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Protocol of Muscular Assessment in Patients With Severe Obstructive Sleep

Apnea Syndrome. A Case-Control Study (Preprint)

Article in JMIR Research Protocols · May 2021

DOI: 10.2196/30500

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 JMIR RESEARCH PROTOCOLS Borrmann et al

Protocol

Muscular Assessment in Patients With Severe Obstructive Sleep

Apnea Syndrome: Protocol for a Case-Control Study

Paz Francisca Borrmann1*, BSc; Carlos O'Connor-Reina2,3*, MD, PhD; Jose M Ignacio4,5*, MD, PhD; Elisa Rodriguez
Ruiz4,5*, RNC; Laura Rodriguez Alcala2,3*, MD, PhD; Florencia Dzembrovsky1*, BSc; Peter Baptista6, MD, PhD;
Maria T Garcia Iriarte7, MD, PhD; Carlos Casado Alba8*, MDS; Guillermo Plaza9,10,11*, MD, PhD
1
Phonoaudiology Section, Otorhinolaryngology Department, Hospital Italiano de Buenos Aires, Buenos Aires, Argentina
2
Otorhinolaryngology Department, Hospital Quironsalud Marbella, Marbella (Malaga), Spain
3
Otorhinolaryngology Department, Hospital Quironsalud Campo de Gibraltar, Palmones, Cadiz, Spain
4
Pulmonology Department, Hospital Quironsalud Marbella, Malaga, Spain
5
Pulmonology Department, Hospital Quironsalud Campo de Gibraltar, Palmones, Cadiz, Spain
6
Otorhinolaryngology Department, Clinica Universitaria de Navarra, Pamplona, Spain
7
Otorhinolaryngology Department, Hospital Universitario Virgen de Valme, Sevilla, Spain
8
School of Medicine, Clinica Universitaria de Navarra, Pamplona, Spain
9
Otorhinolaryngology Department, Hospital Universitario Sanitas la Zarzuela, Madrid, Spain
10
Otorhinolaryngology Department, Hospital Universitario Fuenlabrada, Madrid, Spain
11
School of Medicine, Universidad Rey Juan Carlos I, Madrid, Spain
*
these authors contributed equally

Corresponding Author:
Carlos O'Connor-Reina, MD, PhD
Otorhinolaryngology Department
Hospital Quironsalud Marbella
Avda Severo Ochoa 22
Marbella (Malaga)
Spain
Phone: 34 952774200
Email: [email protected]

Abstract
Background: Myofunctional therapy is currently a reasonable therapeutic option to treat obstructive sleep apnea-hypopnea
syndrome (OSAHS). This therapy is based on performing regular exercises of the upper airway muscles to increase their tone
and prevent their collapse. Over the past decade, there has been an increasing number of publications in this area; however, to
our knowledge, there are no studies focused on patients who can most benefit from this therapy.
Objective: This protocol describes a case-control clinical trial aimed at determining the muscular features of patients recently
diagnosed with severe OSAHS compared with those of healthy controls.
Methods: Patients meeting set criteria will be sequentially enrolled up to a sample size of 40. Twenty patients who meet the
inclusion criteria for controls will also be evaluated. Patients will be examined by a qualified phonoaudiologist who will take
biometric measurements and administer the Expanded Protocol of Orofacial Myofunctional Evaluation with Scores (OMES),
Friedman Staging System, Epworth Sleepiness Scale, and Pittsburgh Sleep Quality Index questionnaires. Measures of upper
airway muscle tone will also be performed using the Iowa Oral Performance Instrument and tongue digital spoon devices.
Evaluation will be recorded and reevaluated by a second specialist to determine concordance between observers.
Results: A total of 60 patients will be enrolled. Both the group with severe OSAHS (40 patients) and the control group (20
subjects) will be assessed for differences between upper airway muscle tone and OMES questionnaire responses.
Conclusions: This study will help to determine muscle patterns in patients with severe OSAHS and can be used to fill the gap
currently present in the assessment of patients suitable to be treated with myofunctional therapy.
Trial Registration: ISRCTN Registry ISRCTN12596010; https://www.isrctn.com/ISRCTN12596010
International Registered Report Identifier (IRRID): PRR1-10.2196/30500

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(JMIR Res Protoc 2021;10(8):e30500) doi: 10.2196/30500

KEYWORDS
myofunctional therapy; sleep apnea; sleep disordered breathing; speech therapy; phenotype; sleep; therapy; protocol; muscle;
assessment; case study; exercise; airway; respiratory

