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Obstructive sleep apnea syndrome (OSAS) is a common disorder which involves upper airway collapse during sleep. Positional Obstructive Sleep Apnoea Syndrome is a sub-type of OSAS characterized by obstructive apneas mainly in the supine sleeping position. Mandibular advancement devices (MAD) are recommended as first-line therapy for mild to moderate OSA or as second-line treatment for severe OSA for patients who do not tolerate or respond to CPAP. A case report of severe positional OSAS (AHI/h 34.7) is presented. The clinical case was studied by CBCT head and neck scan and nocturnal polysomnography and treated with a mandibular advancement device, positional therapy, dietary adjustments and instructions for proper sleep hygiene. After the treatment the patient’s AHI decreased to 5.1 and his general health improved. The MAD appliance represents a valid therapeutic alternative in subjects with severe OSAS who do not tolerate CPAP. A combined approach to the pathology can improve the patient’s overall health and quality of life.
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G. Laganà
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Annali di stomatologia
computed tomography
Clinical management of a severe case of positional obstructive sleep apnea syndrome
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Original Article
2022-08-09
2022-08-09
2019-12-01
2023-12-04
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Ann Stomatol (Roma)
G. Laganà
2019
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Department of Systems Medicine, University of Rome ‘Tor Vergata’, Via Montpellier 1, 00133 Rome, Italy; UniCamillus - Saint Camillus International University of Health and Medical Sciences via Sant’ Alessandro 8, 00131 Rome, Italy
1971-1441
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computed tomography
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Clinical management of a severe case of positional obstructive sleep apnea syndrome
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                                                                                                  Original article


Clinical management of a severe case of positional
obstructive sleep apnea syndrome


Venza Nicolò1                                               tem control over ventilation (3). The presence of some
Malara Arianna1                                             syndromes also predisposes the onset of sleep disor-
Palmacci Daniel1                                            ders (4). Brain dysfunctions might also manifest, such as
Laganà Giuseppina1,2                                        excessive daytime sleepiness and lack of concentration
                                                            and these are the most common causes of traffic ac-
1
    Department of Systems Medicine, University of Rome      cidents (5). The prevalence of OSA has been estimat-
    ‘Tor Vergata’, Via Montpellier 1, 00133 Rome, Italy     ed to be 14% of men and 5% of women (6). Despite
2
    UniCamillus - Saint Camillus International University   its prevalence, OSA is still an underdiagnosed medical
    of Health and Medical Sciences via Sant’ Alessandro     condition, and more than 85% of patients with clinically
    8, 00131 Rome, Italy                                    significant OSA are never diagnosed (7). The presence
                                                            of sleep disturbances is often associated with the pres-
                                                            ence of sleep bruxism (8). Positional Obstructive Sleep
Corresponding author:                                       Apnoea Syndrome (POSAS) is a sub-type of OSAS in
Nicolò Venza                                                which obstructive apneas occur mainly in the supine
Department of Systems Medicine, University of Rome          sleeping position (more than 50% of apnea episodes).
‘Tor Vergata’, Via Montpellier 1, 00133 Rome, Italy.        Polysomnography is the gold standard diagnostic test
Email: venza.nicolo@gmail.com                               for the diagnosis of OSA in whom there is a concern for
                                                            OSA based on a comprehensive sleep evaluation that
Abstract                                                    include informations on sleep posture (9). The apnea-hy-
                                                            popnea index (AHI) is used to diagnose OSAS, and it is
Obstructive sleep apnea syndrome (OSAS) is a com-           calculated as the number of apneas and hypopneas per
mon disorder which involves upper airway collapse           hour during sleep (10). Mandibular advancement devic-
during sleep. Positional Obstructive Sleep Apnoea           es (MAD) are recommended as first-line therapy for mild
Syndrome is a sub-type of OSAS characterized by             to moderate OSA or as second-line treatment for severe
obstructive apneas mainly in the supine sleeping po-        OSA for patients who do not tolerate or respond to CPAP
sition. Mandibular advancement devices (MAD) are            (11). Specifically, MADs interact with the mandible to
recommended as first-line therapy for mild to moder-        the tongue, pharyngeal dilator muscles, and indirectly
ate OSA or as second-line treatment for severe OSA          the soft palate. By moving the mandible forward, these
for patients who do not tolerate or respond to CPAP.        structures that make up the lumen of the oropharynx are
A case report of severe positional OSAS (AHI/h 34.7)        extended forward as well, thereby increasing the up-
is presented. The clinical case was studied by CBCT         per airway space (12). According to the literature Cone
head and neck scan and nocturnal polysomnography            Beam Computed Tomography (CBCT) with its low effec-
and treated with a mandibular advancement device,           tive radiation dose and low scanning time represents an
positional therapy, dietary adjustments and instruc-        effective technique for a 3D complete evaluation, when
tions for proper sleep hygiene. After the treatment the     utilizing a large field of view protocol, for a comprehen-
patient’s AHI decreased to 5.1 and his general health       sive head and neck evaluation (13).
improved. The MAD appliance represents a valid
therapeutic alternative in subjects with severe OSAS        A case report of severe OSAS in a middle age man,
who do not tolerate CPAP. A combined approach to            studied by CBCT head and neck scan and nocturnal pol-
the pathology can improve the patient’s overall health      ysomnography and treated by customized and titrabled
and quality of life.                                        MAD, is presented.

