Keywords
Myofunctional therapy, OSA, protocol, muscles upper airway, Iowa Oral Performance Instrument
Myofunctional therapy, OSA, protocol, muscles upper airway, Iowa Oral Performance Instrument
The aetiology of OSA (obstructive sleep apnoea) is multifactorial and includes anatomical and physiological factors. The upper airway dilator muscles are crucial for maintaining pharyngeal patency and may contribute to the incidence of this medical condition.1 OSA patients have been shown to have sensory-motor deficits located in the muscles of the upper respiratory airways.2,3 These deficits are associated with apraxia, hypotonia, and changes in muscle fibre type from Type I to Types IIa and IIb, leading to early fatigue of the UA (upper airway) muscles.4
The diagnosis and severity of OSA are based on the Apnoea-Hypopnoea Index (AHI)5: 5 to 15 in one hour, it is defined as mild OSA; 15 to 30 is moderate OSA; and more than 30 is considered severe OSA.
According to the latest international consensus document on obstructive sleep apnoea, the objectives of OSA treatment are aimed at resolving signs and symptoms of the disease, restoring sleep quality, normalising the AHI, maximising oxyhaemoglobin saturation, lowering the risk of complications, and reducing the costs of the condition. In addition, a multidisciplinary therapeutic approach is recommended, in which all the medical, surgical or physical options available for the treatment of OSA must be complementary and not exclusive.6
Therefore, with this protocol we would like to underline the importance of a systematised evaluation performed by the specialists involved in a Sleep Unit.
The Tongue+ protocol involves the following steps (Figure 1).
Hospital Quirón Salud Marbella.
A detailed medical history focusses on the symptoms, risk factors and comorbidities which are most frequently associated with OSA, as well as a physical examination, weight-height measurement, calculation of the body mass index (BMI) and questionnaires to evaluate somnolence and quality of life.
Patients who meet the suspicion criteria for OSA would be offered a diagnostic test to be performed via a polysomnography (PSG) or a respiratory polygraphy (RP), with this decision being made according to the clinical features of each patient. The diagnostic examination will measure the Apnoea-Hypopnea Index (AHI) per hour of sleep, the desaturation index per hour of sleep and CT 90 (percentage of sleep time when the patient has a saturation lower than 90%), and leg movements, while restless legs syndrome will be assessed, and the number of snores will be measured by means of a microphone. In the case of the polysomnography, in addition to the cardiorespiratory variables, the REM and non-REM sleep phases, the number of cycles in each phase, sleep latency, sleep efficiency and microarousals will be measured. This test will show us whether the patient has apnoea, its degree of severity and the possible component of alveolar hypoventilation.
After this first assessment, the pulmonologist will refer the patient to the Otorhinolaryngology Department (ENT) to assess the anatomy and functionality of the upper airways and, in the event of detecting obesity, they will be transferred to the Nutrition and Dietetics Unit. We have known for many years that a reduction in body mass index (BMI) of between 5-10% significantly reduces the AHI.7
This step involves an anatomical assessment which will include performing a fibreoptic nasolaryngoscopy, an oropharyngoscopy, and a Müller manoeuvre.
In addition, intercurrent pathology which may aggravate the condition (pharyngolaryngeal reflux, cervical and laryngeal masses, etc.) will be studied and treated sequentially.
For the functional assessment of the UA muscles, the patient will undergo a lingual stereognosis test and a muscular force test with a tongue digital spoon and an IOPI (Iowa Oral Performance Instrument).
Lingual frenulum will be assessed using the Hazelbaker8 and/or Marchesani tests9,10 and possible apraxia of speech will be evaluated using the OMES-modified questionnaire (Extended data42).
⇒ If the patient has an anatomical condition, for example, septal deviation or Grade III-IV tonsils, an operation aimed at re-permeabilising the upper airway will be indicated.
⇒ If they have a functional disorder only of a muscle tone type, oropharyngeal exercises will be offered with the Airway Gym application, as well as advice from a specialised trainer. If this motor disorder is more complex (secondary apraxia of speech), the patient will be referred to a speech therapist.
