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Comparison of Oropharyngeal Dysphagia Before and After Botulinum Toxin Injection in Cervical Dystonia

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Abstract

Cervical dystonia (CD) is the most common form of focal dystonia with Botulinum neurotoxin (BoNT) being a frequent method of treatment. Dysphagia is a common side effect of BoNT treatment for CD. Instrumental evaluation of swallowing in CD using standardized scoring for the videofluoroscopic swallowing study (VFSS) and validated and reliable patient-reported outcomes measures is lacking in the literature. (1) to determine if BoNT injections change instrumental findings of swallowing function using the Modified Barium Swallow Impairment Profile (MBSImP) in individuals with CD; (2) to determine if BoNT injections change self-perception of the psychosocial handicapping effects of dysphagia in individuals with CD, using the Dysphagia Handicap Index (DHI); (3) to determine the effect of BoNT dosage on instrumental swallowing evaluation and self-reported swallowing outcomes measures. 18 subjects with CD completed a VFSS and the DHI before and after BoNT injection. There was a significant increase in pharyngeal residue for pudding consistency after BoNT injection, p = 0.015. There were significant positive associations between BoNT dosage and self-perception of the physical attributes of the handicapping effect of dysphagia, the grand total score and patient self-reported severity of dysphagia on the DHI; p = 0.022; p = 0.037; p = 0.035 respectively. There were several significant associations between changes in MBSImP scores and BoNT dose. Pharyngeal efficiency of swallowing may be affected by BoNT for thicker consistencies. Individuals with CD perceive greater physical handicapping effects of dysphagia with increased amounts of BoNT units and have greater self-perceptions of dysphagia severity with increased amounts of BoNT units.

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References

  1. Defazio G, Jankovic J, Giel J, Papapetropoulos S. Descriptive epidemiology of cervical dystonia. Tremor Other Hyperkinet Mov. 2013. https://doi.org/10.7916/D8OC4TGJ.

    Article  Google Scholar 

  2. Dauer W, Burke R, Greene P, Fahn S. Current concepts on the clinical features, aetiology and management of idiopathic cervical dystonia. Brain. 1998;121:547–60. https://doi.org/10.1093/brain/121.4.547.

    Article  PubMed  Google Scholar 

  3. Jankovic J, Schwartz K, Donovan D. Botulinum toxin treatment of cranial-cervical dystonia, spasmodic dysphonia, other focal dystonias and hemifacial spasm. J Neurol Neurosurg Psychiatry. 1990;53:633–9. https://doi.org/10.1136/jnnp.53.8.633.

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  4. Fahn S. The varied clinical expressions of dystonia. Neuro Clinics. 1984;2(3):541–4. https://doi.org/10.1016/S0733-8619(18)31090-9.

    Article  CAS  Google Scholar 

  5. Nutt JG, Muenter MD, Aronson A, Kurland LT. Melton LJ Epidemiology of focal and generalized dystonia in rochester. Minnesota Mov Disord. 1988;3(3):188–94. https://doi.org/10.1002/mds.870030302.

    Article  CAS  PubMed  Google Scholar 

  6. Chan J, Brin M, Fahn S. Idiopathic cervical dystonia:clinical characteristics. Mov Disord. 1991;6(2):119–26. https://doi.org/10.1002/mds.870060206.

    Article  CAS  PubMed  Google Scholar 

  7. Berman BD, Junker J, Shelton E, Sillau SH, Jinnah HA, Perlmutter JS, Espay AJ, et al. Psychiatric associations of adult-onset focal dystonia phenotypes. J Neurol Neurosurg Psychiatry. 2017;88:595–602. https://doi.org/10.1136/jnnp-2016-315461.

    Article  PubMed  Google Scholar 

  8. Klingelhoefer L, Martino D, Martinez-Martin P, Sauerbier A, Rizos A, Jost W, et al. Nonmotor symptoms and focal cervical dystonia: observations from 102 patients. Basal Ganglia. 2014;4:117–20. https://doi.org/10.1016/j.baga.2014.10.002.

    Article  Google Scholar 

  9. Camfield L, Ben-Shlomo Y, Warner T. Impact of cervical dystonia on quality of life. Mov Disord. 2002;17(4):831–45. https://doi.org/10.1002/mds.10127.

    Article  Google Scholar 

  10. Ben-Shlomo Y, Canfield L, Warner T. What are the determinants of quality of life in people with cervical dystonia? J Neurol Neurosurg Psychiatry. 2002;72:608–14.

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  11. LeDoux MS, Brady KA. Secondary cervical dystonia associated with structural lesions of the central nervous system. Mov Disord. 2003;18(1):60–9. https://doi.org/10.1002/mds.10301.

    Article  PubMed  Google Scholar 

  12. Filip P, Lungu OV, Shaw D, Kasparek T, Bares M. The mechanisms of movement control and time estimation in cervical dystonia patients. Neural Plast. 2013. https://doi.org/10.1155/2013/908741.

