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* From the Respiratory Research Unit (Ms. Skinner, and Drs. Kingshott and Taylor), Dunedin School of Medicine, University of Otago, Dunedin; and Tom McKendrick Sleep Laboratory (Mr. Jones), Dunedin Hospital, Dunedin, New Zealand.
Correspondence to: Margot A. Skinner, MPhEd, School of Physiotherapy, University of Otago, PO Box 56, Dunedin, New Zealand; e-mail: mskinner{at}gandalf.otago.ac.nz
| Abstract |
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Design: Subjects received treatment with CMSC or nCPAP each for 1 month in random order. The study was analyzed on an intention-to-treat basis.
Setting: Tom McKendrick Sleep Laboratory, Dunedin Hospital.
Participants:Ten adult subjects with mild-to-moderate OSA (apnea-hypopnea index [AHI], 24 ± 13/h slept [mean ± SD]) completed the study.
Interventions: The CMSC was designed to prevent mandibular movement and hold the head in slight extension, thus preventing the postural changes that might contribute to OSA. Positioning of the CMSC was confirmed by an externally applied cervical range of motion (CROM) instrument and by cephalometry. Subjects were carefully instructed in the use of each device and completed a symptom diary. After 1 month, subjects underwent polysomnography with each of the allocated devices in situ, and symptom questionnaires were administered.
Measurements and results:Treatment success (AHI
10/h slept) with CMSC was achieved in only 2 of 10 subjects, partial success (AHI > 10/h to
15/h slept) was achieved in 2 subjects, and in 6 of 10 subjects there was no benefit. In contrast, treatment success was achieved in 7 of 10 subjects receiving nCPAP. Mean AHI was 29.4 ± 13.4/h at baseline, 26.9 ± 17.2/h slept with CMSC, and 9.9 ± 8.0/h slept with nCPAP (p = 0.001). No significant differences in sleep architecture or sleep efficiency were achieved using nCPAP compared to CMSC. The efficacy of the CMSC in maintaining the desired head position was confirmed by cephalometry and the CROM instrument.
Conclusions: Our results, although negative, provide important evidence that control of head and neck posture, perhaps adopted as a second-line treatment, is not helpful in the management of OSA. It appears that other anatomic and physiologic factors have a dynamic overriding influence on upper airway closure compared to simple skeletal relationships.
Key Words: cephalometry continuous positive airway pressure neck collar obstructive sleep apnea posture
| Introduction |
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During sleep, the influence of gravity and loss of muscle tone further enhance narrowing of the upper airway,9 thus increasing the risk of apneic and hypopneic events. Given that the upper airway comprises both rigid and flexible structures, the actual site of obstruction may be multiple and variable.10 Thus the anatomic positions of the hyoid bone, mandible, head, and neck may be critical.11
In awake subjects, airway resistance is higher in patients with OSA than in normal subjects; as the mandible is progressively advanced forward, airway resistance decreases.12 . Similarly, it has been shown that whereas neck flexion significantly increases airway resistance in patients with OSA, maintaining the head in extension preserves airway patency.13 Hiyama et al14 have shown in normal subjects that using a cervical collar, jaw closure and thus airway patency may be maintained during sleep.
Against this background, therapeutic devices such as the mandibular advancement splint have been designed to protrude the mandible forward, and with it the tongue, as a means of maintaining airway patency during sleep. This results in a significant reduction in the severity of OSA.15 16 17 The success of this approach raises the possibility that other conservative treatments, designed to prevent posteroinferior movement of the mandible and tongue, may have similar beneficial effects.
Taken together, these data suggest that a device such as a cervicomandibular support collar (CMSC) designed to prevent downward displacement of the mandible as well as maintaining the head in slight extension during sleep may be beneficial in OSA. The aim of this cross-over study was to compare the effect of CMSC therapy in mild-to-moderate OSA with standard therapy, nasal continuous positive airway pressure (nCPAP).18
| Materials and Methods |
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Inclusion criteria were mild-to-moderately severe OSA and apnea-hypopnea index (AHI) of 10 to 60/h of total sleep time. Exclusion criteria were a medical history of cardiovascular, neurologic, or psychological disorders affecting sleep; coexisting sleep disorders; and known cervical or temporomandibular joint dysfunction and/or pain.