⇒ If no anatomical factors are found from the beginning, we recommend performing a drug-induced sleep endoscopy (DISE) in which we assess the various therapeutic options (surgery, myofunctional therapy, mandibular advancement devices or continuous positive airway pressure (CPAP).
Once the patient shows OSA reflected in the sleep study, the pulmonologist will refer the patient to the Nutrition and Dietetics Department if BMI >30, and to the otolaryngologist to assess the functionality and anatomy of the patient's UA to optimise CPAP adherence in cases which meet the current criteria for CPAP6:
AHI: ≥15/h with:
- Excessive daytime sleepiness (Epworth >10)11
- Episodes of nighttime suffocation
- Intense snoring
- Insomnia
- Morning headache
- Impaired professional or academic performance
- High blood pressure (especially if it is resistant or refractory)
In patients without an indication for CPAP due to an AHI ≥15/h without the criteria described above, AHI 5-15/h, or patients who refuse treatment even with an indication, sleep specialists will evaluate alternative treatments.
At our unit, the ENT specialist will be the second level of specialised care, no less important, and oversees evaluating the treatments to optimise CPAP and/or the different treatments available for sleep apnoea: (ENT or maxillofacial surgery, mandibular advancement device (MAD), positional therapy, myofunctional therapy).
In an average time of 15-20 minutes, we must offer a complementary diagnosis so that the patient understands their sleep problem and offer them the guarantee of finding the best individual treatment.
To do this, we carry out a systematic appointment which consists of the following assessments:
1) Questionnaires:
In the case of treatment with CPAP, it will be carried out according to the patient's symptoms. To expedite the appointment, the patient will previously fill out the following questionnaires:
- Epworth questionnaire on daytime sleepiness.10
- NOSE survey to assess the symptoms of nasal obstruction. A result of >50% would indicate the need for surgical treatment.12 In patients who undergo several nasal operations with a negative impact on their respiratory quality, we will assess a possible hyperventilation syndrome,11 counting the number of breaths taken per minute, and the Nijmegen questionnaire.13
- Pittsburgh Sleep Quality Index.14
- Berlin Questionnaire to assess the risk of sleep apnea.15
- OMES-modified questionnaire for the identification of a myofunctional disorder.16
2) Personal history:
We collect the main cardiovascular diseases and jobs with a high risk of accidents due to drowsiness.
3) Anatomical examination:
4) Cervical circumference: a circumference of >41 cm in men and >39.5 cm in women suggests a high probability of developing OSA.17
5) Facial skeletal assessment: ANGLE CLASSIFICATION (1907)18:
The position of the upper first molar with respect to the lower one is assessed during the bite.
• Class I: NORMAL OCCLUSION. Both molars contact (A normal overbite of about 2 mm remains in front) (less than 20% of the population).
• Class II: PROGNATHISM. The lower first molar is behind the upper one (protruding jaw). Concave or long face.
• Class III: PROGNATHISM. The lower first molar is anterior to the upper one (jaw forward). Convex or short face.
6) Temporomandibular joint dysfunction (TMJD)
It is important to identify temporomandibular joint dysfunction in sOSA patients with an indication for a MAD, as there are devices available which simultaneously treat sOSA and disc displacement with TMJD reduction.19
7) Oral cavity and oropharynx:
- Partial or total edentulism would contraindicate the use of a MAD and lingual retention appliances could be chosen.
- Lip sealing: At the appointment, we examine lingual sealing and tongue position in the upper incisor area (palatal ridges) at rest and swallowing. A dysfunctional swallow would include tongue thrust or tongue interposition (clinical criterion of atypical swallowing). The presence of an open mouth will indicate a patient with oral respiration that translates into a functional and craniofacial development problem.20
- Assessment of the palate, uvula and pillars if considering pharyngoplasty surgery, tonsil size (I-IV).
- Friedman classification: Observation of the size and position of the tongue and palate is used to classify patients into stages according to Friedman, where Grade I refer to the possibility of visualising the hard and soft palate, uvula, pillars and tonsils. Grade II allows visualisation of the uvula but not the tonsils. Grade III prevents the uvula from being seen and Grade IV allows visualisation of the hard palate only.
