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Volume 11 No. 10
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Sleep Medicine Pearls

A Young Man Running Out of Treatment Options

http://dx.doi.org/10.5664/jcsm.5104

Ankur Girdhar, MD1; Richard B. Berry, MD1; Scott Ryals, MD1; Emily Beck, MD1; Mary Wagner, MD2
1Division of Pulmonary, Critical Care, and Sleep Medicine, Malcom Randall VA medical center, Gainesville, FL; 2Division of Pediatric Pulmonology, University of Florida, Gainesville, FL

A 33-year-old male combat veteran came to the clinic for evaluation of snoring, apneas and daytime sleepiness. He was diagnosed with obstructive sleep apnea (OSA) at the age of 22, after he was witnessed to have apneas by other military veterans who were deployed with our patient while he was on active duty. His initial study had revealed an apnea-hypopnea index (AHI) of 16/hour and a respiratory disturbance index (RDI) of 19/h. At that time, due to active deployment overseas and personal preference, he chose to explore surgical options rather than positive airway pressure (PAP) therapy. He underwent a turbinate reduction and a nasal septoplasty. He did not have a repeat sleep study, but these surgeries helped improve his sleep symptoms significantly for several years following surgery.

However, about five years later, he began experiencing significant sleep symptoms. He had gained only 5 pounds since his initial sleep study. A repeat sleep study revealed an AHI of 20/h with an RDI of 24/h. At this time he underwent a PAP titration study, but unfortunately could not tolerate any mask interface on his face. He ascribed this inability to tolerate the mask due to claustrophobia and anxiety related to his posttraumatic stress disorder (PTSD). Since PAP therapy was not an option for this patient he was referred to oto-rhino-laryngology for consideration of surgical options. He underwent a repeat turbinate reduction which once more led to partial relief in his sleep symptoms following surgery. He again did not have a follow-up sleep study to assess the effect of surgery on the AHI.

Several years after his second surgery, his symptoms returned and he presented to our sleep clinic. He reported snoring, breathing pauses, unrefreshing sleep, hypertension, and daytime sleepiness. His Epworth Sleepiness Scale score was 13/24. He was distressed by frequent nocturnal awakenings and breathing pauses, and he believed these were increasing the nocturnal anxiety and nightmares. He had also gained about 15 pounds since his first sleep study. He was taking prazosin 5 mg to help reduce the frequency of his nightmares and alprazolam 0.5 mg as needed for relief of anxiety. In the clinic he agreed to undergo a repeat diagnostic sleep study, but strongly refused a PAP titration or PAP treatment. While he was not opposed to other surgical options, he was not very enthusiastic having additional upper airway surgery. On physical examination in the clinic he was found to have mild retrognathia, poor dentition (with only a few remaining teeth in each dental arch), an enlarged uvula, and a Mallampati score of 3. His weight was 221 pounds, neck circumference was 16 inches, and BMI was 30.90 kg/m2. The remainder of the physical examination was unremarkable.

We discussed weight loss with the patient and ordered a full channel diagnostic polysomnogram. The results of this study are shown in Table 1. Hypopneas were scored based on the recommended hypopnea definition in the American Academy of Sleep Medicine scoring manual.1

Diagnostic polysomnogram.

 

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Table 1

Diagnostic polysomnogram.

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Based on the results of this study, the patient had significantly worsened sleep disordered breathing. He wanted treatment, but was firm on his decision to avoid PAP therapy and hesitant to undergo further surgery.

QUESTION: What other novel options are available to treat this patients' sleep apnea?

ANSWER: Treatment options include mask desensitization or oral pressure therapy.

DISCUSSION

PAP intolerance is a very common problem encountered in sleep clinics.2 Intolerance can be related to a number of issues including discomfort or poor fit of the mask interface. This leads to exploration of other options for treating patients with OSA. Our patient underwent nasal surgery on two occasions. Nasal surgery with turbinate reduction for treatment of OSA has been analyzed in a meta-analysis.3 This analysis showed that while this surgery was effective in reducing the severity of symptoms, the success rate after this surgery (defined as AHI improvement of more than 50% and a postoperative AHI ≤ 20 events/h) was only 16.7%. An observational study of the effects of nasal surgery on sleep apnea found that the combination of septoplasty and turbinate reduction was more effective than septoplasty alone.4 However, only 27% of patients undergoing both septoplasty and turbinate reduction had a postoperative drop in AHI to less than 20/h and/or a 50% reduction in AHI. Even though multiple alternatives are available for patients with mild obstructive sleep apnea, patients with more severe OSA have limited options as the most effective therapy to date is PAP.5,6 However, acceptance and adherence to PAP treatment remains much lower than desired.2,7,8

