ARTICLE IN PRESS Sleep Medicine ■■ (2015) ■■–■■

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Pneumoparotid associated with a mandibular advancement device for obstructive sleep apnea Marta Cabello a,*, Emilio Macías b, Alejandro Fernández-Flórez c, Marian Martínez-Martínez d, Juan Cobo b, Félix de Carlos b a

Sleep Unit, Respiratory Division, Marqués de Valdecilla University Hospital, Santander, Spain Department of Surgery and Medical-Surgical Specialities, Area of Orthodontics, Faculty of Medicine, University of Oviedo, Spain c Department of Radiology, Marqués de Valdecilla University Hospital, Santander, Spain d Sleep Unit, Department of Clinical Neurophysiology, Marqués de Valdecilla University Hospital, Santander, Spain b

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Article history: Received 6 March 2015 Accepted 9 April 2015 Available online Keywords: Pneumoparotid Mandibular advancement device Obstructive sleep apnea Oral appliance Side effects

1. Introduction to the case A 42-year-old man with moderate obstructive sleep apnea (OSA) was treated with a mandibular advancement device (MAD) because of intolerance to continuous positive airway pressure (CPAP). This appliance is composed of two-piece splints attached to each other by a metal bar connector located in the middle of both splints (Fig. 1). It serves to push the mandible forward relative to the maxilla. After three months using the MAD, he complained about right subauricular swelling with moderate pain and tenderness. On clinical examination, the patient was afebrile, and right sub-mandibular angle swelling, pain, and subcutaneous crepitus were found. His wife referred that he was sleeping without snoring, but he was blowing all night and sometimes she heard an intense whistle. No other mechanism of intraoral pressure increase was disclosed. Non-steroidal antiinflammatory drugs were administered and two diagnostic procedures were performed (Figs. 2 and 3). 2. Image analysis Figure 1 shows the oral appliance used by the patient for OSA treatment with a fenestrated metal bar joining the two-piece splint at 70% of the maximum advancement.

* Corresponding author. Sleep Unit, Respiratory Division, University Hospital Marqués de Valdecilla, Av. Valdecilla, s/n, 39008 Santander, Cantabria, Spain. Tel.: +34 942 202520; fax: +34 942 202541. E-mail address: mc[email protected] (M. Cabello).

Figure 2 shows an right parotid ultrasound revealing a normal morphology of the gland with multiple hyperechogenic bright areas into the parenchyma representing pneumoparotid (Fig. 2A). Air in the parotid duct appears as a thin hyperechogenic line (Fig. 2B). No lithiasis was found. Figure 3 shows a non-contrast facial CT scan confirming the presence of numerous air bubbles in the right parotid (Fig. 3A) and gas along the right Stensen’s duct (Fig. 3B).

3. Discussion Pneumoparotid is defined by the presence of air in the salivary ducts and acini of the parotid gland. In the absence of a gasproducing bacterial parotitis, gas in the parotid gland is assumed to be due to the re-flux of pressurized air from the mouth into the Stensen’s duct, with excessive increase in intraoral pressure. There are different causes of pneumoparotid, including dental instrumentation bouts of prolonged coughing, forced clearing of the nares, rapid decompression of a scuba diver, doing a spirometry, occupational hazard (glass blowers and players of wind instruments, balloon blowers), or inveterate gum chewing [1]. On the other hand, MADs represent the main non-CPAP alternative for patients with OSA, and are recommended for patients with mild to moderate OSA and those who do not tolerate CPAP [2]. MADs are inserted into the mouth and protrude from the lower jaw, increasing vertical opening during sleep in order to reduce airway

http://dx.doi.org/10.1016/j.sleep.2015.04.020 1389-9457/© 2015 Elsevier B.V. All rights reserved.

Please cite this article in press as: Marta Cabello, et al., Pneumoparotid associated with a mandibular advancement device for obstructive sleep apnea, Sleep Medicine (2015), doi: 10.1016/j.sleep.2015.04.020

ARTICLE IN PRESS 2

M. Cabello et al./Sleep Medicine ■■ (2015) ■■–■■

Fig. 1. Mandibular advancement device. Frontal view (A) and lateral view (B).

Fig. 2. Right parotid ultrasound showing gas into the parotid gland (A, arrows) and parotid duct (B, arrow).

Fig. 3. Facial CT scan showing pneumoparotid (A, arrow) and gas into the Stensen’s duct (B, arrows).

obstruction, apneas, and snoring [3]. The most commonly reported side effects such as tooth pain, dental crown damage, excessive salivation, dry mouth, and temporomandibular joint pain are usually mild and transient [4]. Nevertheless, objective complications such as tooth movement, skeletal changes, and occlusal alteration have also been reported [5].

In our patient, the only cause of pneumoparotid was the blowing and whistles at night related to the MAD. This oral appliance may induce resistance to expiration, leading to an increase in the intraoral pressure. To our knowledge, this side-effect of MAD has not been previously described, and we suggest that it should be considered when oral devices for OSA are designed.

Please cite this article in press as: Marta Cabello, et al., Pneumoparotid associated with a mandibular advancement device for obstructive sleep apnea, Sleep Medicine (2015), doi: 10.1016/j.sleep.2015.04.020

ARTICLE IN PRESS M. Cabello et al./Sleep Medicine ■■ (2015) ■■–■■

Conflict of interest The authors declare that they have no conflict of interest. The ICMJE Uniform Disclosure Form for Potential Conflicts of Interest associated with this article can be viewed by clicking on the following link: http://dx.doi.org/10.1016/j.sleep.2015.04.020. References

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[2] Marklund M, Verbraecken J, Randerath W. Non-CPAP therapies in obstructive sleep apnoea: mandibular advancement device therapy. Eur Respir J 2012;39(5):1241–7. [3] Woodson BT. Non-pressure therapies for obstructive sleep apnea: surgery and oral appliances. Respir Care 2010;55(10):1314–21. [4] Qaseem A, Holty JE, Owens DK, et al. Management of obstructive sleep apnea in adults: a clinical practice guideline from the American College of Physicians. Ann Intern Med 2013;159(7):471–83. [5] Martínez-Gomis J, Willaert E, Nogues L, et al. Five years of sleep apnea treatment with a mandibular advancement device. Side effects and technical complications. Angle Orthod 2010;80(1):30–6.

[1] Luaces R, Ferreras J, Patiño B, et al. Pneumoparotid: a case report and review of the literature. J Oral Maxillofac Surg 2008;66(2):362–5.

Please cite this article in press as: Marta Cabello, et al., Pneumoparotid associated with a mandibular advancement device for obstructive sleep apnea, Sleep Medicine (2015), doi: 10.1016/j.sleep.2015.04.020

Pneumoparotid associated with a mandibular advancement device for obstructive sleep apnea.

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