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Alcohol responsiveness in laryngeal dystonia: a survey study

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Abstract

Laryngeal dystonia (LD) is a task-specific focal dystonia of unknown pathophysiology affecting speech production. We examined the demographics of anecdotally reported alcohol use and its effects on LD symptoms using an online survey based on Research Electronic Data Capture (REDCap™) and National Spasmodic Dysphonia Association’s patient registry. From 641 participants, 531 were selected for data analysis, and 110 were excluded because of unconfirmed diagnosis. A total of 406 patients (76.5 %) had LD and 125 (23.5 %) had LD and voice tremor (LD/VT). The consumption of alcohol was reported by 374 LD (92.1 %) and 109 LD/VT (87.2 %) patients. Improvement of voice symptoms after alcohol ingestion was noted by 227 LD (55.9 % of all patients) and 73 LD/VT (58.4 %), which paralleled the improvement observed by patient’s family and/or friends in 214 LD (57.2 %) and 69 LD/VT (63.3 %) patients. The benefits lasted 1–3 h in both groups with the maximum effect after 2 drinks in LD patients (p = 0.002), whereas LD/VT symptoms improved independent of the consumed amount (p = 0.48). Our data suggest that isolated dystonic symptoms, such as in LD, are responsive to alcohol intake and this responsiveness is not attributed to the presence of VT, which is known to have significant benefits from alcohol ingestion. Alcohol may modulate the pathophysiological mechanisms underlying abnormal neurotransmission of γ-aminobutyric acid (GABA) in dystonia and as such provide new avenues for novel therapeutic options in these patients.

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References

  1. (2010) Dietary guidelines for Americans. In: Agriculture UDo, Services UDoHaH (eds) US Government Printing Office, Washington, DC

  2. Ali SO, Thomassen M, Schulz GM, Hosey LA, Varga M, Ludlow CL, Braun AR (2006) Alterations in CNS activity induced by botulinum toxin treatment in spasmodic dysphonia: an H215O PET study. J Speech Lang Hear Res 49:1127–1146

    Article  PubMed  Google Scholar 

  3. Asgeirsson H, Jakobsson F, Hjaltason H, Jonsdottir H, Sveinbjornsdottir S (2006) Prevalence study of primary dystonia in Iceland. Mov Disord 21:293–298

    Article  PubMed  Google Scholar 

  4. Baylor CR, Yorkston KM, Eadie TL, Maronian NC (2007) The psychosocial consequences of BOTOX injections for spasmodic dysphonia: a qualitative study of patients’ experiences. J Voice 21:231–247

    Article  PubMed Central  PubMed  Google Scholar 

  5. Biary N, Koller W (1985) Effect of alcohol on dystonia. Neurology 35:239–243

    Article  CAS  PubMed  Google Scholar 

  6. Blitzer A (2010) Spasmodic dysphonia and botulinum toxin: experience from the largest treatment series. Eur J Neurol 17(Suppl 1):28–30

    Article  PubMed  Google Scholar 

  7. Blitzer A, Brin MF, Stewart CF (1998) Botulinum toxin management of spasmodic dysphonia (laryngeal dystonia): a 12-year experience in more than 900 patients. Laryngoscope 108:1435–1441

    Article  CAS  PubMed  Google Scholar 

  8. Boecker H, Wills AJ, Ceballos-Baumann A, Samuel M, Thompson PD, Findley LJ, Brooks DJ (1996) The effect of ethanol on alcohol-responsive essential tremor: a positron emission tomography study. Ann Neurol 39:650–658

    Article  CAS  PubMed  Google Scholar 

  9. Borges V, Ferraz HB, de Andrade LA (2000) Alcohol-sensitive hereditary essential myoclonus with dystonia: a study of 6 Brazilian patients. Neurol Sci 21:373–377