- Lingual frenulum: The presence of a lingual frenulum is a limiting factor in the success of myofunctional therapy. The lingual frenulum can be examined according to various protocols. One of them is the Marchesani protocol (the one most used in the adult population), where the patient is asked to place their tongue behind the maxillary incisors and open their mouth without lifting it. This mouth opening is measured and the patient is then asked to open their mouth fully with their tongue down. If there is a difference of more than 50% between the two measurements, that patient has a pathological lingual frenulum.21
8) Fibreoptic nasolaryngoscopy (FNL): we will assess septum deviations, hypertrophy of the inferior turbinates, intranasal masses or polyps, epiglottis, larynx and, above all, basilingual hypertrophy (BLH) with the Friedman Grades classification (2016)22:
- I: None to minimal
- II: Slight: <50% filling vallecula
- III Moderate: >50% effacement of vallecula
- IV Severe: unable to visualise the epiglottis
- In addition, we use the FNL to assess the situation of the UA with snoring: tonsillar collapse, vibration of the veil, and - with the Müller manoeuvre23 - the level of retropalatal or retrolingual collapse. Another new concept which we introduced in the assessment of OSA is the presence of indirect signs in the ENT examination of laryngopharyngeal reflux where its correct treatment may affect an improvement in multidisciplinary treatment.24
The success of our unit is favoured by the simultaneous study of the anatomical and functional problem. We recommend periodic measurements of the muscle tone of the lingual muscles and the perioral muscles with the IOPI (Iowa Oral Performance Instrument) as a way of identifying the hypotonic patient most susceptible to improvement with myofunctional therapy.25,26
The main measurements of the IOPI focus on assessing the strength of the lingual muscles and the strength of the perilabial muscles. This is conducted after the compression of a balloon probe connected to the device which the patient puts into their mouth. To measure the maximum anterior lingual force, the patient is asked to make a maximum compression with their tongue against the balloon resting on the papilla. This value would correspond to the tone of the genioglossus muscle. Three measurements are taken, with the patient resting one minute between each one and the highest value obtained taken as a reference. For the buccinator muscles, the balloon is placed between the gingival mucosa and the cheek, and the patient is asked to make a contraction with it. The value obtained would correspond to the tone of the buccinator muscle. The measurements are obtained in kilopascals (kPa). There are tables which reference the values obtained with the normal population, which allow the patient to know the situation of their musculature at the time of the test and where it should ideally be for their age and sex.27 It has been shown that patients with sleep-disordered breathing present lower-than-average values for this test and that an increase in its values also translates into an improvement in sleep events.28
In addition to the IOPI, our team designed the tongue digital spoon (TDS) as a substitute tool for the IOPI due to its high cost (Figure 2). The TDS is a handheld instrument with spoon that can be found on online shopping platforms. It consists of a handle where the Tare and Hold buttons are located. Pressing the Hold button gives us the highest set value, equivalent to the IOPI peak pressure. To carry out the measurements, the spoon is inverted and a 1 cm2 circular sticker is placed on the back, obtaining a marked circumference. To measure tongue strength, the patient holds the spoon by the handle and, with their elbow resting on a flat surface, brings the spoon closer to their tongue at an elbow angle of approximately 30°. The patient must tare the device by pressing the Tare key, marking 0.0 g, and then presses the marked circumference with the tip of their tongue. Once done, with the index finger of the hand holding the handle, the patient presses the Hold button. This test is performed entirely by the patient to avoid movements with the spoon which may interfere with the results. The normality values compared with the IOPI have been published.29 As with the IOPI, three measurements are taken, with the patient resting one minute between each one and the highest value obtained taken as a reference.
We identify a severe myofunctional problem at <100 mg/cm2. To achieve adequate muscle tone values, patients must achieve greater than 400 mg/cm2.
Once a myofunctional problem has been identified, we will recommend oropharyngeal exercises to the patient with the Airway Gym mobile application.30,31
Once a patient has been diagnosed with a functional problem and myofunctional therapy has been prescribed with the application, we refer them to a speech therapy unit to perform an OMES-modified questionnaire, see the functionality of the lingual frenulum with the Hazelbaker questionnaire, and correct possible errors when doing exercises.
