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Oral appliance therapy for obstructive sleep apnea in multiple system atrophy with floppy epiglottis: a case series of three patients

  • Sleep Breathing Physiology and Disorders • Original Article
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Abstract

Purpose

A recent study demonstrated that continuous positive airway pressure (CPAP) may exacerbate obstructive sleep apnea (OSA) in patients with multiple system atrophy (MSA) and a floppy epiglottis (FE) as the CPAP promotes downward displacement of the epiglottis into the laryngeal inlet. In this case series, we examined the effectiveness of an oral appliance (OA) for treating OSA in three patients with MSA and an FE.

Methods

Patients with MSA were demonstrated to have an FE on fiberoptic laryngoscopy under sedation using intravenous propofol. The therapeutic intervention was fitting an OA. Polysomnography (PSG) was performed subsequently with the OA in place.

Results

In three patients with MSA, some parameters used to assess the severity of OSA improved with an OA. Both apnea–hypopnea index (AHI) and arousal index (ArI) decreased while wearing the OA in two cases while in the third case, apnea index (AI) and cumulative time at peripheral oxygen saturation (SpO2) below 90% (CT90) decreased, but AHI and ArI increased. The only side effects were transient TMJ discomfort, masseter muscle pain, and tooth discomfort.

Conclusion

OA therapy using a two-piece type mandibular advancement device (MAD) may be a useful treatment intervention for patients with OSA who have MSA and FE.

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Data availability

The datasets generated during and/or analyzed during the current study are available from the corresponding author on reasonable request after approval by the Ethical Board of Niigata University Graduate School of Medical and Dental Sciences (Oral Life Science).

References

  1. Gilman S, Low PA, Quinn N, Albanese A, Ben-Shlomo Y, Fowler CJ, Kaufmann H, Klockgether T, Lang AE, Lantos PL, Litvan I, Mathias CJ, Oliver E, Robertson D, Schatz I, Wenning GK (1999) Consensus statement on the diagnosis of multiple system atrophy. J Neurol Sci 163:94–98. https://doi.org/10.1016/s0022-510x(98)00304-9

    Article  CAS  PubMed  Google Scholar 

  2. Gilman S, Wenning GK, Low PA, Brooks DJ, Mathias CJ, Trojanowski JQ, Wood NW, Colosimo C, Durr A, Fowler CJ, Kaufmann H, Klockgether T, Lees A, Poewe W, Quinn N, Revesz T, Robertson D, Sandroni P, Seppi K, Vidailhet M (2008) Second consensus statement on the diagnosis of multiple system atrophy. Neurology 71:670–676. https://doi.org/10.1212/01.wnl.0000324625.00404.15

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  3. Krismer F, Wenning GK (2017) Multiple system atrophy: insights into a rare and debilitating movement disorder. Nat Rev Neurol 13:232–243. https://doi.org/10.1038/nrneurol.2017.26

    Article  PubMed  Google Scholar 

  4. Wenning GK, Geser F, Krismer F, Seppi K, Duerr S, Boesch S, Kollensperger M, Goebel G, Pfeiffer KP, Barone P, Pellecchia MT, Quinn NP, Koukouni V, Fowler CJ, Schrag A, Mathias CJ, Giladi N, Gurevich T, Dupont E, Ostergaard K, Nilsson CF, Widner H, Oertel W, Eggert KM, Albanese A, del Sorbo F, Tolosa E, Cardozo A, Deuschl G, Hellriegel H, Klockgether T, Dodel R, Sampaio C, Coelho M, Djaldetti R, Melamed E, Gasser T, Kamm C, Meco G, Colosimo C, Rascol O, Meissner WG, Tison F, Poewe W, European Multiple System Atrophy Study G (2013) The natural history of multiple system atrophy: a prospective European cohort study. Lancet Neurol 12(264):274. https://doi.org/10.1016/S1474-4422(12)70327-7

    Article  Google Scholar 

  5. Low PA, Reich SG, Jankovic J, Shults CW, Stern MB, Novak P, Tanner CM, Gilman S, Marshall FJ, Wooten F, Racette B, Chelimsky T, Singer W, Sletten DM, Sandroni P, Mandrekar J (2015) Natural history of multiple system atrophy in the USA: a prospective cohort study. Lancet Neurol 14:710–719. https://doi.org/10.1016/S1474-4422(15)00058-7