    Article  PubMed  PubMed Central  Google Scholar 

  13. Sadnicka A, Patani B, Saifee TA, Kassavetis P, Parees I, Korlipara P, Bhatia KP, Rothwell JC, Galea JM, Edwards MJ. Normal motor adaptation in cervical dystonia: a fundamental cerebellar computation is intact. Cerebellum. 2014;13:558–67. https://doi.org/10.1007/s12311-014-0569-0.

    Article  PubMed  PubMed Central  Google Scholar 

  14. Filip P, Gllea C, Lehericy S, Bertasi E, Popa T, Marecek R, Lungu OV, Kasparek T, Vanicek J, Bares M. Disruption in cerebellar and basal ganglia networks during a visuospacial task in cervical dystonia. Mov Disord. 2017;32(5):757–68. https://doi.org/10.1002/mds.26930.

    Article  PubMed  Google Scholar 

  15. Porcacchia P, Alvarez de Toledo P, Rodriguez-Baena A, Martin-Rodriguez JF, Palomar FJ, Vargas-Gonzalez L, Jesus S, Koch G, Mir P. Abnormal cerebellar connectivity and plasticity in isolated cervical dystonia. Pulse One. 2019. https://doi.org/10.1371/journal.pone.0211367.

    Article  Google Scholar 

  16. Munchau A, Corna S, Gresty A, Bhatia KP, Palmer JD, Dressler D, Quinn NP, Rothwell JC, Bronstein AM. Abnormal interaction between vestibular and voluntary head control in patients with spasmodic torticollis. Brain. 2001;124:47–59. https://doi.org/10.1093/brain/124.1.47.

    Article  CAS  PubMed  Google Scholar 

  17. Obermann M, Vollrath C, de Greiff A, Gizewski ER, Diener HC, Hallett M, Maschke M. Sensory disinhibition on passive movement in cervical dystonia. Mov Disord. 2010;25(15):2627–33. https://doi.org/10.1002/mds.23321.

    Article  PubMed  Google Scholar 

  18. Leube B, Rudnicki D, Ratzlaff T, Kessler KR, Benecke B, Auburger G. Idiopathic torsion dystonia: assignment of a gene to chromosome 18p in a German family with adult onset, autosomal dominant inheritance and purely focal distribution. Hum Mol Genet. 1996;5(10):1673–7. https://doi.org/10.1093/hmg/5.10.1673.

    Article  CAS  PubMed  Google Scholar 

  19. Xiao J, Uitti RL, Zhao Y, Vemula SR, Perlmutter JS, Wszolek ZK, Maraganore DM, Auberger G, Leube B, Lehnhoff K, LeDoux M. Mutations in C1Z1 cause adult onset primary cervical dystonia. Ann Neurol. 2012;71:458–69. https://doi.org/10.1002/ana.23547.

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  20. Eidelberg D, Moeller JR, Antonini A, Kazumata K, Nakamura T, Dhawan V, Spetsieris P, deLeon D, Bressman SB, Fahn S. Functional brain networks in DYT1 dystonia. Ann Neurol. 1998;44:303–12. https://doi.org/10.1002/ana.410440304.

    Article  CAS  PubMed  Google Scholar 

  21. Marras C, Van den Eeden SK, Fross RD, Benedict-Albers KS, Klingman J, Leimpeter AD, Nelson LM, et al. Minimum incidence of primary cervical dystonia in a multiethnic health care population. Neurology. 2007;69:676–80. https://doi.org/10.1212/01.wnl.0000267425.51598.c9.

    Article  CAS  PubMed  Google Scholar 

  22. Contarino M, Van Den Dool J, Balash Y, Bhatia K, Giladi N, Kolman JH, Lokkegaard A, et al. Clinical practice: evidence-based recommendations for the treatment of cervical dystonia with botulinum toxin. Front Neurol. 2017. https://doi.org/10.3389/fneur.2017.00035.

    Article  PubMed  PubMed Central  Google Scholar 

  23. Haussermann P, Marczoch S, Klinger C, Landgrebe M, Conrad B, Ceballos-Baumann A. Long-term follow-up of cervical dystonia patients treated with botulinum toxin A. Mov Disord. 2004;19(3):303–8. https://doi.org/10.1002/mds.10659.

    Article  PubMed  Google Scholar 

  24. Patel S, Martino D. Cervical dystonia:from pathophysiology to pharmacotherapy. Behav Neurol. 2013;26:275–82. https://doi.org/10.3233/BEN-2012-120270.

    Article  PubMed  PubMed Central  Google Scholar 

  25. Yahalom G, Fay-Karmon T, Livneh V, Israeli-Korn S, Ephraty L, Hassin-Baer S. Botulinum injections for idiopathic cervical dystonia: a longitudinal study. Neurotox Res. 2021;39:1352–9. https://doi.org/10.1007/s12640-021-00378-2.

    Article  CAS  PubMed  Google Scholar 

  26. Kessler KR, Skutta M, Benecke R. Long-term treatment of cervical dystonia with botulinum toxin A: efficacy, safety, and antibody frequency. J Neurol. 1999;246:265–74. https://doi.org/10.1007/s004150050345.

    Article  CAS  PubMed  Google Scholar 

  27. Riski JE, Horner J, Nashold BS. Swallowing function in patients with spasmodic torticollis. Neurology. 1990;40:1443–5. https://doi.org/10.1212/WNL.40.9.1443.