Study Design
Subjects received treatment with a CMSC or nCPAP each for 1 month in random order. The CMSC device and the nCPAP masks were selected from a range of sizes as appropriate for the individual. Each subject was carefully instructed in the use of each device, and was given a symptom diary at the beginning of each treatment period. At the end of each treatment period, subjects underwent full-night polysomnography with the allocated device in situ, and symptom questionnaires were administered. At the completion of the study, a clinical evaluation of outcomes was undertaken, and each subject was offered long-term therapy depending on results and treatment preference.
CMSC
The CMSC (Fig 1
) was a modified Headmaster Collar (Symmetric Designs; Salt Spring Island, BC, Canada). The CMSC was designed to retain the head in the natural head position (NHP)19
and prevent jaw opening during sleep. The collar comprised a fabric-covered, semicircular acrylic and latex ring, which was molded to fit under the mandible and rest on the upper third of the manubrium sterni. It was held in place by a fabric-covered foam strap with self-adhesive attachments. A chin piece supported the lower jaw in the closed position with the teeth held in light occlusion, and an anterior support tube was attached between the mandible and sternal portions to provide rigid support and prevent neck flexion beyond the NHP.
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nCPAP
At the commencement of the nCPAP treatment arm, each subject was allocated an autotitrating machine (Autoset T, Sullivan; ResMed Ltd; North Ryde, NSW, Australia) and an Ultra Mirage nasal mask. Minimum CPAP pressure was set at 4 cm H2O, and maximum CPAP pressure was set at 20 cm H2O. The Autoset T permits automatic titration of the continuous positive airway pressure (CPAP) required to maintain airway patency while asleep. After 3 to 5 nights of monitoring, data were downloaded from the Autoset T to a computer in the sleep laboratory. The 95th percentile pressure was then used to set the pressure on a ResMed S6 Elite (ResMed Ltd) CPAP machine, which was then given to each subject for the remainder of the trial period.
Cephalometric Radiographs
Cephalograms were obtained at the commencement of the CMSC study arm with and without the CMSC in situ, with each subject lying supine as for the NHP,19
using the orthoposition method.20
A fluid level was used to register the head posture, and care was taken to limit changes in cervical position during the introduction of the cephalostats. A chain suspended from the film cassette registered the true vertical plane. Subjects were instructed to hold the teeth in light occlusion, and cephalograms were obtained at the end of expiration.
All supine lateral cephalometric landmarks were coordinated with the true horizontal and vertical lines. Conventional bony landmarks (Fig 2 , top) were marked on cephalometric tracing film and digitized with a reflex metrograph. Reference lines and angles (Fig 2 , bottom) were measured to the nearest 0.01 mm or degree.
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The definitions of apnea and hypopnea used in our study to calculate the respiratory disturbance index/AHI followed the American Association of Sleep Medicine guidelines.1
A thermistor was used to define airflow characteristics. An apnea was defined as cessation of airflow lasting at least 10 s. Changes in thoracoabdominal movements were used to score hypopneas, given that the thermistor is unable to quantify the reduction in airflow, or to distinguish between obstructive and central events. A reduction in any of these signals by
50% from a rolling baseline, and persisting for
10 s, or a reduction in thoracoabdominal movement with an accompanying oxygen desaturation of at least 3% and/or associated with an arousal, defined the significant respiratory events.