In certain populations, particularly in veterans who have the posttraumatic stress disorder (PTSD), initiating PAP can be difficult.9,10 This is often due to anxiety or panic generated by an apparent constraint from a mask interface.9,11 PTSD is a common diagnosis in patients receiving care at VA medical centers. This diagnosis is made using the DSM-5 criteria12 and scales such as the Clinician-Administered PTSD Scale for DSM-5 (CAPS-5),13 which is considered a gold standard in PTSD assessment. This scale can additionally assess the severity of PTSD, which can help identify patient who are more predisposed to having claustrophobia. Anxiety-deconditioning therapy such as “mask desensitization” has been used for patients with intolerance to a mask interface.14 This entails getting patients comfortable with putting the mask on their face without using PAP, initially for short periods of time every day with a gradual increase in the time spent with the mask in place. Subsequently the patient uses the PAP with the mask during the non-sleeping hours and then gradually during the sleeping hours. This technique has been shown to be moderately successful in the general population.14 To our knowledge, there have been no studies or case reports of effectiveness of this therapy in military veterans.

Oral appliances, including mandibular advancement devices (MADs) and tongue retaining devices are other available options. MADs are constructed by dentists according to particular specifications of the patient's oral structures. Usual recommendations suggest that patient should have good dentition with at least 6–10 teeth in each arch for jaw advancement devices.15 Unfortunately our patient did not have adequate dentition for this type of treatment. Tongue retaining devices can still be used in edentulous patients or those with poor dentition, but they have been found to have a lower compliance rate and easily come out during the night.16 Surgical procedures such as uvulopharyngopalatoplasty (UPPP) or maxillary mandibular advancement (MMA) offer additional alternatives to PAP. Depending on the type of procedure and severity of sleep apnea, they carry a substantial failure rate in addition to being invasive, irreversible interventions. Hypoglossal nerve stimulation17 using a small stimulus generator (about the size of a pacemaker) placed beneath the skin on the upper chest, a breathing sensor lead, and a stimulation lead is another FDA approved treatment option available for patients who do not tolerate CPAP and have a BMI less than 32 kg/m2. This treatment is currently available at a few VA medical centers. It works by electrical stimulation of the genioglossus muscle, the largest upper airway dilator muscle, causing tongue protrusion and stiffening of the anterior pharyngeal wall, and thereby relieving obstruction in the airway of OSA patients.

In recent years a novel treatment option called oral pressure therapy (OPT, Winx, Apnicure, Redwood, CA) has become available and is approved by the Federal Drug Administration for treatment of OSA. OPT works by applying a low level of suction via a mouthpiece which is placed between the teeth and over the patient's tongue (Figure 1). Suction retains the tongue and displaces the soft palate forward, away from the posterior pharyngeal wall maintaining an open upper airway while the patient is sleeping. MRI studies have shown that this device can lead to significant increase in the retropalatal airway space.18 The oral mouthpiece is attached by a tube running through the patient's lips to a bedside console delivering a controlled amount of suction. The console has the ability to record daily use on a data card (time with adequately applied level of suction). The interface is less cumbersome than needed for CPAP and may improve acceptance for some patients by avoiding a mask interface on the patient's face. A range of mouthpieces is available to fit a wide spectrum of dental arch sizes. Proper size is chosen based on a dental imprint after patients bite down on a wax wafer. Of note, only one molar in each quadrant is required for OPT. Patients with dentition inadequate for use of a mandibular retaining device may be treated with OPT. This treatment would also aid patients with OSA that have facial pain syndromes such as trigeminal neuralgia who cannot tolerate a mask on their face. On the other hand patients with bruxism are likely not good candidates for OPT, as the mouthpiece may be damaged.

(A) The general set up of the OPT machine showing the compact size of the console with less constraining interface. (B) Soft, rubberized mouthpiece, which is less cumbersome than the CPAP mask. (C) Cross section showing the placement of mouthpiece on the tongue increasing the retro-palatal space.

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Figure 1

(A) The general set up of the OPT machine showing the compact size of the console with less constraining interface. (B) Soft, rubberized mouthpiece, which is less cumbersome than the CPAP mask. (C) Cross section showing the placement of mouthpiece on the tongue increasing the retro-palatal space.

