Case Report
Use of an individual mandibular advancement device for
an obstructive sleep apnoea patient with facial paralysis:
a short-term follow-up case report
B. PIS¸KIN*, C. SIPAHI*, M. KO
¨ SE
†, O
¨ . KARAKOC¸
‡, H. S¸EVKETBEYOG
˘ LU
§& G. K. ATAC
¸
¶*Gulhane Military Medical Academy, Department of Prosthodontics, Ankara,†MediSina Hospital, Van,‡Gulhane Military Medical Academy,
Departments of Otolaryngology, Head and Neck Surgery, Ankara,§Konya Military Hospital, Konya and¶Department of Radiology, University of Ufuk, Ankara, Turkey
SUMMARYThis case report aimed to describe the fabrication procedure and treatment efficacy of an individual, one-piece, non-adjustable mandibular advancement device (MAD) for a moderate obstruc-tive sleep apnoea patient with facial paralysis (FP). Mandibular advancement device was fabricated with autopolymerising acrylic resin. The intermax-illary relations were recorded such as to fix the mandible at a protruded position with increased vertical dimension. Initial evaluation of the MAD was made with axial magnetic resonance imaging and polysomnography on the first day of usage. Following evaluations were made on the third and
sixth month. After a follow-up period of 6 months, Apnoea⁄ Hypopnea Index (AHI) significantly decreased from 26Æ7 to 3Æ0. However, the average oxygen saturation did not improve as expected initially. The MAD therapy decreased the AHI scores of a patient with FP. At the end of a follow-up period of 6 months, the patient did not report any serious complaint except temporary tooth pains. KEYWORDS: obstructive sleep apnoea, apnoea–hypop-nea index, individual mandibular advancement device, facial paralysis
Accepted for publication 18 December 2011
Introduction
Obstructive sleep apnoea (OSA) is a life-threatening disorder characterised by repetitive cessation of respi-ration, owing to partial or complete obstruction of upper airway, generally resulting with arterial blood oxygen desaturation during sleep (1–5).
A number of craniofacial, dentofacial and oro-facial abnormalities, such as reduced cranial base length, steep mandibular plane, large tongue, tonsillar hyper-trophy, enlarged pharyngeal walls, micrognathia, ret-rognathia, enlarged and elongated soft palate, large and long uvula, elongated maxillary teeth and inferior positioning of hyoid bone have been associated with OSA (4–10). Besides anatomical disorders, vocal fold paralysis that accompanies with OSA, is also reported (11, 12). However, there is an absence of literature
concerning the management of OSA along with the paralysis of certain head and neck regions. Facial paralysis (FP) is one of the most common paralysis types of head and neck region. Facial paralysis may take origin from systemic illnesses such as malign hypertension or diabetes mellitus, and it may also occur after trauma, and oral, otolaryngologic, cosmetic or oncologic surgery (13–15). Some FP cases have been reported after cerebellopontine angle surgery (16).
Therapeutic approaches to OSA include avoidance of alcohol, weight loss, pharyngeal and hypopharyngeal surgery, tracheostomy, continuous positive airway pressure (CPAP) or pharmacologic agents. However, restrictions arise from mentioned approaches owing to the lack of patient compliance, serious complications or drug side effects (17, 18).
Journal of Oral Rehabilitation 2012 39; 472–478
treatment choice for mild- to moderate OSA patients (17, 19–22).
Through the survey of literature, a report concerning MAD application for the treatment of OSA patients with FP is not encountered. This case report aimed to present the fabrication procedure and treatment effi-cacy of an individual MAD for a moderate obstructive sleep aponea patient with FP.
Case
A 36-year-old Caucasian male patient registered to Otolaryngology, Head and Neck Surgery clinics with the complaint of anxiety, excessive daytime sleepiness and witnessed sleep apnoea. In his medical history, he had the diagnose of cerebellopontine angle tumour and had a subtotal mass excision procedure by suboccipital paramedian approach for his complaints of tinnitus and hearing loss at a neurosurgery clinic, 2 years ago. Histopathologic evaluation revealed the diagnosis of Schwannoma. The patient presented the FP after the surgery.
The examination of the patient revealed a grade 2 FP according to House–Brackman Scale (Fig. 1a), a slight nistagmus when looking at right and left sides, and dysdiadokokinesis on the left side. The Romberg’s test was positive.
was performed to investigate the sleep disturbance, revealed moderate OSA with Apnoea⁄ Hypopnea Index (AHI) of 26Æ7. All apnoeas were obstructive except three central apnoeas which were seen during the total sleep time.
