist with Tongue-Tie Identification. Arch. Dis. Child. Fetal Neonatal Ed. 2015, 100, F344–F349. [Google Scholar] [CrossRef] [Green Version]
  16. Rodriguez-Alcalá, L.; Ignacio-García, J.; Serrano Angulo, M.S.; Casado Morente, J.C.; Benjumea Flores, F.; O’Connor-Reina, C. Tongue+ Protocol for the Diagnosis of Obstructive Sleep Apnoea in Quirónsalud Marbella Hospital. F1000Research 2022, 11, 322. [Google Scholar] [CrossRef]
  17. Chinnadurai, S.; Francis, D.O.; Epstein, R.A.; Morad, A.; Kohanim, S.; McPheeters, M. Treatment of Ankyloglossia for Reasons Other than Breastfeeding: A Systematic Review. Pediatrics 2015, 135, e1467-74. [Google Scholar] [CrossRef] [Green Version]
  18. Yuen, H.M.; Au, C.T.; Chu, W.C.W.; Li, A.M.; Chan, K.C.-C. Reduced Tongue Mobility: An Unrecognized Risk Factor of Childhood Obstructive Sleep Apnea. Sleep 2022, 45, zsab217. [Google Scholar] [CrossRef]
  19. Kim, K.B.; Movahed, R.; Malhotra, R.; Stanley, J. Management of Obstructive Sleep Apnea an Evidence-Based, Multidisciplinary Textbook, 1st ed.; Kim, K.B., Movahed, R., Malhotra, R.K., Stanley, J.J., Eds.; Springer: Cham, Switzerland, 2021; ISBN 978-3-030-54146-0. [Google Scholar]
  20. Bikov, A.; Dragonieri, S. Obstructive Sleep Apnea Epidemiology, Pathomechanism and Treatment; MDPI AG PP: Basel, Switzerland, 2020; ISBN 978-3-03936-079-6. [Google Scholar]
  21. Guimarães, K.C.; Drager, L.F.; Genta, P.R.; Marcondes, B.F.; Lorenzi-Filhoy, G. Effects of Oropharyngeal Exercises on Patients with Moderate Obstructive Sleep Apnea Syndrome. Am. J. Respir. Crit. Care Med. 2009, 179, 962–966. [Google Scholar] [CrossRef] [Green Version]
  22. Camacho, M.; Certal, V.; Abdullatif, J.; Zaghi, S.; Ruoff, C.M.; Capasso, R.; Kushida, C.A. Myofunctional Therapy to Treat Obstructive Sleep Apnea: A Systematic Review and Meta-Analysis. Sleep 2015, 38, 669–675. [Google Scholar] [CrossRef]
  23. Aiello, K.D.; Caughey, W.G.; Nelluri, B.; Sharma, A.; Mookadam, F.; Mookadam, M. Effect of Exercise Training on Sleep Apnea: A Systematic Review and Meta-Analysis. Respir. Med. 2016, 116, 85–92. [Google Scholar] [CrossRef] [PubMed] [Green Version]
  24. Ieto, V.; Kayamori, F.; Montes, M.I.; Hirata, R.P.; Gregório, M.G.; Alencar, A.M.; Drager, L.F.; Genta, P.R.; Lorenzi-Filho, G. Effects of Oropharyngeal Exercises on Snoring: A Randomized Trial. Chest 2015, 148, 683–691. [Google Scholar] [CrossRef] [PubMed] [Green Version]
  25. Mediano, O.; Romero-Peralta, S.; Resano, P.; Cano-Pumarega, I.; Sánchez-De-la-torre, M.; Castillo-García, M.; Martínez-Sánchez, A.B.; Ortigado, A.; García-Río, F. Obstructive Sleep Apnea: Emerging Treatments Targeting the Genioglossus Muscle. J. Clin. Med. 2019, 8, 1754. [Google Scholar] [CrossRef] [PubMed] [Green Version]
  26. Rodríguez-Alcalá, L.; Martínez, J.M.L.; Baptista, P.; Ríos Fernández, R.; Javier Gómez, F.; Parejo Santaella, J.; Plaza, G. Sensorimotor Tongue Evaluation and Rehabilitation in Patients with Sleep-Disordered Breathing: A Novel Approach. J. Oral Rehabil. 2021, 48, 1363–1372. [Google Scholar] [CrossRef] [PubMed]
  27. O’connor-Reina, C.; Garcia, J.M.I.; Alcala, L.R.; Ruiz, E.R.; Iriarte, M.T.G.; Morente, J.C.C.; Baptista, P.; Plaza, G. Improving Adherence to Myofunctional Therapy in the Treatment of Sleep-Disordered Breathing. J. Clin. Med. 2021, 10, 5772. [Google Scholar] [CrossRef]
  28. O’Connor-Reina, C.; Ignacio Garcia, J.M.; Ruiz, E.R.; Del Carmen Morillo Dominguez, M.; Barrios, V.I.; Jardin, P.B.; Casado Morente, J.C.; Garcia Iriarte, M.T.; Plaza, G. Myofunctional Therapy App for Severe Apnea–Hypopnea Sleep Obstructive Syndrome: Pilot Randomized Controlled Trial. JMIR mHealth uHealth 2020, 8, e23123. [Google Scholar] [CrossRef]