Outcome Measures
Sleep habits, daytime sleepiness, snore symptoms, exercise capacity, general health, and medication use at presentation were recorded using standardized questionnaires: a general health questionnaire, the Medical Outcomes Study Short-Form 36 (SF-36),25
the Functional Outcomes of Sleep Questionnaire (FOSQ),26
the Scottish National Sleep Laboratory symptom questionnaire (SQ),27
and the Epworth sleepiness scale (ESS).28
Throughout the study, sleep habits, the period of time wearing the device at night, subjective levels of sleepiness/alertness on waking, daytime hypersomnolence, side effects, and any other relevant symptoms were recorded in the self-reported diary. At the end of each period, subjects completed an adverse effects questionnaire, which comprised 19 adverse effects commonly associated with nCPAP therapy such as a dry mouth, sore jaw, and poor mask fit. These were also applied to the CMSC therapy. Each effect was ranked on a scale of 0 to 3 (0 = no effect, 1 = mild effect but did not disturb sleep, 2 = sleep disturbed, 3 = could not use device) At the end of the study, subjects were asked to rate the overall benefit of each therapy on a 10-point linear scale (1 = no benefit, to 10 = maximum benefit).
Anthropometric data including body height and weight, neck circumference (at the fourth cervical vertebra), and abdominal circumference (mid point between the tenth rib and iliac crest) were measured at baseline and at the end of each treatment period. Objective CPAP compliance time was downloaded from the Autoset T and the ResMed S6 Elite into a computer in the laboratory.
Analysis of Results
The primary outcome for treatment success was defined as an AHI
10/h of total sleep time. Partial success was defined as an AHI in the range > 10/h to
15/h slept. An AHI > 15/h slept was defined as treatment failure.
Results were analyzed on an intention-to-treat basis. Paired t tests were undertaken for the mean differences between the changes from baseline for each of the two treatments. Paired cephalometric data with and without the CMSC were compared using t tests. Stepwise linear regression analysis was undertaken to examine the relationship between cephalometric variables and changes in AHI. Multivariate analysis was used to determine any order effect. Because multiple comparisons were made, a significance level of 0.01 was used. The study protocol was approved by the Otago Ethics Committee, and all subjects gave written informed consent.
| Results |
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10/h slept) was achieved in 2 of 10 subjects, partial success (AHI > 10 to
15/h slept) was achieved in a further 2 subjects, and in the remaining 6 subjects there was no significant benefit, given that the AHI remained > 15/h slept with treatment. In three subjects, the AHI with CMSC was higher than the AHI at baseline. The mean adherence to CMSC therapy was 89 ± 23% of the total available study nights. Mean nightly duration of use of the CMSC was 5.2 ± 1.2 h (Table 3
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The difference in the mean change from baseline AHI to AHI with the nCPAP was therefore significantly greater than the mean change from baseline AHI to AHI with the CMSC (p = 0.001). The mean difference in the change from baseline in the total number of snores per hour using nCPAP was also significantly greater compared to the mean difference in the change from baseline to the total number of snores per hour using CMSC (p = 0.009) [Table 4 ]. No significant differences in the mean changes in sleep architecture or sleep efficiency were achieved using nCPAP compared to the CMSC (Table 4) . There was no treatment order effect.
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Subjective Outcomes
Results obtained from patient questionnaires and diary entries are summarized in Table 3
. Despite the differences between nCPAP and the CMSC for objective outcome measures, the subjective outcomes were comparable for both devices. There was a trend toward a lower mean ESS score using the CMSC, but the results were not significant (Table 3
, Fig 3
). Results for other measures of daytime symptoms and sleep symptoms (SF-36, FOSQ, SQ) compared to baseline were similar with each of the two therapies. There were fewer adverse effects reported with the CMSC than with nCPAP, but there was no preference demonstrated for one therapy over the other (Table 3)
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| Discussion |
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10/h slept) was used as the primary outcome measure to define treatment success. We failed to demonstrate that the CMSC was an effective treatment. Only 2 of 10 patients responded successfully with the CMSC, and the mean AHI for this group did not change (mean AHI at baseline, 29.4 ± 13.4/h slept; mean AHI with CMSC, 26.9 ± 17.2/h slept). These results contrasted strongly with the mean decrease in AHI obtained using nCPAP (9.9 ± 8.0/h slept). This outcome was disappointing, given the need to provide effective alternative therapy to nCPAP in a significant number of patients.29 Our study was based on evidence that during sleep, downward movement of the mandible has an influence on airway patency2 14 30 and that airway resistance increases with neck flexion.13 31 32 The CMSC was designed to prevent mandibular movement, and to hold the head in slight extension, thus preventing the postural changes that might contribute to OSA. Both CROM and cephalometric measures demonstrated that these two objectives were adequately achieved and retained throughout the CMSC treatment period.