Keywords: OSA, Mandibular advancement devices,
polysomnography, computed tomography.                       Clinical case and therapeutic approach
                                                            A 41 years old Caucasian male, M.M., from the Depart-
Introduction                                                ment of Orthodontics referred by the Neurophysiopa-
                                                            thology Department of Tor Vergata Hospital, with a chief
Obstructive sleep apnea syndrome (OSAS) is a com-           complaint of snoring, numerous episodes of nocturnal
mon and treatable disorder, which involves upper airway     apnea and daytime sleepiness. The patient had histo-
collapse during sleep and results in intermittent hypox-    ry of arterial hypertension and depression. The patient
aemia and sleep fragmentation (1,2). This disorder is       was undergoing pharmacological treatment for his de-
the result of a complex interaction between anatomic        pressive state. He had previously been treated for sleep
factors, sleep-related factors, and central nervous sys-    apnea with CPAP. This treatment had not shown satis-
Annali di Stomatologia 2019; X (1-4): 17-21                                                                              17
     Clinical management of a severe case of positional obstructive sleep apnea syndrome

     factory results due to the patient’s poor tolerance to the        ADVANCER (Leone orthodontic products, Sesto Fior-
     therapy. The patient’s body mass index was 27,13 (over-           entino, Firenze, Italy). Bimaxillary device with lateral
     weight) with a neck circumference of 45 cm, and he was            connectors composed of a tube and piston mechanism
     being treated by a nutritionist to improve alimentation           with the possibility of adjusting the protrusion very accu-
     and lose weight. The patient was also instructed in prop-         rately, with 7 mm of maximum elongation. Vestibular pin
     er sleep hygiene to improve his habits. The upper airway          for elastic bands were added to the device to prevent
     appeared normal in ear-nose-throat and laryngoscopy               the patient from keeping his mouth open while sleeping
     examinations. The patient’s neck and tongue muscles               (Fig. 1a).
     were enlarged. Moreover the patient shows a reduced               The MAD was periodically adjusted during monthly
     patency of the nasal airways passages due to hypertro-            checks in order to obtain the maximum possible mandib-
     phy of the nasal turbinates. The patient does not have            ular advancement in the absence of patient discomfort.
     any voluptuous habit. At the oral examination the patient         After six months of using MAD and NS Sleep Positioner,
     showed right and left full class I with dentoalveolar bi-         the patient’s apnea improved the patient reports a clear
     retrusion and increased OVB, signs of dental bruxism              improvement in symptoms (ESS = 1), restful sleep, ab-
     and normal-shaped arches. The patient besides shows               sence of asthenia upon awakening, absence of snoring
     macroglossia (Mallampati grade 3), normal size of the             and / or apneic episodes. The patient does not show
     uvula and soft palate, type 1 tonsillar grading. Epworth          any kind of discomfort, absence of signs or symptoms
     sleepiness score (ESS) was 11. CBCT examinations was              about temporomandibular dysfunctions. The second pol-
     performed with NewTom VGi EVO unit (NewTom 3G, QR                 ysomnographic examination with MAD in situ revealed
     s.r.l.; AFP Imaging, Elmsford, NY, USA). The 3D recon-            only nine apnea episodes and no one were central. The
     struction and evaluation of the upper airways showed an           patient’s AHI decreased to 5.1 (Fig. 2). Moreover, the
     anatomical narrowing of the hypopharynx which predis-             patient’s overall health improved owing to dietary adjust-
     poses the collapse of the airways during sleep. Written           ments. The patient appeared satisfied with the results
     consent was obtained from the patient.                            of the therapy. A second TCCB with MAD in situ scan
     Upon polysomnographic examination, the subject was                showed an increased dimension of the hypopharynx
     diagnosed with positional preference OSAS. During the             (Fig. 3).
     test, he had 141 apneas, 1 of which were central. The
     patient’s apnea hypopnea index (AHI) was 34.7, a value            Discussion
     that meets the criterion for severe sleep apnea.
     Because the patient refused CPAP therapy due to dis-              The patient was treated using a mandibular advance-
     comfort and considering the strong positional compo-              ment device, positional therapy, dietary adjustments and
     nent of apneas, treatment options for reduction of the            instructions for proper sleep hygiene according with the
     OSAS included a customized and titrabled MAD (Figure              existing evidenced-based clinical practice guidelines