Nasal CPAP treatment was initiated after determina-tion of sleep disorder. Unfortunately, the patient did not tolerate the CPAP treatment because of epitaxis and frequent sleep fragmentation. It was decided that any kind of pharyngeal surgery may not prevent the patient from pharyngeal collapse during sleep, owing to coex-istent FP. Under these circumstances, the patient was referred to the dental department of the hospital to receive an OA treatment.
Clinical intra-oral and extraoral examination revealed decreased volume of oral cavity owing to enlarged tongue and slightly restricted mouth opening with jaw deviation from the midline to non-paralysed side (Fig. 1b). The patient did not have a complaint of bilateral temporamandibular joint (TMJ) or ear pain. There were no audible clicking or crepitus and pain except a mild tenderness in masticatory muscles in the palpation of the masticatory muscles and bilateral TMJ. It was considered that oropharyngeal or hypopha-ryngeal collapses could manifest during sleep, owing to enlarged tongue along with reduced volume of oral cavity and altered muscle activity because of FP.
(a) (b)
The final treatment decision was to fabricate an individual, non-adjustable, one-piece MAD to increase the volume of intra-oral cavity for the enlarged tongue, advancing the mandible anteriorly to prevent pharyn-geal collapse and to prevent the jaw deviation to non-paralysed side during sleep.
Materials and methods
Fabrication of MAD
Anatomic impressions were made with irreversible hydrocolloid impression material (Tulip Color Switch*), individual impression trays were fabricated with resin based light curing material†, and functional impressions were made with condensation silicone material (Zeta-plus, Oranwash L‡). The master models were cast with type III dental stone (Moldano§). Maxillary and man-dibular occlusal splints were fabricated on isolated (Isolant⁄ C.M.S¶) master models with autopolymerising
acrylic resin (Ortocryl**). Splints were prepared such as to cover occlusal and incisal surfaces of the dentition for prevention, to minimise the orthodontic side effects of MAD and to provide comfortable usage combined with satisfactory retention and stabilisation during sleep.
Intermaxillary relationships were determined follow-ing the fabrication of splints. The horizontal
intermax-illary relation was recorded at approximately 75% of maximal mandibular protrusion. (Fig. 2a–c).
The physiologic vertical dimension was increased up to 5 mm, and the recorded intermaxillary relationship was mounted on an articulator (Artex Type NK††). Autopolymerising acrylic resin was applied to fix maxillary and mandibular occlusal splints from first premolar up to second molar teeth bilaterally. After the MAD was completed, it was inserted into the mouth with minor adjustments. The patient was informed about how to use the appliance (Fig. 3).
Evaluation of MAD
Pharyngeal magnetic resonance (MR) imaging was performed on supine position, while the patient was awake with and without MAD to evaluate the enlarg-ing effect of the device on the upper airway before initiation of the treatment. Full-night polysomnograph-ic records were also taken with the devpolysomnograph-ice. Epworth Sleepiness Scale was performed before applying the MAD and at the third month and sixth month follow-up.
Initial evaluation of the device was conducted with pharyngeal MR imaging. The oropharyngeal column of the patient was scanned by 1.5 T superconductive magnet MR equipment (Signa Excite 1.5 T‡‡). The slice thickness was 5 mm with 1-mm gaps.
T1 and T2 weighted axial and sagittal images were taken for measurement and evaluation of oro-pharyngeal cavity. Air column cross-sectional area was measured from accumulative counting of every
(a) (b) (c)
Fig. 2. (a) Vertical lines were marked on right lower pre-molar and upper occlusal splint at centic occlusion, (b) maximum protrusion, (c) approximately 75% of maximum protrusion.
*Cavex, Haarlem, Holland.
†
Vertex, Zeist, The Netherlands.
‡Zhermack, Rovigo, Italy.
§Heraeus Kulzer, Hanau, Germany.
¶
Dentsply, UK.
**Dentaurum, Ispringen, Germany.
††
Amann Girrbach, Pforzheim, Deutschland.
cross-sectional area of oropharynx drawn manually by an experienced radiologist with GE software on work-station from digital images. Every consecutive axial section of T1 W image starting from the base of nasopharynx to the root of the tongue was counted.
Full-night attended type 1 PSG (Embla S4000, Rem-brandt and Somnologica software§§) evaluation was made in sleep laboratory, while MAD was in the mouth during sleep on the first day of application. The subsequent PSG assessments were made at third and sixth month. Polysomnographys were scored according to the 2007 American Academy of Sleep Medicine Manual for the Scoring of Sleep and Related Events.