Previously, Choi et al13 demonstrated in awake subjects that there is a significant difference in the mechanical resistance to airflow caused by changes in jaw and neck position, notably flexion, and that neck flexion is greater in OSA subjects. Studies undertaken by Kushida et al31 32 provided further support for our working hypothesis. These authors showed a significant improvement in AHI in subjects with OSA using a specially designed pillow to encourage neck extension. However, the results for individual subjects were inconsistent, and the clinical relevance of very simple posture control in the management of OSA remained unclear. For this reason, we adopted a more rigorous approach.
In our study, careful attention was paid to maintaining appropriate head and neck posture. The CMSC was fitted with the head in slight extension, measured using the external CROM instrument. Thus, cephalometric measurements for craniohorizontal variables obtained in supine position (Table 5) demonstrated that the position of the head in relation to the true horizontal and in relation to the cervical column remained in 10° of extension, and was significantly different to the baseline angles measured. The CMSC prevented head flexion beyond the NHP when subjects attempted to do so. Further, the chin piece of the CMSC supported the lower jaw and prevented downward rotation of the mandible. Theoretically, this ought also to have contributed positively to the maintenance of airway patency during sleep. Despite these features of the CMSC design, our results strongly suggest that maintaining the postural relationships of the cranium, mandible, and cervical spine during sleep is insufficient to maintain airway patency. Clearly, other anatomic and physiologic factors must have a dynamic overriding influence compared to simple skeletal relationships.
It is interesting to note that there was a very positive subjective response to this form of treatment. Our subjects completed a number of questionnaires relating to quality of life and sleep habits. Despite the striking differences between the CMSC and nCPAP in controlling OSA, there was a nonsignificant trend toward reduced daytime hypersomnolence, as measured by the ESS, with the collar. Also fewer adverse side effects were reported by subjects when using the CMSC. Clearly, a strong placebo effect was obtained, and our findings confirm the results of a previous study33 that have showed there are no clear-cut relationships between objective polysomnographic improvements and daytime function in patients with OSA. These outcomes highlight the importance of rigorous controlled crossover studies to assess the value of more conservative treatments to manage OSA.
Initially, our study was designed to include a sample size of 20 adults. However, the study was terminated prematurely after the results for 10 subjects were analyzed and confirmed that the CMSC was failing to achieve clinically important reductions in the AHI, despite subjective benefit. There was no "washout" period between the two study arms. It is unlikely that this would have altered the outcomes given the complete lack of objective benefit from the CMSC: no carry-over benefit was occurring.
Our results, although negative, provide important evidence that control of head and neck posture, perhaps adopted as a second-line treatment in the management of OSA, is unhelpful. Although nCPAP is clearly effective, its use is often restricted by the frequency of poor patient acceptance, or cost. Mandibular advancement splints are often recommended as an alternative to nCPAP, but their long-term efficacy is uncertain. As a consequence, sleep physicians and patients alike are seeking new, innovative therapies that are cost-effective and are well tolerated. There may yet be a role for the CMSC as an alternative to the chin strap in assisting jaw closure during nCPAP therapy. However, our data suggest that while elimination of postural abnormalities may have some subjective impact, they are in themselves insufficient to control OSA.
| Footnotes |
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This work was performed at the Respiratory Research Unit, Dunedin School of Medicine, University of Otago, Dunedin, New Zealand.
The study was funded by the University of Otago School of Physiotherapy and the Otago Respiratory Research Trust.
Received for publication December 3, 2002. Accepted for publication August 15, 2003.
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