  29. O’Connor Reina, C.; Plaza Mayor, G.; Ignacio-Garcia, J.M.; Baptista Jardin, P.; Garcia-Iriarte, M.T.; Casado-Morente, J.C. Floppy Closing Door Epiglottis Treated Successfully with an Mhealth Application Based on Myofunctional Therapy: A Case Report. Case Rep. Otolaryngol. 2019, 2019, 4157898. [Google Scholar] [CrossRef]
  30. Carrasco-Llatas, M.; Matarredona-Quiles, S.; De Vito, A.; Chong, K.B.; Vicini, C. Drug-Induced Sleep Endoscopy: Technique, Indications, Tips and Pitfalls. Healthcare 2019, 7, 93. [Google Scholar] [CrossRef] [Green Version]
  31. Page, M.J.; McKenzie, J.E.; Bossuyt, P.M.; Boutron, I.; Hoffmann, T.C.; Mulrow, C.D.; Shamseer, L.; Tetzlaff, J.M.; Akl, E.A.; Brennan, S.E.; et al. The PRISMA 2020 Statement: An Updated Guideline for Reporting Systematic Reviews. BMJ 2021, 372. [Google Scholar] [CrossRef]
  32. Clark, J.; Glasziou, P.; Del Mar, C.; Bannach-Brown, A.; Stehlik, P.; Scott, A.M. A Full Systematic Review Was Completed in 2 Weeks Using Automation Tools: A Case Study. J. Clin. Epidemiol. 2020, 121, 81–90. [Google Scholar] [CrossRef]
  33. Ouzzani, M.; Hammady, H.; Fedorowicz, Z.; Elmagarmid, A. Rayyan-a Web and Mobile App for Systematic Reviews. Syst. Rev. 2016, 5, 210. [Google Scholar] [CrossRef] [PubMed] [Green Version]
  34. Borrmann, P.F.; O’Connor-Reina, C.; Ignacio, J.M.; Ruiz, E.R.; Alcala, L.R.; Dzembrovsky, F.; Baptista, P.; Garcia Iriarte, M.T.; Alba, C.C.; Plaza, G. Muscular Assessment in Patients with Severe Obstructive Sleep Apnea Syndrome: Protocol for a Case-Control Study. JMIR Res. Protoc. 2021, 10, e30500. [Google Scholar] [CrossRef] [PubMed]
  35. Adams, V.; Mathisen, B.; Baines, S.; Lazarus, C.; Callister, R. A Systematic Review and Meta-Analysis of Measurements of Tongue and Hand Strength and Endurance Using the Iowa Oral Performance Instrument (IOPI). Dysphagia 2013, 28, 350–369. [Google Scholar] [CrossRef] [PubMed]
  36. Kezirian, E.J.; Hohenhorst, W.; De Vries, N. Drug-Induced Sleep Endoscopy: The VOTE Classification. Eur. Arch. Otorhinolaryngol. 2011, 268, 1233–1236. [Google Scholar] [CrossRef] [PubMed]
  37. De Vito, A.; Carrasco Llatas, M.; Ravesloot, M.J.; Kotecha, B.; De Vries, N.; Hamans, E.; Maurer, J.; Bosi, M.; Blumen, M.; Heiser, C.; et al. European Position Paper on Drug-Induced Sleep Endoscopy: 2017 Update. Clin. Otolaryngol. 2018, 43, 1541–1552. [Google Scholar] [CrossRef]
  38. De Vito, A.; Carrasco Llatas, M.; Vanni, A.; Bosi, M.; Braghiroli, A.; Campanini, A.; de Vries, N.; Hamans, E.; Hohenhorst, W.; Kotecha, B.T.; et al. European Position Paper on Drug-Induced Sedation Endoscopy (DISE). Sleep Breath. 2014, 18, 453–465. [Google Scholar] [CrossRef]
  39. Zaghi, S.; Valcu-Pinkerton, S.; Jabara, M.; Norouz-Knutsen, L.; Govardhan, C.; Moeller, J.; Sinkus, V.; Thorsen, R.S.; Downing, V.; Camacho, M.; et al. Lingual Frenuloplasty with Myofunctional Therapy: Exploring Safety and Efficacy in 348 Cases. Laryngoscope Investig. Otolaryngol. 2019, 4, 489–496. [Google Scholar] [CrossRef] [Green Version]
  40. Baxter, R.; Merkel-Walsh, R.; Baxter, B.S.; Lashley, A.; Rendell, N.R. Functional Improvements of Speech, Feeding, and Sleep After Lingual Frenectomy Tongue-Tie Release: A Prospective Cohort Study. Clin. Pediatr. 2020, 59, 885–892. [Google Scholar] [CrossRef]
  41. Bussi, M.T.; de Castro Corrêa, C.; Cassettari, A.J.; Giacomin, L.T.; Faria, A.C.; Moreira, A.P.S.M.; Magalhães, I.; da Cunha, M.O.; Weber, S.A.T.; Zancanella, E.; et al. Is Ankyloglossia Associated with Obstructive Sleep Apnea? Braz. J. Otorhinolaryngol. 2021, 88, S156–S162. [Google Scholar] [CrossRef]