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A multicenter study19 done in the past to investigate the effectiveness of OPT has shown that with the use of this therapy a clinically significant response (defined as treatment AHI ≤ 10/h and ≤ 50% of the control value) was seen in 20 of the 63 subjects evaluated. Median pre-intervention AHI was 27.5/h at the time of enrollment in to the study. With the use of OPT this was reduced to only a median of 13.4/h on the first treatment night, and 14.8/h on 28th treatment night of therapy. Adherence over 28-day periods was good with a mean usage of 6.0 ± 1.4 h use per night. Even though a clinically significant response in the AHI occurred in less than half of the study subjects, it was effective in some patients with both moderate (9/18) and severe (7/30) OSA. Exclusions to entry into the study included obesity (BMI > 40 kg/m2) and nasal obstruction. It is also important to note that a significant number of potential subjects were excluded due to inability to demonstrate an acceptable amount of sleep and duration of effective suction pressure on a screening night at home. A single-night feasibility study found that the number of awakenings and the percentage of sleep time spent in N1 sleep were significantly reduced while percentage of sleep time spent in N2, N3, and REM were significantly increased. OPT is generally well tolerated but initial side effects are commonly noted.20 In the feasibility study, device-related side effects were reported by 32% of the subjects, but 92% of the side effects were mild. The most common side effect in the multicenter study19 was oral tissue discomfort or irritation. A dry mouth and dental discomfort were also reported. In 43% of patients, side effects resolved with continued treatment.

To determine if OPT therapy will be effective in a particular patient, a treatment sleep study (Winx titration study) in which patient uses OPT instead of PAP is performed. The patient's respiratory events, oxygen desaturations, and sleep stages are recorded in usual fashion. The level of suction applied by the machine is automatic and set by a proprietary algorithm. The Winx titration study is used to document effectiveness and the ability to deliver suction during sleep. While mouthpiece size may be changed for comfort or the ability to deliver adequate suction, the level of pressure cannot be changed by the sleep technologist. A significant reduction in AHI to ≤ 10/h and patient's tolerance and relief of sleep symptoms can be a valuable guide to success with this treatment.

At the present time the availability of OPT is limited to a few durable medical equipment companies. Many third party payers are also not currently covering the cost of this therapy. However, the treatment is available in the VA Health Care System through the Prosthetics service and is currently available at a few centers as an additional treatment option for patients not able to use PAP therapy. Difficulties with providing OPT therapy include the need to have a number of different mouthpieces sizes on hand in the sleep center and developing proficiency in sizing patents for the proper mouthpiece. Since our patient had limited treatment options for his severe obstructive sleep apnea, a Winx titration study was performed to assess if this device would effectively treat his sleep apnea. The results of this study are shown in Table 2.

Winx titration.

 

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Table 2

Winx titration.

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The patient liked this therapy and remarked that his sleep on this device was much better than usual. He was issued this therapy and he tolerated it well. During OPT, the user breathes exclusively through the nose, and, in the multicenter trial, one of the exclusion criteria was a peak nasal inspiratory flow rate of less than 75 L/minute. Our patient's two prior nasal airway surgeries may have increased the likelihood of good clinical outcome with OPT. Machine adherence data confirmed nightly use of the device with average use over five hours in the first month of treatment. He reported sustained relief from his sleep symptoms and his Epworth Sleepiness Scale score improved to 5/24 on follow-up.

SLEEP MEDICINE PEARLS

  1. PAP treatment may be challenging in some patients with PTSD due to claustrophobia.

  2. Mask desensitization therapy can be used to increase mask acceptance in motivated patients with claustrophobia.

  3. Oral pressure therapy provides a treatment alternative for OSA patients with claustrophobia.

  4. Effectiveness of OPT in a particular patient can be gauged by performing a sleep study while patient is using this device. Although OPT is effective in all severities of OSA, treatment is successful in less than half of the patients. Positive airway pressure is more reliably effective than OPT in patients with moderate to severe OSA. Long-term studies of adherence and effectiveness of OPT are not available.

  5. OPT can be a treatment option for sleep apnea patients with poor dentition and those with claustrophobia. The effectiveness of OPT must be demonstrated by sleep monitoring.

DISCLOSURE STATEMENT

This was not an industry supported study. The authors have indicated no financial conflicts of interest.

ABBREVIATIONS

AHI

apnea-hypopnea index

BMI

body mass index

ESS

Epworth Sleepiness Scale

FDA

Federal Drug Administration

MAD

mandibular advancement device

OPT

oral pressure therapy

OSA

obstructive sleep apnea

PAP

positive airway pressure

PTSD

posttraumatic stress disorder

RDI

respiratory disturbance index

UPPP

uvulopharyngopalatoplasty

WASO

wake after sleep onset

CITATION

Girdhar A, Berry RB, Ryals S, Beck E, Wagner M. A young man running out of treatment options. J Clin Sleep Med 2015;11(10)1239–1242.

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