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The close relationship between life-threatening breathing disorders and urine storage dysfunction in multiple system atrophy

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Abstract

Survival of multiple system atrophy (MSA) depends on whether a variety of sleep-related breathing problems as well as autonomic failure (AF) occur. Since the brainstem lesions that cause respiratory and autonomic dysfunction overlap with each other, these critical manifestations might get worse in parallel. If so, the detection of AF, which is comparatively easy, might be predictive of a latent life-threatening breathing disorder. In 15 patients with MSA, we performed autonomic function tests composed of postural challenges and administered a questionnaire on bladder condition, as well as polysomnography and laryngoscopy during wakefulness and under anesthesia. Polysomnographic variables such as the apnea-hypopnea index (AHI) and oxygen saturation (SpO2) and the findings of laryngoscopy were compared with the degree of cardiac and urinary autonomic dysfunction. AHI, mean SpO2 and the lowest SpO2 showed significant correlations with urine storage dysfunction. In addition, patients with vocal cord abductor paralysis (VCAP) or central sleep apnea (CSA) contributing to nocturnal sudden death had more severe storage disorders than those without. On the other hand, no significant relationship between polysomnographic variables and orthostatic hypotension was observed except in the case of mean SpO2. These results indicate that life-threatening breathing disorders have a close relationship with AF, and especially urine storage dysfunction. Therefore, longitudinal assessment of deterioration of the storage function might be useful for predicting the latent progress of VCAP and CSA.

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References

  1. Watanabe H, Saito Y, Terao S, Ando T, Kachi T, Mukai E et al (2002) Progression and prognosis in multiple system atrophy. An analysis of 230 Japanese patients. Brain 125:1070–1083

    Article  PubMed  Google Scholar 

  2. O’Sullivan SS, Massey LA, Williams DR, Silveira-Moriyama L, Kempster PA, Holton JL et al (2008) Clinical outcomes of progressive supranuclear palsy and multiple system atrophy. Brain 131:1362–1372

    Article  PubMed  Google Scholar 

  3. 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

    CAS  PubMed  Google Scholar 

  4. Brown LK (1994) Respiratory dysfunction in Parkinson’s disease. Clin Chest Med 15:715–727

    CAS  PubMed  Google Scholar 

  5. Silber MH, Levine S (2000) Stridor and death in multiple system atrophy. Mov Disord 15:699–704

    Article  CAS  PubMed  Google Scholar 

  6. Isozaki E, Naito A, Horiguchi S, Kawamura R, Hayashida T, Tanabe H (1996) Early diagnosis and stage classification of vocal cord abductor paralysis in patients with multiple system atrophy. J Neurol Neurosurg Psychiatry 60:399–402

    Article  CAS  PubMed  Google Scholar 

  7. Benarroch EE (2007) Brainstem respiratory control: substrates of respiratory failure of multiple system atrophy. Mov Disord 22:155–161

    Article  PubMed  Google Scholar 

  8. Benarroch EE (2007) Brainstem respiratory chemosensitivity. New insights and clinical implications. Neurology 68:2140–2143

    Article  PubMed  Google Scholar 

  9. Tsuda T, Onodera H, Okabe S, Kikuchi Y, Itoyama Y (2002) Impaired chemosensitivity to hypoxia is a marker of multiple system atrophy. Ann Neurol 52:367–371

    Article  CAS  PubMed  Google Scholar 

  10. Bannister R, Gibson W, Michaels L, Oppenheimer DR (1981) Laryngeal abductor paralysis in multiple system atrophy. A report on three necropsied cases, with observations on the laryngeal muscles and the nuclei ambigui. Brain 104:351–368

    Article  CAS  PubMed  Google Scholar 

  11. Isozaki E, Matsubara S, Hayashida T, Oda M, Hirai S (2000) Morphometric study of nucleus ambiguus in multiple system atrophy presenting with vocal cord abductor paralysis. Clin Neuropathol 19:213–220

    CAS  PubMed  Google Scholar 

  12. Isono S, Shiba K, Yamaguchi M, Tanaka A, Hattori T, Konno A et al (2001) Pathogenesis of laryngeal narrowing in patients with multiple system atrophy. J Physiol 536:237–249

    Article  CAS  PubMed  Google Scholar 

  13. Merlo IM, Occhini A, Pacchetti C, Alfonsi E (2002) Not paralysis, but dystonia causes stridor in multiple system atrophy. Neurology 58:649–652