In addition, this appointment assesses suspicions of secondary lingual praxis which would make it difficult to use the application and perform the lingual stereognosis test. We have shown that the use of the Airway Gym® application produces improvements in the sensory-motor function of the tongue in patients with sleep-related breathing disorders, due to the continuous stimulation of the brain based on proprioceptive training required to localise the responses when performing the exercises.32
Recently, we have been encouraging feedback from all our patients, so that the patient is aware of and forms part of the chronicity of their disease.
We first provide a measure of upper airway tone with IOPI and the tongue digital spoon (TDS), after performing a drug-induced sleep endoscopy (DISE) which gives the patient a visual concept of the behaviour of their muscles during sleep. Depending on the type of collapse detected during the DISE, we recommend certain exercises with our app, and we review this patient monthly for new IOPI and TDS measurements. If the measurements obtained by the IOPI and the TDS have not improved, it probably indicates a poor performance of the exercises, so the patient will be asked to repeat them at an appointment and will probably need a correction. With our Airway Gym app, patients can directly contact their 'coach' about the accuracy of the exercises. In some cases, at the request of the patients, we repeat the DISE to confirm anatomical changes after performing oropharyngeal exercises.32
We perform a drug-induced sleep endoscopy in all patients who (i) reject CPAP and request alternative treatments; (ii) patients who undergo nasal surgery as a complementary study of their sleep disorder and as a control of treatment with myofunctional therapy; and above all, (iii) have sarcopenia and do not present significant anatomical alteration.
The DISE is performed in the operating room. In order to show it to the patient at the next appointment, we use a microphone recorder to record snoring/apnoea episodes and to subsequently verify the VOTE34 rating among several ENTs. The patient is sedated using two sprays of xylocaine in each nostril to prevent stimulation of the fibreoptic nasoscope. The anaesthetic infusion is administered intravenously with propofol, starting at 200 ml/hour and quadrupled when the patient has snoring episodes. The resulting figure is used as the hourly infusion rate in ml/hour.33
We follow a systemic procedure which entails assessing the velum, oropharynx, tongue base, epiglottis (VOTE)34 classification after documenting the patient's first three episodes of apnoea and, subsequently, performing chin-lift manoeuvres and the mandibular advancement or march Esmarch manoeuvre35 when the patient is in the supine decubitus position. Then we assess the improvement or other in the left and right lateral decubitus position and repeat chin-lift and Smarch manoeuvres.
Our experience in providing an online version of the video of a DISE with its corresponding explanation implies 80% higher adherence to a myofunctional treatment than when it is not performed.
We have presented our multidisciplinary vision for the diagnosis and treatment of the public health problem known as obstructive sleep apnoea (OSA).
This protocol sets out to systematise the work of Sleep Units to raise patient awareness and let them have a say in the most favourable type of therapy.
The importance of an anatomical and functional assessment of the upper airway helps the patients understand better, resulting in greater therapeutic adherence. This is why we recommend myofunctional therapy (MT) and DISE as a diagnostic and treatment strategy for OSA.
In the study by O'Connor et al., the Airway Gym app includes MT exercise education for severe sleep apnoea patients, helping to improve the AHI and upper airway muscle tone.25 This is the first trial in the literature which shows significant correlations between the increase in tone, measured by the IOPI, and improvements in AHI in patients with severe OSAHS.
Therefore, at our unit we offer DISE as a pre- and post-treatment tool with myofunctional therapy after 90 sessions, in order to achieve more feedback among patients and offer future treatment options for severe OSA, which is the greatest treatment-related challenge in sleep apnoea, especially in patients with poor tolerance to CPAP.
However, in the recently published review on the role of myofunctional therapy in sleep-related breathing disorders,36 the authors conclude that the available evidence shows a positive effect of MT in reducing sleep apnoea, measured by polysomnography and clinical variables (including snoring). There is no evidence of the usefulness of MT to treat upper airway resistance syndrome,37,38 the duration of MT effects, or with respect to which MT protocol is best. Despite these gaps in knowledge, the available evidence suggests that MT is a safe form of treatment.28
Given this, our experience shows that we achieved effectiveness after 90 treatment sessions, and that patients with anatomical changes in the DISE could be candidates for other types of treatment such as MAD, pharynx/palate surgery, chin rest, etc.