  42. Bargiel, J.; Gontarz, M.; Gąsiorowski, K.; Marecik, T.; Szczurowski, P.; Zapała, J.; Wyszyńska-Pawelec, G. Miofrenuloplasty for Full Functional Tongue Release in Ankyloglossia in Adults and Adolescents—Preliminary Report and Step-by-Step Technique Showcase. Medicina 2021, 57, 848. [Google Scholar] [CrossRef]
  43. Jaikumar, S.; Srinivasan, L.; Kennedy Babu, S.P.K.; Gandhimadhi, D.; Margabandhu, M. Laser-Assisted Frenectomy Followed by Post-Operative Tongue Exercises in Ankyloglossia: A Report of Two Cases. Cureus 2022, 14, e23274. [Google Scholar] [CrossRef] [PubMed]
  44. Friedman, M. Friedman Tongue Position and the Staging of Obstructive Sleep Apnea/Hypopnea Syndrome. In Sleep Apnea and Snoring: Surgical and Non-Surgical Therapy; Elsevier Ltd.: Philadelphia, PA, USA, 2009. [Google Scholar]
  45. Walsh, F.; Kelly, D. Partial Airway Obstruction after Lingual Frenotomy. Anesth. Analg. 1995, 80, 1066–1067. [Google Scholar] [PubMed]
  46. Genther, D.J.; Skinner, M.L.; Bailey, P.J.; Capone, R.B.; Byrne, P.J. Airway Obstruction after Lingual Frenulectomy in Two Infants with Pierre-Robin Sequence. Int. J. Pediatr. Otorhinolaryngol. 2015, 79, 1592–1594. [Google Scholar] [CrossRef]
  47. Giudice, A.; Barone, S.; Belhous, K.; Morice, A.; Soupre, V.; Bennardo, F.; Boddaert, N.; Vazquez, M.P.; Abadie, V.; Picard, A. Pierre Robin Sequence: A Comprehensive Narrative Review of the Literature over Time. J. Stomatol. Oral Maxillofac. Surg. 2018, 119, 419–428. [Google Scholar] [CrossRef] [PubMed]
  48. Kezirian, E. Myofunctional Therapy and Frenuloplasty Are Not Appropriate Treatments for Obstructive Sleep Apnea. Available online: https://www.youtube.com/watch?v=29fOg5eDe2c (accessed on 14 December 2022).
  49. Lin, H.-C.; Hwang, M.S.; Liao, C.-C.; Friedman, M. Taste Disturbance Following Tongue Base Resection for OSA. Laryngoscope 2016, 126, 1009–1013. [Google Scholar] [CrossRef] [PubMed]
  50. Li, H.-Y.; Lee, L.-A.; Kezirian, E.J. Coblation Endoscopic Lingual Lightening (CELL) for Obstructive Sleep Apnea. Eur. Arch. Otorhinolaryngol. 2016, 273, 231–236. [Google Scholar] [CrossRef]
  51. Shah, R.R.; Thaler, E.R. Base of Tongue Surgery for Obstructive Sleep Apnea in the Era of Neurostimulation. Otolaryngol. Clin. N. Am. 2020, 53, 431–443. [Google Scholar] [CrossRef]
  52. Lin, H.-C.; Friedman, M. Transoral Robotic OSA Surgery. Auris Nasus Larynx 2021, 48, 339–346. [Google Scholar] [CrossRef]
  53. Justin, G.A.; Chang, E.T.; Camacho, M.; Brietzke, S.E. Transoral Robotic Surgery for Obstructive Sleep Apnea: A Systematic Review and Meta-Analysis. Otolaryngol. Neck Surg. Off. J. Am. Acad. Otolaryngol. Neck Surg. 2016, 154, 835–846. [Google Scholar] [CrossRef]
  54. Kim, J.; Poole, B.; Cen, S.Y.; Sanossian, N.; Kezirian, E.J. Transoral Robotic Surgery (TORS) Versus Non-TORS Tongue Resection for Obstructive Sleep Apnea. Laryngoscope 2021, 131, E1735–E1740. [Google Scholar] [CrossRef]
  55. Villa, M.P.; Evangelisti, M.; Barreto, M.; Cecili, M.; Kaditis, A. Short Lingual Frenulum as a Risk Factor for Sleep-Disordered Breathing in School-Age Children. Sleep Med. 2020, 66, 119–122. [Google Scholar] [CrossRef] [PubMed]
  56. Correa, E.J.; Rodriguez-Alcalá, L.; Conti, D.M.; Rabino, A.; Baptista Jardin, P.M.; García-Iriarte, M.T.; Plaza, G. What Are We Missing in Adult Obstructive Sleep Apnea Clinical Evaluation? Review of Official Guidelines. Int. J. Orofac. Myol. Myofunctional Ther. 2022, 49, 1–10. [Google Scholar] [CrossRef]