Results
Air column cross-sectional area at the retrolingual region was 7Æ08 cm2 without MAD and improved to 8Æ61 cm2after MAD application. Increased oropharyn-geal cross-sectional area after MAD application is shown in the Fig. 4a,b. Apnoea⁄ Hypopnea Index decreased gradually from 26Æ7 to 3Æ0 at the sixth month follow-up. Mean oxygen saturation was same as the initial value. There was no significant change in mean oxygen saturation. The PSG values are depicted at Table 1. When it is compared with the first diagnostic PSG, total sleep time decreased in follow-up PSGs. Epworth Sleepiness Scale of patients improved from 14 to 11 at the third month follow-up, and it was 9 at the sixth month.
Oral appliances, particularly MADs, are becoming popular treatment alternatives for OSA patients owing to its ease of use, simplicity of fabrication procedure and low cost (26–28).
Plenty of different designs have been suggested for fabrication of MADs. Mainly, these differences are relevant to the type of construction material, number of pieces, protrusion range of mandible and amount of vertical opening (22, 28).
Mandibular advancement devices could be produced with soft thermoplastic or hard acrylic materials. It has not been suggested any differences of treatment efficacy between MADs fabricated with these two different type materials. Some OAs that permit lateral jaw movement may reduce the risk of adverse effects and improve patient adherence (22, 29–32). In our case, to get rid of the disadvantage of FP, such as deviation of mandible to paralysed part of the face during sleep, a non-adjustable, one-piece MAD which has been fabricated from hard acrylic material is designed.
One of the most important discussions in the liter-ature regarding MAD design is the amount of mandib-ular protrusion. Mehta et al. (33) compared the effect of the protrusion range on the success rate with two different MADs, while one of them did not protrude the mandible (placebo device), the other one did. However, the placebo device did not decrease AHI, while the device with protrusion improved it. Marklund et al. (34) achieved satisfactory results in their patients with MADs at the 41–88% maximum protrusion. Clark et al. (35) achieved striking improvements on respiratory disturbance index from 48Æ4 to 12Æ3, with a modified Herbst appliance which has more than 75% of maxi-mum mandibular protrusion. Walker-Engstro¨m et al. (36) compared efficacy of two different MADs which have different protrusion amounts, 50% and 75%. They determined that the device with 75% protrusion amount was more effective than the other one without side effects. In our case, intermaxillary relationship was
Fig. 3. Fabricated mandibular advancement device.
recorded approximately at the 75% of maximal pro-trusion to avoid the side effects of MAD because of muscle tenderness from FP.
In the literature, another challenging issue on MAD fabrication is the amount of vertical opening. Bloch et al. (29) compared a Herbst appliance that produced 4–6 mm of interincisal opening with a Monobloc device fixed at 5–10 mm of interincisal distance; both of them produced identical levels of protrusion and equally effective on decreasing AHI. Pitsis et al. (37) suggested that the amount of vertical opening induced by the appliance does not have a striking impact on treatment efficacy. Nevertheless, same authors alleged that slightly increased vertical opening could have been tolerated handily by patients. Furthermore, Johal and Battagel (38) noted that the overincreased vertical dimension could decrease the space between the base of the tongue and the posterior pharyngeal wall. Owing to
the consideration of all these arguments, in the present case, existing vertical dimension was increased by almost 5 mm to provide enough space for MAD’s body and prevent any muscular complication on the non-paralysed side.
Several success criteria could be used for the evalu-ation of treatment outcome; however, treatment suc-cess of a MAD is generally defined by a reduction in the total Apnoea–Hypopnea Index to <10, and decrease in the Apnoea–Hypopnea Index by 50% (28, 39, 40).
In our case, while AHI decreased from 26Æ7 to 3Æ0, the mean oxygen saturation did not improve as expected. This may be explained as follows: oxygen saturation is not directly associated with AHI but the pulmonary capacity. Other factors contributing to desaturation include underlying pulmonary disease and duration of the event. Total sleep time also decreased in the follow-up PSG’s. This may be associated with the
(a) (b)
Fig. 4. (a) T1 W axial magnetic resonance (MR) image from retrolingual region before mandibular advancement device (MAD) application, (b) T 1 W axial MR image from retrolingual region after MAD application.