    Article  CAS  PubMed  Google Scholar 

  10. Brin MF, Blitzer A, Stewart C (1998) Laryngeal dystonia (spasmodic dysphonia): observations of 901 patients and treatment with botulinum toxin. Adv Neurol 78:237–252

    CAS  PubMed  Google Scholar 

  11. de Yebenes JG, Vazquez A, Martinez A, Mena MA, del Rio RM, de Felipe C, del Rio J (1988) Biochemical findings in symptomatic dystonias. Adv Neurol 50:167–175

    PubMed  Google Scholar 

  12. Deuschl G, Wenzelburger R, Loffler K, Raethjen J, Stolze H (2000) Essential tremor and cerebellar dysfunction clinical and kinematic analysis of intention tremor. Brain 123(Pt 8):1568–1580

    Article  PubMed  Google Scholar 

  13. Frucht SJ, Bordelon Y, Houghton WH, Reardan D (2005) A pilot tolerability and efficacy trial of sodium oxybate in ethanol-responsive movement disorders. Mov Disord 20:1330–1337

    Article  PubMed  Google Scholar 

  14. Garibotto V, Romito LM, Elia AE, Soliveri P, Panzacchi A, Carpinelli A, Tinazzi M, Albanese A, Perani D (2011) In vivo evidence for GABA(A) receptor changes in the sensorimotor system in primary dystonia. Mov Disord 26:852–857

    Article  PubMed  Google Scholar 

  15. Gasser T, Bereznai B, Muller B, Pruszak-Seel R, Damrich R, Deuschl G, Oertel WH (1996) Linkage studies in alcohol-responsive myoclonic dystonia. Mov Disord 11:363–370

    Article  CAS  PubMed  Google Scholar 

  16. Gudin M, Vaamonde J, Rodriguez M, Obeso JA (1993) Alcohol sensitive dystonia. Mov Disord 8:122–123

    Article  CAS  PubMed  Google Scholar 

  17. Gurey LE, Sinclair CF, Blitzer A (2013) A new paradigm for the management of essential vocal tremor with botulinum toxin. Laryngoscope 123:2497–2501

    CAS  PubMed  Google Scholar 

  18. Harris P, Taylor R, Theilke R, Payne J, Gonzalez N, Conde J (2009) Research electronic data capture (REDCap)—a metadata driven methodology for providing translational research informatics support. J Biomed Inform 42:377–381

    Article  PubMed Central  PubMed  Google Scholar 

  19. Haslinger B, Erhard P, Dresel C, Castrop F, Roettinger M, Ceballos-Baumann AO (2005) “Silent event-related” fMRI reveals reduced sensorimotor activation in laryngeal dystonia. Neurology 65:1562–1569

    Article  CAS  PubMed  Google Scholar 

  20. Haubenberger D, McCrossin G, Lungu C, Considine E, Toro C, Nahab FB, Auh S, Buchwald P, Grimes GJ, Starling J, Potti G, Scheider L, Kalowitz D, Bowen D, Carnie A, Hallett M (2013) Octanoic acid in alcohol-responsive essential tremor: a randomized controlled study. Neurology 80:933–940

    Article  PubMed Central  CAS  PubMed  Google Scholar 

  21. Hedrich K, Meyer EM, Schule B, Kock N, de Carvalho Aguiar P, Wiegers K, Koelman JH, Garrels J, Durr R, Liu L, Schwinger E, Ozelius LJ, Landwehrmeyer B, Stoessl AJ, Tijssen MA, Klein C (2004) Myoclonus-dystonia: detection of novel, recurrent, and de novo SGCE mutations. Neurology 62:1229–1231

    Article  CAS  PubMed  Google Scholar 

  22. Hess C, Saunders-Pullman R (2006) Movement disorders and alcohol misuse. Addict Biol 11:117–125

    Article  PubMed  Google Scholar 

  23. Izdebski K, Dedo HH, Boles L (1984) Spastic dysphonia: a patient profile of 200 cases. Am J Otolaryngol 5:7–14

    Article  CAS  PubMed  Google Scholar 

  24. Koller WC, Busenbark K, Miner K (1994) The relationship of essential tremor to other movement disorders: report on 678 patients. Essential Tremor Study Group. Ann Neurol 35:717–723