     1a) and a NS Sleep Positioner (Advanced Brain Moni-               (14, 15). The patient presented in the basal PSG an AHI
     toring, Carlsbad, CA) composed of a plastic device fas-           of 34.7 events/h. Before starting the therapy with the
     tened on the back of the neck with an adjustable rubber           MAD, the patient tried CPAP for a few nights, once it
     strap secured by a magnetic clasp (Fig. 1b).                      is the primary treatment indication for severe sleep ap-
     Customized MAD was obtained through the Gorge                     nea. Nevertheless, he abandoned it as the discomfort
     Gauge at maximum advancement without muscular pain                was the no-compliance reason. After titrating the MAD,
     for patients. The MAD chosen was the TELESCOPIC                   the results of the control polysomnographic, with the


      a




      b




                                                                  Figure 1. (a) Intraoral views of the MAD appliance with vestibular
                                                                  elastic bands (b) Night Shift Positioner.


18                                                                                          Annali di Stomatologia 2019; X (1-4): 17-21
V. Nicolò et al.


  a




  b




Figure 2. (a) Polysomnographic report before treatment (b) after treatment.




Annali di Stomatologia 2019; X (1-4): 17-21                                   19
     Clinical management of a severe case of positional obstructive sleep apnea syndrome


      a                                                              b




     Figure 3. (a) 3D CBCT initial reconstruction and evaluation of the upper airways (b) with MAD in situ.




     oral appliance in situ, showed an objective decrease in             position is not only important to avoid the simple fall
     the rates of respiratory obstructive events. The full night         of the soft tissues by gravity but also for the effects
     PSG showed an AHI = 5.1 events/h. Patients with severe              on chest pressure. Joosten S.A. and colleagues (23)
     OSA have an increased cardiovascular risk, showing in-              demonstrated a 13% functional residual capacity (FRC)
     creased prevalence of hypertension, stroke, arrhythmia,             decrease in the OSA group when moving from lateral to
     and aortic events, although these findings are not always           supine. This reduction of FRC influences upper airway
     consistent (16-18). Without CPAP adherence, they must               collapsibility with resultant changes in upper airway
     be treated with alternative therapies. As suggested in the          pressure (24). The reduction in lung volume causes an
     literature, the efficacy of the OSAS therapy must be eval-          increase in critical pressure (Pcrit) which influences the
     uated in relation to the patient’s compliance (19). The             collapse of the upper airways causing the apnea event.
     main objective of MAD therapy is to reduce or normalize             Additionally, a reduced lung volume increases the ring
     the AHI. There are some predictors that help dentists in            gain of the ventilatory control system during reduced
     the attempt to identify which patients will benefit more            sleep lung oxygen and carbon dioxide deposits which
     from the treatment. However, the predictors are still not           can then function of respiratory instability in the supine
     fully clinically reliable (20). In the present case the pa-         position of sleep (25).
     tient was 41 years old, overweight and higher AHI. This             In this case report, the patient was placed on a weight-
     outcome was not compatible with some strong predic-                 loss program with a nutritionist in order to manage the
     tors such as lower AHI, lower age, lower BMI and higher             BMI. Although the effect of weight-loss on OSAS out-
     mandible protrusion (21). Despite this, the patient had an          comes is not well studied, weight management is rou-
     excellent response to MAD therapy.                                  tinely recommended. A decrease in BMI is associated
     In this study the patient wore a NS Sleep Positioner in             with improved metabolic outcomes in obese subjects
     order to avoid supine position. Van Kesteren et al. (22)            and should be routinely encouraged notwithstanding is
     demonstrated that, especially in POSA patients, the                 potential benefits regarding OSAS (26). Chirinos JA et
     head position has a fundamental role in the genesis                 al. (27) in his study has shown that CPAP, associated
     of apnoeas. Simply with head rotation, the anteropos-               with a weight-loss program, increases insulin sensitivity
     terior collapse of the tongue due to gravity forces can             and reduces serum triglyceride levels, but no improve-
     be avoided and the airway can be stiffened. The body                ment was observed with CPAP treatment alone, which