Table 1. Polysomnography (PSG) results of the patient
Total sleep time (min) Sleep efficiency (%) Sleep stages (%) AHI (per h) Oxygen desaturation events (per h) Mean oxygen saturation (%) Lowest oxygen saturation (%) Stage 1 Stage 2 Stage 3 REM
Diagnostic PSG values
434Æ5 90 2Æ8 81Æ6 3Æ2 12Æ5 26Æ7 35Æ2 91Æ8 84Æ0
First night with MAD values
361Æ0 92 5 72 5 18 7Æ1 38 93Æ0 86Æ0
Third month with MAD values
287Æ5 88 5Æ2 77 6Æ2 11 5Æ6 31 92Æ1 84Æ0
Sixth month with MAD values
384Æ0 94 3Æ2 82Æ6 5Æ3 12Æ1 3Æ0 21 91Æ9 85Æ0
teeth. There was no sign of inflammation on gingiva. A small amount of selective grinding was applied on the MAD for elimination of pain.
Conclusion
The MAD therapy decreased the AHI scores of a patient with FP. At the end of a follow-up period of 6 months, the patient did not report any serious complaint except temporary tooth pains.
References
1. Avrahami E, Solomonovich A, Englender M. Axial CT measurements of the cross-sectional area of the oropharynx in adults with obstructive sleep apnea syndrome. Am J Neuroradiol. 1996;17:1107–1111.
2. Guilleminault C, Hill MV, Simmons FB. Obstructive sleep apnea: electromyographic and fiber optic studies. Exp Neurol. 1978;62:48–67.
3. Strollo P, Rogers RM. Current concepts: obstructive sleep apnea. N Engl J Med. 1996;334:99–104.
4. Ayappa I, Rapoport DM. The upper airway in sleep: physiol-ogy of the pharynx. Sleep Med Rev. 2003;7:9–33.
5. Baik UB, Suzuki M, Ikeda K, Sugawara J, Mitani H.
Relationship between cephalometric characteristics and
obstructive sites in obstructive sleep apnea syndrome. Angle Orthod. 2002;72:124–134.
6. Schwab R, Pasirstein M, Pierson R, Mackley A, Hachadoorian R, Arens R et al. Identification of upper airway anatomic risk factors for obstructive sleep apnea with volumetric magnetic resonance imaging. Am J Respir Crit Care Med. 2003;168: 522–530.
7. Schellenberg JB, Mailsin G, Schwab RJ. Physical findings and the risk for obstructive sleep apnea. The importance of oropharyngeal structures. Am J Respir Crit Care Med. 2000;162:740–748.
8. Battagel JM, Johal A, Smith AM, Kotecha B. Postural variation in oropharyngeal dimensions in subjects with sleep disordered breathing: a cephalometric study. Eur J Orthod. 2002;24:263–276.
9. Zonato AI, Martinho FL, Bittencourt LR, Brasil OOC, Grego¢rio LC, Tufik S. Head and neck physical examination:
in patients with bilateral vocal cord paralysis. Eur Arch Otorhinolaryngol. 2003;260:183–185.
13. Lydiatt DD. Medical malpractice and facial nevre paralysis. Arch Otolaryngol Head Neck Surg. 2003;129:50–53. 14. Mavrikakis I. Facial nevre palsy: anatomy, etiology,
evalua-tion, and management. Orbit. 2008;27:466–474.
15. Vasconcelos BCE, Bessa-Nogueira RV, Maurette PE, Carneiro SCSA. Facial nevre paralysis after impacted lower third molar surgery: a literature review and case report. Med Oral Patol Oral Cir Bucal. 2006;11:175–178.
16. Maglio G, Amico RD, Forino M. Results and complications of facial reanimation following cerebellopontine angle surgery. Eur Arch Otorhinolaryngol. 2001;258:45–48.
17. Barthlen GM, Brown LK, Wiland MR, Sadeh JS, Patwari J, Zimmerman M. Comparison of three oral appliances for treatment of severe obstructive sleep apnea syndrome. Sleep Med. 2000;1:299–305.
18. Hudgel DW. Treatment of obstructive sleep apnea. A review. Chest. 1996;109:1346–1358.
19. Hans MG, Nelson S, Luks VG, Lorkovich P, Baek SJ. Comparison of two dental devices for treatment of obstructive sleep apnea syndrome (OSAS). Am J Orthod Dentofac Orthop. 1997;111:562–570.
20. George PT. Selecting sleepdisordered–breathing appliances Biomechanical considerations. J Am Dent Assoc. 2001;132: 339–347.
21. Cistulli PA, Gotsopoulos H, Marklund M, Lowe AA. Treat-ment of snoring and obstructive sleep apnea with mandib-ular repositioning appliances. Sleep Med Rev. 2004;8: 443–457.
22. Chan ASL, Lee RWW, Cistulli PA. Dental appliance treatment for obstructive sleep apnea. Chest. 2007;132:693–699. 23. Stepnowsky CJ, Moore PJ. Nasal CPAP treatment for
obstruc-tive sleep apnea. Developing a new perspecobstruc-tive on dosing strategies and compliance. J Psychosom Res. 2003;54:599– 605.