  57. Murias, I.; Grzech-Leśniak, K.; Murias, A.; Walicka-Cupryś, K.; Dominiak, M.; Deeb, J.G.; Matys, J. Efficacy of Various Laser Wavelengths in the Surgical Treatment of Ankyloglossia: A Systematic Review. Life 2022, 12, 558. [Google Scholar] [CrossRef] [PubMed]
  58. Villa, M.P.; Evangelisti, M.; Martella, S.; Barreto, M.; Del Pozzo, M. Can Myofunctional Therapy Increase Tongue Tone and Reduce Symptoms in Children with Sleep-Disordered Breathing? Sleep Breath. 2017, 21, 1025–1032. [Google Scholar] [CrossRef] [PubMed]
Jcm 12 00201 g001 550
Figure 1. Systematic review identification and selection of studies on ankyloglossia and sleep apnea.
Figure 1. Systematic review identification and selection of studies on ankyloglossia and sleep apnea.
Jcm 12 00201 g001
Jcm 12 00201 g002 550
Figure 2. Normal position of the tongue.
Figure 2. Normal position of the tongue.
Jcm 12 00201 g002
Jcm 12 00201 g003 550
Figure 3. Ankyloglossia: The tongue is not in contact with the hard palate (*) and is invading the retroglossal space (**).
Figure 3. Ankyloglossia: The tongue is not in contact with the hard palate (*) and is invading the retroglossal space (**).
Jcm 12 00201 g003
Jcm 12 00201 g004 550
Figure 4. After frenotomy, the tongue takes a normal position.
Figure 4. After frenotomy, the tongue takes a normal position.
Jcm 12 00201 g004
Table
Table 1. Summary of systematic review on OSA and tongue-tie.
Table 1. Summary of systematic review on OSA and tongue-tie.
AuthorYearStudyAgeIntervention/ExpositionOutcomeAssessmentConclusion
Guilleminault et al.[11]2016Retrospective 3 to 12 yearsCorrelation ankyloglossia and obstructive sleep apneaCorrelation of ankyloglossia with sleep apneaSleep questionnaires and polysomnography in a sleep laboratoryA short lingual frenulum left untreated at birth is associated with obstructive sleep apnea at later stage.
Zaghi et al.[39]2019Retrospective cohort29 months to 79 years (n = 348)MFT + frenuloplastyMouth breathing, snoring, clenching, myofascial tensionQoL Likert scale—like questionnairesSafe and potentially effective for mouth breathing, snoring, clenching and myofascial tension.
Messner et al.[5]2020Consensus statementChildrenCase reportsCorrelation of ankyloglossia with sleep apneaConsensusThere is no evidence that ankyloglossia causes sleep apnea.
Baxter[40]2020Prospective13 months to 13 yearsMFT + frenectomySpeech, feeding, sleepQoL Likert scale—like questionnairesCombination of MFT with frenectomy shows functional improvements in speech, feeding, and sleep.
Bussi et al.[41]2021Systematic reviewChildrenIs ankyloglossia associated with obstructive sleep apnea?An untreated shortened lingual frenulum at birth is associated with sleep apnea.Systematic review Lingual frenuloplasty associated with myofunctional therapy is effective in the treatment of snoring.
Bargiel et al.[42]2021Prospective18 to 37 yearsMFT + miofrenuloplastyTongue mobility, swallowing, speech, snoring, neck muscle tensionTongue mobility (Tongue range motion ratio) and patient referred symptom reportMiofrenuloplasty is and advanced, but effective and highly predictable procedure.
Jaikumar et al.[43]2022Case report14 and 20 year oldMFT + laser-assisted frenectomyTongue mobilityTongue mobilityCombination of MFT with frenectomy shows benefits and better results.
Disclaimer/Publisher’s Note: The statements, opinions and data contained in all publications are solely those of the individual author(s) and contributor(s) and not of MDPI and/or the editor(s). MDPI and/or the editor(s) disclaim responsibility for any injury to people or property resulting from any ideas, methods, instructions or products referred to in the content.