(OMES), involving functional exploration of all of the

Introduction stomatognathic functions to obtain a score, this has proven to
Background be inferior in patients with OSAHS compared with controls
[16,17]. A myofunctional therapist uses this evaluation to
Obstructive sleep apnea-hypopnea syndrome (OSAHS) is a improve the examined items that are in deficit and subsequently
significant public health issue characterized by repetitive performs specific exercises to improve them. However, the
episodes of airway obstruction during sleep associated with OMES test is based on subjective evaluations, contains many
snoring, sleep fragmentation, daytime sleepiness, and increased items, and is difficult to reproduce. In our opinion, a more
cardiovascular risk [1,2]. It is well established that the most concise, objective, and reproducible evaluation is required. This
effective treatment for OSAHS is continuous positive airway opinion stems from our experience of measurements with the
pressure (CPAP) [3], which has variable patient compliance. Iowa Oral Performance Instrument (IOPI) of the genioglossus
CPAP virtually eliminates OSAHS and snoring, reduces daytime muscle and the orbicular muscle [18,19]. Together with
sleepiness, and improves subjective sleep quality [3,4]. measurement of the motor tone of the genioglossus muscle using
The etiology of OSAHS is multifactorial, including anatomical a tongue digital spoon (TDS) [20], these simple measurements
and physiological factors. The upper airway dilator muscles are may provide patients with information about their condition,
crucial for maintaining pharyngeal patency and may contribute serve as therapy response parameters, and objectively transmit
to the incidence of this medical condition [5,6]. results between professionals, which can also be based in
electronic health facilities [21].
Other treatments for OSAHS include a mandibular advancement
device (MAD), conventional surgery, CO2 or radiofrequency Aim
laser, or hypoglossal nerve stimulation. There is also some The aim of this study is to evaluate the muscle patterns of
evidence for pharmacological treatments with oxybutynin and patients with severe OSAHS. The use of the OMES protocol
atomoxetine, which are currently showing promising results can be complemented by the values obtained through the IOPI
[7]. Clinical trials have been carried out with theophylline, and the TDS.
acetazolamide, and desipramine to reduce the collapse of the
upper airway, but without clear effectiveness [8,9]. Objectives
The main objective is to evaluate the function of the
Myofunctional therapy is a treatment applied to patients with stomatognathic musculature of patients with OSAHS by using
orofacial myofunctional disorders that can interfere with the the OMES protocol, TDS, and IOPI.
development or functioning of orofacial structures and functions
[10]. Reviews of studies on myofunctional therapy show benefits The secondary objectives are to: (1) use this protocol to evaluate
by promoting changes in dysfunctional muscles of the upper whether there are differences between the muscles of patients
airway [11]; therefore, this treatment has been proposed to with OSAHS and healthy controls; (2) use the IOPI to measure
reduce the severity of OSAHS and associated symptoms in tongue strength and resistance with the genioglossus and
adults [12]. Myofunctional therapy also has potential to promote buccinator muscle tone, and evaluate whether there are
a decrease in the Apnea-Hypopnea Index (AHI), reduce snoring differences from those of healthy patients; and (3) use the TDS
[13], and improve quality of life [14]. In addition, it can be to measure tongue pressure and evaluate whether there are
considered as an adjuvant therapy and an intervention strategy differences from healthy patients.
to support CPAP adherence [15].
However, it is currently unknown which patients are the best
Methods
candidates for myofunctional therapy. Although there are Design
instruments available for patient selection such as the Expanded
We designed a prospective controlled quasiexperimental pilot
Protocol of Orofacial Myofunctional Evaluation with Scores
study on patients with severe OSAHS (Figure 1).

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Figure 1. Flow chart of the study process. IOPI: Iowa Oral Performance Instrument; OMES: Orofacial Myofunctional Evaluation with Scores; OSAHS:
obstructive sleep apnea-hypopnea syndrome; TDS: tongue digital spoon.

