pii: jc- 00265 -15http://dx.doi.org/10.5664/jcsm.5414

CAS E R EPOR T S

Mandibular Advancement Device—Emergent Central Sleep Apnea Can Resolve Spontaneously: A Case Report Arjun Mohan, MD1,2,3; Jennifer Henderson, DO1,2; M. Jeffery Mador, MD1,2 Veterans Affairs Western NY Healthcare System, Buffalo, NY; 2Division of Pulmonary, Critical Care and Sleep Medicine, University at Buffalo, State University of New York-Buffalo, NY; 3Division of Pulmonary, Critical Care and Sleep Medicine, East Carolina University School of Medicine, Greenville, NC

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The development of treatment emergent central sleep apnea (CSA) has been described after almost all obstructive sleep apnea (OSA) therapies. While the course of positive airway pressure (PAP) emergent CSA, is better established; little is known about the natural course of mandibular advancement device (MAD) emergent CSA. Previous reports failed to comment on its natural course or report treatment with advanced ventilator modes such as adaptive servoventilation. We describe spontaneous resolution of MAD emergent CSA in a patient with moderate OSA who refused PAP. We also highlight the need for follow up polysomnography (PSG) after maximal advancement with a MAD and the possible association between MAD emergent CSA and atrial fibrillation. The exact pathophysiology of this phenomenon remains unclear but may relate to high loop gain of the respiratory system resulting in ventilatory overshoot after treatment and atrial fibrillation associated increased susceptibility to periodic ventilation. Keywords: treatment emergent central sleep apnea, mandibular advancement device, spontaneous resolution. Citation: Mohan A, Henderson J, Mador MJ. Mandibular advancement device—emergent central sleep apnea can resolve spontaneously: a case report. J Clin Sleep Med 2016;12(1):137–138.

for pulmonary hypertension. A level 1 polysomnogram demonstrated moderate OSA with associated hypoxemia but no central events (Table 1). Despite strong recommendations, the patient refused PAP due to concerns of claustrophobia. He was subsequently offered a MAD (SomnoDent; SomnoMed, Denton TX). He tolerated maximal mandibular advancement to 7.5 mm (53.7% of total protrusion) over a 3-month period. At this point, a PSG with his MAD showed an AHI that was not significantly different from baseline, but 49% of the respiratory events were central, consistent with treatment-emergent central sleep apnea (Table 1). PAP was offered again, but the patient again refused this therapy as he felt his symptoms had largely resolved with use of his MAD. He was monitored closely for an additional year and a third PSG with his MAD was repeated (Table 1). This showed very mild residual OSA with complete resolution of his central events. Of note, his BMI was stable during this period (lowest noted was 35.5 at the end of 1 year). He was in sinus rhythm during all 3 PSGs. However, long-term Holter monitoring prior to his first study had shown intermittent bouts of atrial fibrillation. The patient had no clinical episodes of paroxysmal atrial fibrillation during the year of treatment with his MAD.

I N T RO D U C T I O N Central sleep apnea (CSA) can be primary (idiopathic) or secondary.1 OSA treatment emergent central sleep apnea has been described after PAP therapy, MAD, and OSA surgeries such as maxillofacial surgery and tracheostomy.2–6 Three prior reports on MAD emergent CSA3–5 failed to comment on its natural course or report treatment with advanced ventilator modes such as adaptive servo ventilation. We report spontaneous resolution of these events in a patient with moderate OSA who refused PAP. We hypothesize that with continued MAD treatment these CSA events can resolve spontaneously, and expectant management may be an option in such cases. We also highlight the need for close clinical and PSG follow up after maximal MAD advancement. R E P O R T O F CAS E A 69-year-old male with a past medical history of diabetes, hypertension, coronary artery disease, and atrial fibrillation requiring multiple ablations was referred to our sleep clinic for OSA evaluation. On presentation, he reported poor sleep quality and constant daytime fatigue. His body mass index was 36.5 and neck size was 17.5 inches. His cardiopulmonary examination was remarkable for an irregularly irregular heartbeat. His home medications included glipizide, lisinopril, metoprolol, simvastatin, warfarin, and aspirin. His last echocardiogram showed mild concentric left ventricular hypertrophy with an ejection fraction > 55%, bi-atrial enlargement, and normal right ventricular size and function with no evidence

D I SCUS S I O N Recurrent apneic events occurring in the absence of respiratory effort is called central sleep apnea.1 MAD-emergent central apneas have been described in the literature since 2006.3 The prevalence and natural history of MAD-emergent central sleep apnea is unknown. Our report highlights that these 137