    Article  CAS  PubMed  Google Scholar 

  28. Ertekin C, Aydogdu I, Secil Y, Kiylioglu N, Tarlaci S, Ozdemirkiran T. Oropharyngeal swallowing in craniocervical dystonia. J Neurol Neurosurg Psychiatry. 2002;73:406–11.

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  29. Comella CL, Tanner CM, DeFoor-Hill L, Smith C. Dysphagia after botulinum toxin injections for spasmodic torticollis: clinical and radiological findings. Neurology. 1992;42:1307–10. https://doi.org/10.1212/WNL.42.7.1307.

    Article  CAS  PubMed  Google Scholar 

  30. Kutschenko A, Klietz M, Paracka L, Kollewe K, Schulte-Sutum A, Janssen T, Schrader C, Wegner F, Dressler D. Dysphagia in cervical dystonia patients receiving optimized botulinum toxin therapy: a single-center retrospective cohort study. Neuro Preclin Neurol Stud. 2020;127:1161–5. https://doi.org/10.1007/s00702-020-02220-z.

    Article  CAS  Google Scholar 

  31. Vilanova TFD, Borges V, Ferraz HB. Specific characteristics of the medical history of swallowing before and after application of botulinum toxin in patients with cervical dystonia. Clinics. 2019;74:e776. https://doi.org/10.6061/clinics/2019/e776.

    Article  PubMed  PubMed Central  Google Scholar 

  32. Martin-Harris B, Brodsky MB, Michele Y, Castell DO, Schleicher M, Sandidge J, Maxwell R, Blair J. MBS measurement tool for swallow impairment—MBSImp: establishing a standard. Dysphagia. 2008;23(4):392–405. https://doi.org/10.1007/s00455-008-9185-9.

    Article  PubMed  PubMed Central  Google Scholar 

  33. Silbergleit A, Schultz L, Jacobson B, Beardsley T, Johnson A. The dysphagia handicap index: development and validation. Dysphagia. 2012;27:46–52. https://doi.org/10.1007/s00455-011-9336-2.

    Article  PubMed  Google Scholar 

  34. Timmerman AA, Speyer R, Heijnen BJ, Klijn-Zwijnenberg IR. Psychometric characteristics of health-related quality-of-life questionnaires in oropharyngeal dysphagia. Dysphagia. 2014;29:183–98. https://doi.org/10.1007/s00455-013-9511-8.

    Article  PubMed  Google Scholar 

  35. Rosenbek JC, Robbins J, Roecker EB, Coyle JL, Wood JL. A penetration-aspiration scale. Dysphagia. 1996;11:93–8. https://doi.org/10.1007/BF00417897.

    Article  CAS  PubMed  Google Scholar 

  36. Ramirez-Castaneda J, Jankovic J, Comella C, Dashtipour K, Fernandez HH, Mari Z. Diffusion, spread and migration of botulinum toxin. Mov Disord. 2013;28(3):1775–83. https://doi.org/10.1002/mds.25582.

    Article  CAS  PubMed  Google Scholar 

  37. Brodsky MA, Swope DM, Grimes D. Diffusion of botulinum toxins. Tremor Other Hyperkinetic Mov. 2012. https://doi.org/10.7916/2FD88W3C1M.

    Article  Google Scholar 

  38. Borodic GE, Joseph M, Fay L, Cozzolino D, Ferrante RJ. Botulinum A toxin for the treatment of spasmodic torticollis: dysphagia and regional spread. Head Neck. 1990;12:392–8. https://doi.org/10.1002/hed.2880120504.

    Article  CAS  PubMed  Google Scholar 

  39. Hok P, Veverka T, Hlustik P, Nevrly M, Kanovsky P. The central effects of botulinum toxin in dystonia and spasticity. Toxins. 2021;13:1–21. https://doi.org/10.3390/toxins13020155.

    Article  CAS  Google Scholar 

  40. Weise D, Weise CM, Naumann M. Central effects of botulinum neurotoxin-evidence from human studies. Toxins. 2019;11:1–11. https://doi.org/10.3390/toxins11010021.

    Article  CAS  Google Scholar 

  41. Munchau A, Good CD, McGowen S, Quinn NP, Palmer JD, Bhatia KP. Prospective study of swallowing function in patients with cervical dystonia undergoing selective peripheral denervation. J Neurol Neurosurg Psychiatry. 2001;71:67–72.

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  42. Consky ES, Lang AE. Clinical assessments of patients with cervical dystonia. In: Jankovic J, Hallet M, editors. Therapy with botulinum toxin. Marcel Dekker Inc: New York; 1994. p. 211–37.

    Google Scholar 

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This study was funded by the Research Proposal Development Program, Henry Ford Health.

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Correspondence to Alice K. Silbergleit.

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Silbergleit, A.K., Isabell, K., Turnbull, J. et al. Comparison of Oropharyngeal Dysphagia Before and After Botulinum Toxin Injection in Cervical Dystonia. Dysphagia 38, 1421–1429 (2023). https://doi.org/10.1007/s00455-023-10571-4

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