    CAS  Google Scholar 

  25. Kyllerman M, Forsgren L, Sanner G, Holmgren G, Wahlstrom J, Drugge U (1990) Alcohol-responsive myoclonic dystonia in a large family: dominant inheritance and phenotypic variation. Mov Disord 5:270–279

    Article  CAS  PubMed  Google Scholar 

  26. Levy LM, Hallett M (2002) Impaired brain GABA in focal dystonia. Ann Neurol 51:93–101

    Article  CAS  PubMed  Google Scholar 

  27. Lim SC, Kim JS, An JY, Yoon Kang S (2012) Alcohol-responsive writer’s cramp. Intern Med 51:99–101

    Article  PubMed  Google Scholar 

  28. Lof E, Ericson M, Stomberg R, Soderpalm B (2007) Characterization of ethanol-induced dopamine elevation in the rat nucleus accumbens. Eur J Pharmacol 555:148–155

    Article  PubMed  Google Scholar 

  29. Lou JS, Jankovic J (1991) Essential tremor: clinical correlates in 350 patients. Neurology 41:234–238

    Article  CAS  PubMed  Google Scholar 

  30. Ludlow CL, Adler CH, Berke GS, Bielamowicz SA, Blitzer A, Bressman SB, Hallett M, Jinnah HA, Juergens U, Martin SB, Perlmutter JS, Sapienza C, Singleton A, Tanner CM, Woodson GE (2008) Research priorities in spasmodic dysphonia. Otolaryngol Head Neck Surg 139:495–505

    Article  PubMed Central  PubMed  Google Scholar 

  31. Mahloudji M, Pikielny RT (1967) Hereditary essential myoclonus. Brain 90:669–674

    Article  CAS  PubMed  Google Scholar 

  32. Marjanska M, Lehericy S, Valabregue R, Popa T, Worbe Y, Russo M, Auerbach EJ, Grabli D, Bonnet C, Gallea C, Coudert M, Yahia-Cherif L, Vidailhet M, Meunier S (2013) Brain dynamic neurochemical changes in dystonic patients: a magnetic resonance spectroscopy study. Mov Disord 28:201–209

    Article  PubMed Central  CAS  PubMed  Google Scholar 

  33. Meyer TK, Hu A, Hillel AD (2013) Voice disorders in the workplace: productivity in spasmodic dysphonia and the impact of botulinum toxin. Laryngoscope 123(Suppl 6):S1–S14

    Article  CAS  PubMed  Google Scholar 

  34. Mubaidin AF (2000) Alcohol with xylocaine for treatment of eyelid dystonia. Eur J Neurol 7:213–215

    Article  CAS  PubMed  Google Scholar 

  35. Nestoros JN (1980) Ethanol specifically potentiates GABA-mediated neurotransmission in feline cerebral cortex. Science 209:708–710

    Article  CAS  PubMed  Google Scholar 

  36. Novakovic D, Waters HH, D’Elia JB, Blitzer A (2011) Botulinum toxin treatment of adductor spasmodic dysphonia: longitudinal functional outcomes. Laryngoscope 121:606–612

    Article  CAS  PubMed  Google Scholar 

  37. Quinn NP (1996) Essential myoclonus and myoclonic dystonia. Mov Disord 11:119–124

    Article  CAS  PubMed  Google Scholar 

  38. Ridding MC, Sheean G, Rothwell JC, Inzelberg R, Kujirai T (1995) Changes in the balance between motor cortical excitation and inhibition in focal, task specific dystonia. J Neurol Neurosurg Psychiatry 59:493–498

    Article  PubMed Central  CAS  PubMed  Google Scholar 

  39. Schweinfurth JM, Billante M, Courey M (2002) Risk factors and demographics in patients With spasmodic dysphonia. Laryngoscope 112:220–223