    Article  PubMed  PubMed Central  Google Scholar 

  6. Munschauer FE, Loh L, Bannister R, Newsom-Davis J (1990) Abnormal respiration and sudden death during sleep in multiple system atrophy with autonomic failure. Neurology 40:677–679. https://doi.org/10.1212/wnl.40.4.677

    Article  CAS  PubMed  Google Scholar 

  7. Sadaoka T, Kakitsuba N, Fujiwara Y, Kanai R, Takahashi H (1996) Sleep-related breathing disorders in patients with multiple system atrophy and vocal fold palsy. Sleep 19:479–484. https://doi.org/10.1093/sleep/19.6.479

    Article  CAS  PubMed  Google Scholar 

  8. Silber MH, Levine S (2000) Stridor and death in multiple system atrophy. Mov Disord 15:699–704. https://doi.org/10.1002/1531-8257(200007)15:4%3c699::aid-mds1015%3e3.0.co;2-l

    Article  CAS  PubMed  Google Scholar 

  9. Ghorayeb I, Yekhlef F, Chrysostome V, Balestre E, Bioulac B, Tison F (2002) Sleep disorders and their determinants in multiple system atrophy. J Neurol Neurosur Ps 72:798–800. https://doi.org/10.1136/jnnp.72.6.798

    Article  CAS  Google Scholar 

  10. Vetrugno R, Provini F, Cortelli P, Plazzi G, Lotti EM, Pierangeli G, Canali C, Montagna P (2004) Sleep disorders in multiple system atrophy: a correlative video-polysomnographic study. Sleep Med 5:21–30. https://doi.org/10.1016/j.sleep.2003.07.002

    Article  PubMed  Google Scholar 

  11. Ferini-Strambi L, Marelli S (2012) Sleep dysfunction in multiple system atrophy. Curr Treat Options Neurol 14:464–473. https://doi.org/10.1007/s11940-012-0189-2

    Article  PubMed  Google Scholar 

  12. Shimohata T, Shinoda H, Nakayama H, Ozawa T, Terajima K, Yoshizawa H, Matsuzawa Y, Onodera O, Naruse S, Tanaka K, Takahashi S, Gejyo F, Nishizawa M (2007) Daytime hypoxemia, sleep-disordered breathing, and laryngopharyngeal findings in multiple system atrophy. Arch Neurol 64:856–861. https://doi.org/10.1001/archneur.64.6.856

    Article  PubMed  Google Scholar 

  13. Ohshima Y, Nakayama H, Matsuyama N, Hokari S, Sakagami T, Sato T, Koya T, Takahashi T, Kikuchi T, Nishizawa M, Shimohata T (2017) Natural course and potential prognostic factors for sleep-disordered breathing in multiple system atrophy. Sleep Med 34:13–17. https://doi.org/10.1016/j.sleep.2017.01.020

    Article  PubMed  Google Scholar 

  14. Shimohata T, Tomita M, Nakayama H, Aizawa N, Ozawa T, Nishizawa M (2011) Floppy epiglottis as a contraindication of CPAP in patients with multiple system atrophy. Neurology 76:1841–1842. https://doi.org/10.1212/WNL.0b013e31821ccd07

    Article  CAS  PubMed  Google Scholar 

  15. (1995) Practice parameters for the treatment of snoring and obstructive sleep apnea with oral appliances. American Sleep Disorders Association. Sleep 18:511–513. https://doi.org/10.1093/sleep/18.6.511

  16. (1999) Sleep-related breathing disorders in adults: recommendations for syndrome definition and measurement techniques in clinical research. The report of an American Academy of Sleep Medicine task force. Sleep 22:667–689

  17. (1992) Eeg arousals: scoring rules and examples: a preliminary report from the sleep disorders atlas task force of the American Sleep Disorders Association. Sleep 15:173–184

  18. Iranzo A, Santamaria J, Tolosa E, Vilaseca I, Valldeoriola F, Marti MJ, Munoz E (2004) Long-term effect of CPAP in the treatment of nocturnal stridor in multiple system atrophy. Neurology 63:930–932. https://doi.org/10.1212/01.wnl.0000137043.76383.a4