Patients with good tolerance to CPAP and an improvement in AHI levels with MT will follow a long-term muscle training treatment until they achieve optimal levels of positive pressure with the lowest AHI. If it is worse, we will assess speech therapy re-education, dietary control in the event of weight gain, and another DISE.
In addition, we have incorporated new strategies for the diagnosis and treatment of OSA on the recent association of OSA and gastroesophageal reflux disease (GERD), especially when proton pump inhibitors are not effective and non-acid reflux is suspected, detected in the DISE or a basic ENT examination. For this, we use the Stanford University Reflux Symptom Index (RSI)39 and the Reflux Findings Score (RFS),40 both prepared by Belafsky et al. in 2001 and 2002. Given this suspicion, we will assess the existence of possible gas reflux. For its diagnosis, we will request a 24-hour multichannel intraluminal impedance, with the treatment being carried out with sodium alginate 500 mg twice a day/orally for six months.41
We have the first DISE-assessed case in literature of severe epiglottitis due to GERD,22 conditioning obstruction of the UA and moderate OSA, which, after the correct diagnosis and treatment of reflux, significantly decreased AHI levels. Therefore, the correct treatment of GERD due to acid or gas reflux is essential for the control of OSA.
Recently, thanks to Guilleminault's work on lingual apraxia2 in child and adult OSA, we have incorporated the study of lingual praxis and stereognosis in certain patients with OSA. Patients with oral respirators without a justifiable obstructive anatomical cause of the UA (obesity, retrognathia, severe tonsillar hypertrophy or septal deviation) would correspond to the percentage of hypotonic patients with poor efficiency of the oropharyngeal musculature. Therefore, the use of IOPI, TDS, DISE and speech therapy assessment of possible apraxia and lingual stereognosis significantly help the treatment of OSA with MT due to a poor muscular response as the pathophysiology of OSA. We have the Airway Gym application, which has shown improvements in the sensory-motor function of the tongue, due to continuous stimulation of the brain based on proprioceptive training to locate the responses when performing the exercises.21
This protocol sets out to establish a full and common assessment of sleep-related breathing disorders from the point of view of the anatomy and functionality of the upper airway to guarantee the long-term treatment of the different phenotypes of OSA patients. We have incorporated new diagnostic and treatment strategies for OSA to guarantee continuous and multidisciplinary assessment and improve the quality of life of patients.
Open Science Framework: Tongue+ protocol for the diagnosis of obstructive sleep apnoea in Quirónsalud Marbella hospital. https://doi.org/10.17605/OSF.IO/4ADRP.42
This project contains the following extended data:
- Tongue protocol.doc (containing the modified OMES questionnaire)
- Multidisciplinary approach (algorithm included in the Protocol)
Data are available under the terms of the Creative Commons Zero “No rights reserved” data waiver (CC0 1.0 Public domain dedication).
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Is the rationale for developing the new method (or application) clearly explained?
Yes
Is the description of the method technically sound?
No
Are sufficient details provided to allow replication of the method development and its use by others?
Partly
If any results are presented, are all the source data underlying the results available to ensure full reproducibility?
No
Are the conclusions about the method and its performance adequately supported by the findings presented in the article?
Partly
Competing Interests: No competing interests were disclosed.
Reviewer Expertise: Sleep apnea, obstructive sleep apnea
Is the rationale for developing the new method (or application) clearly explained?
Partly
Is the description of the method technically sound?
Partly
Are sufficient details provided to allow replication of the method development and its use by others?
Partly
If any results are presented, are all the source data underlying the results available to ensure full reproducibility?
No
Are the conclusions about the method and its performance adequately supported by the findings presented in the article?
Partly
Competing Interests: No competing interests were disclosed.
Reviewer Expertise: Sleep Medicine
Alongside their report, reviewers assign a status to the article:
Invited Reviewers | ||
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Version 1 17 Mar 22 |
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