    PubMed  Google Scholar 

  14. Jin K, Okabe S, Chida K, Abe N, Kimpara T, Ohnuma A et al (2007) Tracheostomy can fatally exacerbate sleep-disordered breathing in multiple system atrophy. Neurology 68:1618–1621

    Article  CAS  PubMed  Google Scholar 

  15. Gilman S, Wenning GK, Low PA, Brooks DJ, Mathias CJ, Trojanowski JQ et al (2008) Second consensus statement on the diagnosis of multiple system atrophy. Neurology 71:670–676

    Article  CAS  PubMed  Google Scholar 

  16. Benarroch EE (1999) Central neurotransmitters and neuromodulators in cardiovascular regulation. Management of postural hypotension. In: Mathias CJ, Bannister R (eds) Autonomic failure: a textbook of clinical disorders of the autonomic nervous system, 4th edn. Oxford University Press, New York, pp 37–44

    Google Scholar 

  17. Kavia RB, Dasgupta R, Fowler CJ (2005) Functional imaging and the central control of the bladder. J Comp Neurol 493:27–32

    Article  PubMed  Google Scholar 

  18. de Groat WC, Araki I, Vizzard MA, Yoshiyama M, Yoshimura N, Sugaya K et al (1998) Developmental and injury induced plasticity in the micturition reflex pathway. Behav Brain Res 92:127–140

    Article  PubMed  Google Scholar 

  19. Sakakibara R, Nakazawa K, Shiba K, Nakajima Y, Uchiyama T, Yoshiyama M et al (2002) Firing patterns of micturition-related neurons in the pontine storage centre in cats. Auton Neurosci 99:24–30

    Article  PubMed  Google Scholar 

  20. Ito T, Sakakibara R, Nakazawa K, Uchiyama T, Yamamoto T, Liu Z et al (2006) Effects of electrical stimulation of the raphe area on the micturition reflex in cats. Neuroscience 142:1273–1280

    Article  CAS  PubMed  Google Scholar 

  21. Köllensperger M, Geser F, Seppi K, Stampfer-Kountchev M, Sawires M, Scherfler C et al (2008) Red flags for multiple system atrophy. Mov Disord 23:1093–1099

    Article  PubMed  Google Scholar 

  22. Wenning GK, Tison F, Seppi K, Sampaio C, Diem A, Yekhlef F et al (2004) Development and validation of the Unified Multiple System Atrophy Rating Scale (UMSARS). Mov Disord 19:1391–1402

    Article  PubMed  Google Scholar 

  23. Sakakibara R, Shinotoh H, Uchiyama T, Sakuma M, Kashiwado M, Yoshiyama M et al (2001) Questionnaire-based assessment of pelvic organ dysfunction in Parkinson’s disease. Auton Neurosci 92:76–85

    Article  CAS  PubMed  Google Scholar 

  24. The Report of an American Academy of Sleep Medicine Task Force (1999) Sleep-related breathing disorders in adults: recommendations for syndrome definition and measurement techniques in clinical research. Sleep 22:667–689

    Google Scholar 

  25. Maurer JT, Stuck BA, Hein G, Hörmann K (2000) Videoendoscopic assessment of uncommon sites of upper airway obstruction during sleep. Sleep Breath 4:131–136

    Article  PubMed  Google Scholar 

  26. Shimohata T, Shinoda H, Nakayama H, Ozawa T, Terajima K, Yoshizawa H et al (2007) Daytime hypoxemia, sleep-disordered breathing, and laryngopharyngeal findings in multiple system atrophy. Arch Neurol 64:856–861

    Article  PubMed  Google Scholar 

  27. Uzawa A, Sakakibara R, Tamura N, Asahina M, Yamanaka Y, Uchiyama T et al (2005) Laryngeal abductor paralysis can be a solitary manifestation of multiple system atrophy. J Neurol Neurosurg Psychiatry 76:1739–1741

    Article  CAS  PubMed  Google Scholar 

  28. Glass GA, Josephs KA, Ahlskog JE (2006) Respiratory insufficiency as the primary presenting symptom of multiple-system atrophy. Arch Neurol 63:978–981

    Article  PubMed  Google Scholar 

  29. Deguchi K, Ikeda K, Shimamura M, Urai Y, Tsukaguchi M, Touge T et al (2007) Assessment of autonomic dysfunction of multiple system atrophy with laryngeal abductor paralysis as an early manifestation. Clin Neurol Neurosurg 109:892–895