    Article  CAS  PubMed  Google Scholar 

  19. Ghorayeb I, Yekhlef F, Bioulac B, Tison F (2005) Continuous positive airway pressure for sleep-related breathing disorders in multiple system atrophy: long-term acceptance. Sleep Med 6:359–362. https://doi.org/10.1016/j.sleep.2004.10.002

    Article  PubMed  Google Scholar 

  20. Shimohata T, Nakayama H, Aizawa N, Nishizawa M (2014) Discontinuation of continuous positive airway pressure treatment in multiple system atrophy. Sleep Med 15:1147–1149. https://doi.org/10.1016/j.sleep.2014.04.013

    Article  PubMed  Google Scholar 

  21. Shimohata T, Aizawa N, Nakayama H, Taniguchi H, Ohshima Y, Okumura H, Takahashi T, Yokoseki A, Inoue M, Nishizawa M (2016) Mechanisms and prevention of sudden death in multiple system atrophy. Parkinsonism Relat Disord 30:1–6. https://doi.org/10.1016/j.parkreldis.2016.04.011

    Article  PubMed  Google Scholar 

  22. Clark GT, Blumenfeld I, Yoffe N, Peled E, Lavie P (1996) A crossover study comparing the efficacy of continuous positive airway pressure with anterior mandibular positioning devices on patients with obstructive sleep apnea. Chest 109:1477–1483. https://doi.org/10.1378/chest.109.6.1477

    Article  CAS  PubMed  Google Scholar 

  23. Ferguson KA, Ono T, Lowe AA, Keenan SP, Fleetham JA (1996) A randomized crossover study of an oral appliance vs nasal-continuous positive airway pressure in the treatment of mild-moderate obstructive sleep apnea. Chest 109:1269–1275. https://doi.org/10.1378/chest.109.5.1269

    Article  CAS  PubMed  Google Scholar 

  24. Ferguson KA, Ono T, Loweal-Majed AAS, Love LL, Fleetham JA (1997) A short-term controlled trial of an adjustable oral appliance for the treatment of mild to moderate obstructive sleep apnoea. Thorax 52:362–368. https://doi.org/10.1136/thx.52.4.362

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  25. Randerath WJ, Heise M, Hinz R, Ruehle KH (2002) An individually adjustable oral appliance vs continuous positive airway pressure in mild-to-moderate obstructive sleep apnea syndrome. Chest 122:569–575. https://doi.org/10.1378/chest.122.2.569

    Article  PubMed  Google Scholar 

  26. Tan YK, L’Estrange PR, Luo YM, Smith C, Grant HR, Simonds AK, Spiro SG, Battagel JM (2002) Mandibular advancement splints and continuous positive airway pressure in patients with obstructive sleep apnoea: a randomized cross-over trial. Eur J Orthod 24:239–249. https://doi.org/10.1093/ejo/24.3.239

    Article  CAS  PubMed  Google Scholar 

  27. Tegelberg A, Wilhelmsson B, Walker-Engstrom ML, Ringqvist M, Andersson L, Krekmanov L, Ringqvist I (1999) Effects and adverse events of a dental appliance for treatment of obstructive sleep apnoea. Swed Dent J 23:117–126

    CAS  PubMed  Google Scholar 

  28. Liu Y, Zeng X, Fu M, Huang X, Lowe AA (2000) Effects of a mandibular repositioner on obstructive sleep apnea. Am J Orthod Dentofacial Orthop 118:248–256. https://doi.org/10.1067/mod.2000.104831

    Article  CAS  PubMed  Google Scholar 

  29. Isono S, Tanaka A, Sho Y, Konno A (1985) Nishino T (1995) Advancement of the mandible improves velopharyngeal airway patency. J Appl Physiol 79:2132–2138. https://doi.org/10.1152/jappl.1995.79.6.2132

    Article  Google Scholar 

  30. Ryan CF, Love LL, Peat D, Fleetham JA, Lowe AA (1999) Mandibular advancement oral appliance therapy for obstructive sleep apnoea: effect on awake calibre of the velopharynx. Thorax 54:972–977. https://doi.org/10.1136/thx.54.11.972