24. Sullivan CE, Issa FG, Berthon-Jones M, Eves L. Reversal of obstructive sleep apnea by continuous positive airway pressure through the nares. Lancet. 1981;1:862–865. 25. Redline S, Adams N, Strauss ME, Roebuck T, Winters M,
Rosenberg C. Improvment of mild sleep-disordered breathing with CPAP compared with conservative therapy. Am J Respir Crit Care Med. 1998;157:858–865.
26. Cartwright R. What’s new in oral appliances for snoring and sleep apnea: an update. Sleep Med Rev. 2001;5:25–32.
27. Clark GT, Blumenfeld I, Yoffe N, Peled E, Lavie P. A Crossover study comparing the efficacy of continuous positive airway pressure with anterior mandibular positioning devices on patients with obstructive sleep apnea. Chest. 1996;109:1477– 1483.
28. Schmidt-Nowara W, Lowe A, Wiegland L, Cartwright R, Perez-Guerra F, Menn S. Oral appliances for the treatment of snoring and obstructive sleep apnea: a review. Sleep. 1995;18:501–510.
29. Bloch KE, Iseli A, Zhang JN, Xie X, Kaplan V, Stoeckli PW et al. A randomized, controlled crossover trial of two oral appliances for sleep apnea treatment. Am J Respir Crit Care Med. 2000;162:246–251.
30. Rose E, Staats R, Virchow C, Jonas IE. A comparative study of two mandibular advancement appliances for the treatment of obstructive sleep apnea. Eur J Orthod. 2002;24:191–198. 31. Marklund M, Stenlund H, Franklin KA. Mandibular
advance-ment devices in 630 men and women with obstructive sleep apnea and snoring. Chest. 2004;124:1270–1278.
32. Clark GT. Mandibular advancement devices and sleep disor-dered breathing. Sleep Med Rev. 1998;2:163–174.
33. Mehta A, Qian J, Petocz P, Darendeliler MA, Cistulli PA. A randomized, controlled study of a mandibular advancement splint for obstructive sleep apnea. Am J Respir Crit Care Med. 2001;163:1457–1461.
34. Marklund M, Franklin KA, Sahlin C, Lundgren R. The effect of a mandibular advancement device on apneas and sleep in patients with obstructive sleep apnea. Chest. 1998;113:707–713. 35. Clark GT, Arand D, Tong D, Chung E. The effect of anterior
mandibular positioning on obstructive sleep apnea. Am Rev Respir Dis. 1993;147:624–629.
36. Walker-Engstro¨m ML, Ringqvist I, Vestling O, Wilhelmsson B,
Tegelberg A˚ . A prospective randomized study comparing two
different degrees of mandibular advancement with a dental appliance in treatment of severe obstructive sleep apnea. Sleep Breath. 2003;7:119–130.
37. Pitsis AJ, Darendeliler MA, Gotsopoulos H, Petocz P, Cistulli PA. Effect of vertical dimension on efficacy of oral appliance therapy in obstructive sleep apnea. Am J Respir Crit Care Med. 2002;166:860–864.
38. Johal A, Battagel JM. Current princibles in the management of obstructive sleep apnoea with mandibular advancement appliances. Br Dent J. 2001;190:532–536.
39. Marklund M, Persson M, Franklin KA. Treatment success with a mandibular advancement device is related to supine-dependent sleep apnea. Chest. 1998;114:1630–1635. 40. Schmidt-Nowara WW, Meade TE, Hays MB. Treatment of
snoring and obstructive sleep apnea with a dental orthosis. Chest. 1991;99:1378–1385.
41. Fritsch KM, Iseli A, Russi EW, Bloch KE. Side effects of mandibular advancement devices for sleep apnea treatment. Am J Respir Crit Care Med. 2001;164:813–818.
42. Pancer J, Al-Faifi M, Hoffstein V. Evaluation of variable mandibular advancement appliance for treatment of snoring and sleep apnea. Chest. 1999;116:1511–1518.
43. Piskin B, Sentut F, Sevketbeyoglu H, Avsever H, Gunduz K, Kose M et al. Efficacy of a modified mandibular advancement device for a totally edentulous patient with severe obstructive sleep apnea. Sleep Breath. 2010;14:81–85.
Correspondence: Cumhur Sipahi, Tevfik Saglam Cad, GATA, Etlik, 06018, Ankara, Turkey. E-mail: cumhursipahi@yahoo.com