Share and Cite

MDPI and ACS Style

Correa, E.J.; O’Connor-Reina, C.; Rodríguez-Alcalá, L.; Benjumea, F.; Casado-Morente, J.C.; Baptista, P.M.; Casale, M.; Moffa, A.; Plaza, G. Does Frenotomy Modify Upper Airway Collapse in OSA Adult Patients? Case Report and Systematic Review. J. Clin. Med. 2023, 12, 201. https://doi.org/10.3390/jcm12010201

AMA Style

Correa EJ, O’Connor-Reina C, Rodríguez-Alcalá L, Benjumea F, Casado-Morente JC, Baptista PM, Casale M, Moffa A, Plaza G. Does Frenotomy Modify Upper Airway Collapse in OSA Adult Patients? Case Report and Systematic Review. Journal of Clinical Medicine. 2023; 12(1):201. https://doi.org/10.3390/jcm12010201

Chicago/Turabian Style

Correa, Eduardo J., Carlos O’Connor-Reina, Laura Rodríguez-Alcalá, Felipe Benjumea, Juan Carlos Casado-Morente, Peter M. Baptista, Manuele Casale, Antonio Moffa, and Guillermo Plaza. 2023. "Does Frenotomy Modify Upper Airway Collapse in OSA Adult Patients? Case Report and Systematic Review" Journal of Clinical Medicine 12, no. 1: 201. https://doi.org/10.3390/jcm12010201

Note that from the first issue of 2016, this journal uses article numbers instead of page numbers. See further details here.

Article Metrics

Back to TopTop