disease, or a history of prior orofacial muscle rehabilitation

Scope of the Study therapy and any prior apnea treatment that may modify the study
This study will involve patients diagnosed and/or treated at the results (surgery, MAD, CPAP) will be excluded.
Pneumology and Otorhinolaryngology Departments of
Quirónsalud Hospital in Marbella (Malaga, Spain) or Hospital Sample Size and Sampling Procedure
Campo de Gibraltar (Palmones, Cadiz, Spain) where the study The effectiveness of use of the OMES protocol in the evaluation
will also be performed. of patients with moderate to severe apnea-hypopnea syndrome
will be evaluated from data previously published in studies
Study Population using this protocol. Following the literature review, patients
This study will include patients diagnosed with sleep will be recruited. The sample size will be 60 subjects (40 in the
apnea-hypopnea at the participating hospitals and who agree to experimental group and 20 in the control group). The sample
participate in the project. size was calculated using the XLSTAT statistical software for
Excel.
Inclusion Criteria
For cases, adults (aged 18-75 years) who have received a The variables that we are going to measure in all patients are
diagnosis of severe OSAHS (AHI>30) without previous reflected in the data collection table shown in Multimedia
experience of the condition and not undergoing treatment owing Appendix 1 using SPSS Info software, including age, sex,
to different circumstances, who had not undergone any previous weight, height, ethnicity, BMI, abdominal circumference (at
treatment for OSAHS, and signed the informed consent form the level of the navel), neck circumference (using a flexible tape
will be included. around the most prominent part while the patient is standing
with their arms by their sides, head erect, and eyes looking
For controls, adults with adequate sleep hygiene, no complaints ahead), IOPI measurement of tongue strength and the buccinator
of snoring or daytime sleepiness, and scoring <7 points on the muscle, AHI, nighttime oxygen desaturation index, lowest
Epworth Sleepiness Scale will be included. overnight oxygen saturation levels, digital spoon measurement
Exclusion Criteria of tongue strength, and OMES protocol.
Cases and controls alike with a cognitive or neurological deficit, A series of questionnaires will be applied to both groups:
inability to answer questionnaires, severe alcohol abuse, Friedman Staging System, Epworth Sleepiness Scale, and
presence of craniofacial malformations, active neoplastic Pittsburgh Sleep Quality Index (see Multimedia Appendix 2).

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An information sheet and information consent will also be TDS Evaluation