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  31. Iwamoto T, Takata Y, Kitamura N, Hasebe D, Kobayashi T, Saito C (2012) Prognostic predictors on the efficacy of oral appliance therapy for obstructive sleep apnea syndrome. Open J Stomatol 2:210–221

    Article  Google Scholar 

  32. Lowe AA, Sjoholm TT, Ryan CF, Fleetham JA, Ferguson KA, Remmers JE (2000) Treatment, airway and compliance effects of a titratable oral appliance. Sleep 23(Suppl 4):S172-178

    PubMed  Google Scholar 

  33. Menn SJ, Loube DI, Morgan TD, Mitler MM, Berger JS, Erman MK (1996) The mandibular repositioning device: role in the treatment of obstructive sleep apnea. Sleep 19:794–800. https://doi.org/10.1093/sleep/19.10.794

    Article  CAS  PubMed  Google Scholar 

  34. Pancer J, Al-Faifi S, Al-Faifi M, Hoffstein V (1999) Evaluation of variable mandibular advancement appliance for treatment of snoring and sleep apnea. Chest 116:1511–1518. https://doi.org/10.1378/chest.116.6.1511

    Article  CAS  PubMed  Google Scholar 

  35. Rose E, Staats R, Virchow C, Jonas IE (2002) A comparative study of two mandibular advancement appliances for the treatment of obstructive sleep apnoea. Eur J Orthod 24:191–198. https://doi.org/10.1093/ejo/24.2.191

    Article  PubMed  Google Scholar 

  36. Schmidt-Nowara W, Lowe A, Wiegand L, Cartwright R, Perez-Guerra F, Menn S (1995) Oral appliances for the treatment of snoring and obstructive sleep apnea: a review. Sleep 18:501–510. https://doi.org/10.1093/sleep/18.6.501

    Article  CAS  PubMed  Google Scholar 

  37. Orru G, Storari M, Scano A, Piras V, Taibi R, Viscuso D (2020) Obstructive sleep apnea, oxidative stress, inflammation and endothelial dysfunction—an overview of predictive laboratory biomarkers. Eur Rev Med Pharmacol Sci 24:6939–6948. https://doi.org/10.26355/eurrev_202006_21685

    Article  CAS  PubMed  Google Scholar 

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Authors and Affiliations

  1. Division of Reconstructive Surgery for Oral and Maxillofacial Region, Faculty of Dentistry & Graduate School of Medical and Dental Sciences, Niigata University, 2-5274 Gakkocho-Dori, Chuo-ku, Niigata, 951-8514, Japan

    Toshihiko Mikami, Tadaharu Kobayashi & Daichi Hasebe

  2. Department of Dentistry and Oral Surgery, Niigata Medical Center, Niigata, Japan

    Toshihiko Mikami

  3. Department of Respiratory Medicine and Infectious Diseases, Niigata University Graduate School of Medical and Dental Sciences, Niigata, Japan

    Yasuyoshi Ohshima

  4. Department of Neurology, National Hospital Organization Nishiniigata Chuo Hospital, Niigata, Japan

    Tetsuya Takahashi

  5. Department of Neurology, Gifu University Graduate School of Medicine, Gifu, Japan

    Takayoshi Shimohata

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  1. Toshihiko Mikami
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  2. Tadaharu Kobayashi
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  3. Daichi Hasebe
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  4. Yasuyoshi Ohshima
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  5. Tetsuya Takahashi
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  6. Takayoshi Shimohata
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Corresponding author

Correspondence to Toshihiko Mikami.

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Ethics approval

All procedures in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards. This study protocol was reviewed and approved by the Ethical Board of Niigata University Graduate School of Medical and Dental Sciences (Oral Life Science).

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Informed consent was obtained from all individual participants included in the study.

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Mikami, T., Kobayashi, T., Hasebe, D. et al. Oral appliance therapy for obstructive sleep apnea in multiple system atrophy with floppy epiglottis: a case series of three patients. Sleep Breath 27, 213–219 (2023). https://doi.org/10.1007/s11325-022-02607-0

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  • DOI: https://doi.org/10.1007/s11325-022-02607-0

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