Journal of Clinical Sleep Medicine, Vol. 12, No. 1, 2016

A Mohan, J Henderson and MJ Mador. Case Report

Furthermore, we postulate an association with comorbid cardiac disease especially atrial fibrillation. Atrial fibrillation has been a comorbid disease in most reported cases.4,5 However, its association with treatment-emergent CSA development has not been well established, although it is a well-established risk factor for the development of non-treatment emergent CSA. It may work in conjunction with other possible mechanisms, such as high loop gain of the respiratory system resulting in ventilatory overshoot after treatment. Reports such as ours are needed to understand the phenomenon of MAD emergent central sleep apnea, as MAD becomes a first line therapy for treatment of mild to moderate sleep apnea.

Table 1—Summary of sleep study reports. STUDY 2 (with MAD)

STUDY 1 Baseline

Obstructive apnea index (events per hour)

After maximal STUDY 3 mandibular (with MAD) advancement 1 year later

10.2

4.7

0.3

Central apnea index (events per hour)

0.0

7.4

0.0

Hypopnea index (events per hour)

6.0

2.8

6.0

Apnea-hypopnea index (events per hour)

16.2

14.9

6.3

% obstructive apneas

64.0

32.0

4.0

0.0

49.0

0.0

% hypopneas

36.0

19.0

96.0

Arousal index (events per hour)

19.6

16.4

8.9

Nadir SaO2 (%)

88.0

89.0

90.0

% total sleep time with SpO2 < 90%

32.7

1.1

0.0

% central apneas

R E FE R E N CES 1. Eckert DJ, Jordan AS, Merchia P, Malhotra A. Central sleep apnea: pathophysiology and treatment. Chest 2007;131:595–607. 2. Javaheri S, Smith J, Chung E. The prevalence and natural history of complex sleep apnea. J Clin Sleep Med 2009;5:205–11. 3. Avidan AY, Guilleminault C, Robinson A. The development of central sleep apnea with an oral appliance. Sleep Med 2006;7:187–91. 4. Kuzniar TJ, Kovacevic-Ristanovic R, Freedom T. Complex sleep apnea unmasked by the use of a mandibular advancement device. Sleep Breath 2011;15:249–52. 5. Gindre L, Gagnadoux F, Meslier N, Fleury B, Gustin JM, Racineux JL. [Central apnea developing during treatment with a mandibular advancement device]. Rev Mal Respir 2006;23:477–80. 6. Corcoran S, Mysliwiec V, Niven AS, Fallah D. Development of central sleep apnea after maxillofacial surgery for obstructive sleep apnea. J Clin Sleep Med 2009;5:151–3.

apneas, similar to those seen with PAP therapy, can resolve over time. Given that spontaneous resolution is noted in 50% to 75% of PAP-emergent central apneas2 after several months of PAP therapy, we suspect spontaneous resolution of MADemergent central apneas occurs more frequently than anticipated. Since MADs are advanced slowly, repeat sleep studies documenting their efficacy are not usually performed for at least 3 months after treatment initiation. Thus, if MAD-emergent CSA behaves similarly to PAP-emergent CSA, central events may have occurred and resolved by the time the MAD efficacy study is performed. However, unlike PAP therapy which is effective immediately, the gradual advancement of MAD devices means that treatment effectiveness may not occur for some time after therapy is initiated. This likely explains why central events were still present after 3 months of therapy and why they resolved spontaneously after more time at effective therapy. The key to resolution with PAP therapy appears to be strict adherence with therapy.

Journal of Clinical Sleep Medicine, Vol. 12, No. 1, 2016

SUBM I SSI O N & CO R R ESPO NDENCE I NFO R M ATI O N Submitted for publication June, 2015 Submitted in final revised form August, 2015 Accepted for publication August, 2015 Address correspondence to: M. Jeffery Mador, VA Western NY Healthcare System, Division of Pulmonary, Critical Care and Sleep Medicine, 3495 Bailey Avenue, Buffalo NY 14215; Tel: (716) 862-8634; Fax: (716)862-6526; Email: mador@ buffalo.edu

D I SCLO S U R E S TAT E M E N T This was not an industry supported study. The authors have indicated no financial conflicts of interest.

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Mandibular Advancement Device-Emergent Central Sleep Apnea Can Resolve Spontaneously: A Case Report.

The development of treatment emergent central sleep apnea (CSA) has been described after almost all obstructive sleep apnea (OSA) therapies. While the...
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