provided and signed by patients. Finally, the tone of the tongue muscles will be measured with
Procedures a digital spoon, taking three measurements and using the highest
obtained. A digital spoon is a kitchen tool used to estimate the
Experimental Design weight of food. To develop the TDS, we used the Soehnle
A myofunctional evaluation of patients diagnosed with OSAHS Cooking Star Digital Measuring Spoon with graduation from
will be performed in the same week as the polysomnography 0.1 grams to 500 grams (ID ID20005876833). This is a handheld
is performed. During this evaluation, patients will be blindly instrument with a spoon that can be found on online shopping
examined by a speech therapist and their examination will be platforms, consisting of a handle where the “tare” and “hold”
recorded on video for subsequent evaluation. buttons are located. Pressing the “hold” button helps to obtain
the highest tared value, equivalent to the IOPI peak pressure.
The patient will sit 1 meter away from the camera with their To carry out the measurements, the spoon is inverted and a
feet flat on the floor and their back supported by the backrest.
The camera (Sony CCD-TRV138 Handycam camcorder) will 1-cm2 circular sticker is placed on the back to obtain a marked
be placed on a tripod at face and shoulder height. circumference. To measure tongue strength, the subject holds
the spoon by the handle and, with their elbow resting on a flat
Evaluation with the OMES protocol (see Multimedia Appendix surface, brings the spoon closer to the tongue with an elbow
3) will then take place, based on the analysis of the following angle of approximately 30°. The subject must then tare the
parameters: (1) appearance/posture; (2) mobility; and (3) device by pressing the “hold” key, marking 0.0 grams. The
functions, including respiration, deglutition, and mastication. subject then presses on the marked circumference with the tip
As a result of this evaluation with the already validated protocol, of their tongue. Once done, with the index finger of the hand
the higher the score, the more normal the patient’s that is holding the handle, the subject presses the “hold” button.
stomatognathic system. This test is performed entirely by the subject to avoid
movements on the spoon that may interfere with the results [20].
IOPI Evaluation
The recordings and the data obtained will also be analyzed by
The IOPI objectively measures maximum tongue and lip another blinded examiner.
strength. Tongue strength is assessed by measuring the
maximum pressure exerted when a person presses a disposable, Distribution of Hospital Visits
standard-sized tongue bulb against the roof of the mouth. Lip
Selection Visit
strength is assessed by measuring the maximum pressure when
the bulb is located between the cheek and closed teeth, and the A patient diagnosed with OSAHS at a pulmonology laboratory
patient contracts the buccinator muscle without biting the bulb. by means of an initial sleep study (with measurement of baseline
Reference values have been obtained for a healthy population AHI, nighttime oxygen desaturation index, and the lowest
and are provided by the manufacturer [18]. nighttime oxygen saturation figures) will be evaluated with
respect to the inclusion and exclusion criteria and then informed
Tongue strength is measured by obtaining maximal tongue about the study. After reading the information and having any
elevation pressures. The patient is instructed to “place this bulb doubts resolved, the patient will accept and sign the inform
in your mouth on the midline of your tongue and push it against consent form in duplicate, taking one copy home.
the roof of your mouth as hard as you can.” To maximize
standard placement, the examiner demonstrates how to place One-off Visit
the bulb along the central groove of the tongue blade. Previous The patient will be evaluated by the speech therapist and fill in
research [18] indicates that maximal measures of tongue strength the sleepiness questionnaires, following which the OMES
and endurance are best assessed with an unconstrained jaw; protocol will be applied and the evaluation will be carried out
participants will be encouraged to gently rest the incisors on with the IOPI and the TDS.
the tubing of the IOPI bulb. Each test lasts 7-10 seconds, and
all participants will be given verbal encouragement from the Statistical Analysis
examiner for the entire test. The test will be performed three The data of the study variables will be collected in a database
times by each participant, with a brief rest of about 30 seconds created for the development of the study. In the statistical
between each test while the examiner records the peak pressure analysis, the sample will be described through the distribution
obtained. The highest pressure across the three trials will be of frequencies for the categorical variables, and through
used as the maximal isometric pressure instead of the mean measures of central tendency and dispersion such as the mean
pressure, which has been used by other researchers [22]. Given (SD) and median (IQR) for quantitative variables. The
the high correlation between the average and maximal pressure distribution of quantitative variables will be examined using
and that both are similarly related to oral-phase swallowing the Kolmogorov-Smirnov test. Bivariate analysis of the
function, the use of maximal pressure is more efficient in a association between categorical variables will be carried out
clinical setting because it requires no calculation. using the χ2 test or Fisher exact test when necessary. The
Subsequently, the muscle tone of the genioglossus muscle and differences between quantitative variables will be analyzed
the buccinator muscle are evaluated, taking three measurements using the Student t test or analysis of variance for two or more
of each and using the highest value. samples, respectively, and nonparametric tests (Mann-Whitney
or Kruskal-Wallis test) will be used if the variables to be
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analyzed do not follow the normal distribution. The possible processed according to Regulation (EU) 2016/679 of the
correlations between the OMES protocol evaluation and the European Parliament and of the Council (April 27, 2016) on
IOPI values and TDS will be assessed using the Spearman rank the protection of natural persons with regard to the processing
correlation coefficient. The consistency and stability of the intra- of personal data and on the free movement of such data, and
and interrater measurements (reliability coefficient) will be Organic Law 3 (December 5, 2018) on the protection of personal
determined using the split-half method. The level of statistical data and guarantee of digital rights.
significance will be set at P<.05.
Usefulness and Applicability
Ethical Aspects The selection criteria for patients with OSAHS may improve
The Research Ethics Committee of the Hospital Provincial de depending on which therapy is more suitable.
Málaga reviewed and approved the protocol and the informed
consent model for the patients (AWGAP-2021-02). Before Results
performing any of the procedures specified in the study protocol,
the participating subjects will have signed and dated the The authors plan to publish the study findings in a peer-reviewed
informed consent form approved by the Research Ethics journal and at topic-related conferences (to be determined at a
Committee. later date). All listed authors or contributors are compliant with
guidelines outlined by the International Committee of Medical
Access to Data and Protection of Data Obtained from Journal Editors for author inclusion in a published work.
the Study Furthermore, to support research transparency and
To guarantee the confidentiality of the study data, the original reproducibility, we will share the deidentified research data
data will be stored at the hospital and only researchers and the after publication of the study results. We will also share the
Research Ethics Committee will have access. deidentified data on Figshare, a repository where users can make
all of their research outputs available in a citable, shareable,
This project will be carried out following the guidelines of the and discoverable manner. To date, we have collected data for
Declaration of Helsinki (Fortaleza, Brazil, 2013) [23] and the 20% of the planned sample. The timeline for data collection to
Standards of Good Clinical Practice. Personal data will be completion of the study is given in Figure 2.
Figure 2. Summary of the results.

The patients’ main anomaly will be determined by their

Discussion responses to the OMES questionnaire, and with the measures
Projected Significance of the upper airway muscles using the IOPI and TDS.