20                                                                                          Annali di Stomatologia 2019; X (1-4): 17-21
V. Nicolò et al.

raises the important issue of the usefulness of adding               12. Kerbrat A, Vinuesa O, Lavergne F, Aversenq E, Graml A,
weight-loss programs to CPAP treatment in order to                       Kerbrat JB, Trost O, Goudot P. Clinical impact of two types
                                                                         of mandibular retention devices - A CAD/CAM design and a
improve the cardiovascular risk factor profile of obese
                                                                         traditional design - On upper airway volume in obstructive
patients with OSA. The same considerations can cer-                      sleep apnea patients. J Stomatol Oral Maxillofac Surg. 2021
tainly also be made in the association between MAD                       Jun 9:S2468-7855(21)00125-7
and weight loss. A meta-analysis that included four ran-             13. Alsufyani NA, Al-Saleh MA, Major PW. CBCT assessment
domized controlled trials assessing the effect of inten-                 of upper airway changes and treatment outcomes of ob-
                                                                         structive sleep apnoea: a systematic review. Sleep Breath.
sive lifestyle interventions on weight change and AHI
                                                                         2013;17(3):911–923.
reduction found that a weight loss of 14 kg was associ-              14. Epstein LJ, Kristo D, Strollo PJ Jr, et al.; Adult Obstruc-
ated with a reduction in AHI of 16 events/h (28).                        tive Sleep Apnea Task Force of the American Academy of
                                                                         Sleep Medicine . Clinical guideline for the evaluation, man-
                                                                         agement and long-term care of obstructive sleep apnea in
Conclusion                                                               adults. J Clin Sleep Med. 2009;5(3):263-276.
                                                                     15. Ranieri S, Laganà G, Cretella Lombardo Elisabetta, Cozza
The mandibular advancement device associated with
                                                                         P. Le problematiche respiratorie nel sonno in età adulta: il
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tions for proper sleep hygiene improved the polysomno-               16. Kimura, H.; Ota, H.; Kimura, Y.; Takasawa, S. Effects of In-
graphic parameters in a case of severe OSA. The MAD                      termittent Hypoxia on Pulmonary Vascular and Systemic Dis-
appliance represent a valid therapeutic alternative in                   eases. Int. J. Environ. Res. Public Health 2019, 16, 3101.
subjects with severe OSAS who do not tolerate CPAP.                  17. Kohler, M.; Pitcher, A.; Blair, E.; Risby, P.; Senn, O.; Forfar,
                                                                         C.; Wordsworth, P.; Stradling, J.R. The impact of obstructive
A combined approach to the pathology can improve the
                                                                         sleep apnea on aortic disease in Marfan’s syndrome. Respi-
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                                                                     18. Laganà G, Venza N, Malara A, Liguori C, Cozza P, Pisano
                                                                         C. Obstructive Sleep Apnea, Palatal Morphology, and Aortic
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Annali di Stomatologia 2019; X (1-4): 17-21                                                                                                 21