    Article  PubMed  Google Scholar 

  30. Isozaki E, Miyamoto K, Osanai R, Hayashida T, Tanabe H (1991) Clinical studies of 23 patients with multiple system atrophy presenting with vocal cord paralysis. Rinsho Shinkeigaku 31:249–254

    CAS  PubMed  Google Scholar 

  31. Sun QJ, Berkowitz RG, Goodchild AK, Pilowsky PM (2002) Serotonin inputs to inspiratory laryngeal motoneurons in the rat. J Comp Neurol 451:91–98

    Article  CAS  PubMed  Google Scholar 

  32. Berkowitz RG, Sun QJ, Goodchild AK, Pilowsky PM (2005) Serotonin inputs to laryngeal constrictor motoneurons in the rat. Laryngoscope 115:105–109

    Article  CAS  PubMed  Google Scholar 

  33. Richerson GB, Wang W, Hodges MR, Dohle CI, Diez-Sampedro A (2005) Homing in on the specific phenotype(s) of central respiratory chemoreceptors. Exp Physiol 90:259–266

    Article  CAS  PubMed  Google Scholar 

  34. Benarroch EE, Schmeichel AM, Low PA, Parisi JE (2004) Involvement of medullary serotonergic groups in multiple system atrophy. Ann Neurol 55:418–422

    Article  PubMed  Google Scholar 

  35. Yamamoto T, Sakakibara R, Uchiyama T, Liu Z, Ito T, Awa Y et al (2009) Questionnaire-based assessment of pelvic organ dysfunction in multiple system atrophy. Mov Disord 24:972–978

    Article  PubMed  Google Scholar 

  36. Ghani KR, Pilcher J, Rowland J, Rowland D, Patel U, Nassiri D, Anson K (2008) Portable ultrasonography and bladder volume accuracy—a comparative study using three-dimensional ultrasonography. Urology 72:24–28

    Article  PubMed  Google Scholar 

  37. Yabe I, Soma H, Takei A, Fujiki N, Yanagihara T, Sasaki H (2006) MSA-C is the predominant clinical phenotype in Japan: analysis of 142 patients with probable MSA. J Neurol Sci 249:115–121

    Article  PubMed  Google Scholar 

  38. Ozawa T, Paviour D, Quinn NP, Josephs KA, Sangha H, Kilford L et al (2004) The spectrum of pathological involvement of the striatonigral and olivopontocerebellar systems in multiple system atrophy: clinicopathological correlations. Brain 127:2657–2671

    Article  PubMed  Google Scholar 

  39. Jellinger KA, Seppi K, Wenning GK (2005) Grading of neuropathology in multiple system atrophy: proposal for a novel scale. Mov Disord 20(Suppl 12):S29–S36

    Article  PubMed  Google Scholar 

  40. Schmidt C, Herting B, Prieur S, Junghanns S, Schweitzer K, Globas C et al (2008) Autonomic dysfunction in different subtypes of multiple system atrophy. Mov Disord 23:1766–1772

    Article  PubMed  Google Scholar 

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Authors and Affiliations

  1. Department of Gastroenterology and Neurology, Faculty of Medicine, Kagawa University, 1750-1 Ikenobe, Miki-cho, Kita-gun, Kagawa, 761-0793, Japan

    Kazushi Deguchi, Kazuyo Ikeda, Masago Tsukaguchi, Yoshiteru Urai, Kazutaka Kurokohchi & Tsutomu Masaki

  2. Department of Otorhinolaryngology, Faculty of Medicine, Kagawa University, 1750-1 Ikenobe, Miki-cho, Kita-gun, Kagawa, 761-0793, Japan

    Rieko Goto & Nozomu Mori

  3. Department of Health Sciences, Faculty of Medicine, Kagawa University, 1750-1 Ikenobe, Miki-cho, Kita-gun, Kagawa, 761-0793, Japan

    Tetsuo Touge

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  1. Kazushi Deguchi
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  2. Kazuyo Ikeda
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  9. Tsutomu Masaki
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Correspondence to Kazushi Deguchi.

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Deguchi, K., Ikeda, K., Goto, R. et al. The close relationship between life-threatening breathing disorders and urine storage dysfunction in multiple system atrophy. J Neurol 257, 1287–1292 (2010). https://doi.org/10.1007/s00415-010-5508-5

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  • DOI: https://doi.org/10.1007/s00415-010-5508-5

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