Although Eckert [24] proposed that one of the phenotypes The TDS is a simple, reproducible, and affordable method to
responsible for initiating sleep apnea is the hypotonic pattern measure the muscle tone of the tongue for this patient group.
in 2016, studies have yet to be performed to confirm this We have pioneered the use of this domestic tool to allow patients
proposal. It is well known that patients with this phenotype can to obtain immediate feedback of their success in performing
benefit from treatment with a hypoglossal pacemaker, myotonic myofunctional therapy exercises. In our opinion, the OMES
medication such as desipramine, and myofunctional therapy. questionnaire is a suitable tool to make a diagnosis for these
We consider that our study will help to identify certain patients patients, but can only be performed by specialized
with severe OSAHS according to distinct anthropometric and phonoaudiologists and requires considerable time. In most
myofunctional features from those of conventional patients (ie, countries, there is a lack of phonoaudiologists and the demand
individuals with obesity or with anatomical issues such as big on their time means that consultations are short. We contend
tonsils). Following our experience using the IOPI [18], we that we can provide this information with the assistance of the
consider that patients with a normal BMI, neck and bell IOPI and TDS that do not require any special training.
circumference, and anatomy of the upper airway will show no The information provided by these two instruments can be
relationship with the position of the tongue or the soft palate. correlated with the information obtained by the OMES. In this
We previously demonstrated that Friedman stage is independent case, these two measures are fast, simple, reproducible, and
from the tone of the muscle of the genioglossus as measured provide objective information to both the patient and examiner.
by the IOPI [18].

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Limitations is obliged to correct the deficits simultaneously with the use of

One of the limitations of this study is that although we are going CPAP. We are strongly opposed to any surgery in patients where
to identify patients according to their singular characteristics, there is a demonstrated myofunctional disorder.
we are not going to be able to demonstrate the effectiveness of Comparison With Prior Work
the exercises performed with myofunctional therapy.
No previous work directed at this matter has been performed
Theoretically, these patients should improve with myofunctional
for effective comparison.
therapy, a hypoglossal pacemaker, or inotropic medication. We
intend to perform that study as a continuation of this proposed Conclusions
trial. This study will help determine the muscle patterns in patients
Our main concern is that if these patients have myofunctional with severe OSAHS and may be used to fill the current gap in
disorders (ie, low OMES, IOPI, or TDS scores), the therapist the identification of patients suitable to be treated with
myofunctional therapy.

Authors' Contributions
PFB, COR, JMI, and ER made significant contributions to data collection, and writing and editing assistance. FD, CCA, and PFB
assisted with translation. GP and MTGI made significant contributions in the design of the study.

Conflicts of Interest
None declared.

Multimedia Appendix 1
PowerPoint presentation with database.
[PPTX File , 3292 KB-Multimedia Appendix 1]

Multimedia Appendix 2
Information sheets; informed consent; and Pittsburg, Friedman, and Epworth questionnaires.
[PDF File (Adobe PDF File), 6303 KB-Multimedia Appendix 2]

Multimedia Appendix 3
OMES questionnaire.
[PDF File (Adobe PDF File), 249 KB-Multimedia Appendix 3]

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Abbreviations
AHI: Apnea-Hypopnea Index
CPAP: continuous positive airway pressure
IOPI: Iowa Oral Performance Instrument
MAD: mandibular advancement device.
OMES: Orofacial Myofunctional Evaluation with Scores
OSAHS: obstructive sleep apnea-hypopnea syndrome.
TDS: tongue digital spoon

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Edited by G Eysenbach; submitted 20.05.21; peer-reviewed by B Piknova; comments to author 11.06.21; revised version received
11.06.21; accepted 11.06.21; published 06.08.21
Please cite as:
Borrmann PF, O'Connor-Reina C, Ignacio JM, Rodriguez Ruiz E, Rodriguez Alcala L, Dzembrovsky F, Baptista P, Garcia Iriarte
MT, Casado Alba C, Plaza G
Muscular Assessment in Patients With Severe Obstructive Sleep Apnea Syndrome: Protocol for a Case-Control Study
JMIR Res Protoc 2021;10(8):e30500
URL: https://www.researchprotocols.org/2021/8/e30500
doi: 10.2196/30500
PMID: 34115605

©Paz Francisca Borrmann, Carlos O'Connor-Reina, Jose M Ignacio, Elisa Rodriguez Ruiz, Laura Rodriguez Alcala, Florencia
Dzembrovsky, Peter Baptista, Maria T Garcia Iriarte, Carlos Casado Alba, Guillermo Plaza. Originally published in JMIR Research
Protocols (https://www.researchprotocols.org), 06.08.2021. This is an open-access article distributed under the terms of the
Creative Commons Attribution License (https://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution,
and reproduction in any medium, provided the original work, first published in JMIR Research Protocols, is properly cited. The
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copyright and license information must be included.

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