    Article  PubMed  Google Scholar 

  40. Simonyan K, Berman BD, Herscovitch P, Hallett M (2013) Abnormal striatal dopaminergic neurotransmission during rest and task production in spasmodic dysphonia. J Neurosci 33:14705–14714

    Article  PubMed Central  CAS  PubMed  Google Scholar 

  41. Simonyan K, Frucht SJ (2013) Long-term effect of sodium oxybate (Xyrem®) in spasmodic dysphonia with vocal tremor. Tremor Other Hyperkinet Mov (N Y)

  42. Simonyan K, Ludlow CL (2010) Abnormal activation of the primary somatosensory cortex in spasmodic dysphonia: an fMRI study. Cereb Cortex 20:2749–2759

    Article  PubMed Central  PubMed  Google Scholar 

  43. Simonyan K, Ludlow CL (2012) Abnormal structure-function relationship in spasmodic dysphonia. Cereb Cortex 22:417–425

    Article  PubMed Central  PubMed  Google Scholar 

  44. Simonyan K, Tovar-Moll F, Ostuni J, Hallett M, Kalasinsky VF, Lewin-Smith MR, Rushing EJ, Vortmeyer AO, Ludlow CL (2008) Focal white matter changes in spasmodic dysphonia: a combined diffusion tensor imaging and neuropathological study. Brain 131:447–459

    Article  PubMed Central  PubMed  Google Scholar 

  45. Sulica L, Louis ED (2010) Clinical characteristics of essential voice tremor: a study of 34 cases. Laryngoscope 120:516–528

    Article  PubMed  Google Scholar 

  46. Valente EM, Misbahuddin A, Brancati F, Placzek MR, Garavaglia B, Salvi S, Nemeth A, Shaw-Smith C, Nardocci N, Bentivoglio AR, Berardelli A, Eleopra R, Dallapiccola B, Warner TT (2003) Analysis of the epsilon-sarcoglycan gene in familial and sporadic myoclonus-dystonia: evidence for genetic heterogeneity. Mov Disord 18:1047–1051

    Article  PubMed  Google Scholar 

  47. White LJ, Klein AM, Hapner ER, Delgaudio JM, Hanfelt JJ, Jinnah HA, Johns MM 3rd (2011) Coprevalence of tremor with spasmodic dysphonia: a case-control study. Laryngoscope 121:1752–1755

    Article  PubMed Central  PubMed  Google Scholar 

  48. Ziemann U, Lonnecker S, Paulus W (1995) Inhibition of human motor cortex by ethanol. A transcranial magnetic stimulation study. Brain 118(Pt 6):1437–1446

    PubMed  Google Scholar 

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Acknowledgments

We thank Estee Rubien-Thomas, BA, and Peter Velickovic for their assistance in data collection. This study was funded by the grant from the National Institute on Deafness and Other Communication Disorders, National Institutes of Health (R01DC012545) to KS. ClinicalTrials.gov Identifier: NCT01961297. DNK was supported by a research fellowship grant from the Foundation for Surgery Reg Worcester Research Fellowship Scholarship, Royal Australasian College of Surgeons.

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  1. Department of Neurology, Icahn School of Medicine at Mount Sinai, One Gustave L. Levy Place, Box 1137, New York, NY, 10029, USA

    Diana N. Kirke, Steven J. Frucht & Kristina Simonyan

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  1. Diana N. Kirke
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  2. Steven J. Frucht
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  3. Kristina Simonyan
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Correspondence to Kristina Simonyan.

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Kirke, D.N., Frucht, S.J. & Simonyan, K. Alcohol responsiveness in laryngeal dystonia: a survey study. J Neurol 262, 1548–1556 (2015). https://doi.org/10.1007/s00415-015-7751-2

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  • DOI: https://doi.org/10.1007/s00415-015-7751-2

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