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Orthostatic Jaw Tremor, a Diagnostic Challenge for Neurologists

Received: 27 June 2020     Accepted: 22 July 2020     Published: 31 August 2020
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Abstract

Jaw tremor is usually associated with tremor or other abnormal involuntary movements affecting additional body parts, but isolated jaw tremor is a rare clinical presentation. A 40-year-old man presented with history of paroxysmal jaw tremor for 3 years with no other neurological symptoms, psychiatric conditions, or a family history of neurological disorders. The tremor occurred only in the resting jaw position, with 10 episodes per day on an average, and lasted from 10 minutes to hours. It disappeared with any voluntary activation of the masseter muscle and did not change (in amplitude or frequency) with distraction maneuvers. The remainder of neurologic examination was unremarkable. The patient underwent extensive testing, including contrast-enhanced brain magnetic resonance imaging, blood tests and neuropsychiatric evaluation by two different neuropsychiatrists which failed to reveal the underlying cause. The needle electromyography examination revealed involuntary, intermittent, and synchronized muscular contractions in both masseter muscles, at a frequency of 13 to 14 Hz. No changes were discovered in the other muscles. He was treated with propranolol, primidone, valproic acid, diazepam, and amitriptyline, but the response was not significant. Injection of 25 units of botulinum toxin type A in the masseter muscle on both sides of the fac was effective five days after the injection. No adverse effects or masseter weakness were observed.

Published in Clinical Neurology and Neuroscience (Volume 4, Issue 3)
DOI 10.11648/j.cnn.20200403.14
Page(s) 61-65
Creative Commons

This is an Open Access article, distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution and reproduction in any medium or format, provided the original work is properly cited.

Copyright

Copyright © The Author(s), 2020. Published by Science Publishing Group

Keywords

Paroxysmal Isolated Jaw Tremor, Primary Orthostatic Tremor, Botulinum Toxin

References
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[2] P. Gonzalez-Alegre, P. Kelkar, T. L Rodnitzky. Isolated high-frequency jaw tremor relieved by botulinum toxin injections. Mov Disord 2006; 21: 1049–1050.
[3] P. F. Sowman, P. D. Thompson, T. S. Miles. Investigation of an unusual, high-frequency jaw tremor with coherence analysis. Mov Disord 2008; 23: 441-443.
[4] Koster B, Lauk M, Timmer J, et al. Involvement of cranial muscles and high intermuscular coherence in orthostatic tremor. Ann Neurol 1999; 45: 384-388.
[5] A. Schrag, K. Bhatia, P. Brownm C. D. Marsden. An unusual jaw tremor with characteristics of primary orthostatic tremor. Mov Disord 1999; 14: 528–530.
[6] E. D. Louis, E. Rios, L. M. Applegate, N. C. Hernandez, H. F Andrews. Jaw tremor: prevalence and clinical correlates in three essential tremor case samples. Mov Disord 2006; 21: 1872–1878.
[7] S. A. Schneider, K. P Bhatia. The entity of jaw tremor and dystonia. Mov Disord 2007; 22: 1491–1495.
[8] Ure RJ, Dhanju S, Lang AE, et al. Unusual tremor syndromes: know in order to recognise. Journal of Neurology, Neurosurgery & Psychiatry 2016; 87: 1191-1203.
[9] D. Junge, J. R Rosenberg, D. M Halliday. Physiological tremor in human jaw-muscle system. Arch Oral Bio 1998; 43: 45–54.
[10] MATLAB. version 9.4.0.813654 (R2018a). Natick, Massachusetts: The MathWorks Inc.; 2018.
[11] P. F. Sowman, KS. Turker. Methods of time and frequency domain examination of physiological tremor in the human jaw. Hum Mov Sci 2005; 24: 657-666.
[12] K. M. Heilman. Orthostatic tremor. Arch Neurol 1984; 41: 880-881.
[13] S. Jaberzadeh, P. Brodin, S. C Flavel, N. J O’Dwyer, M. A Nordstrom, T. S Miles. Pulsatile control of the human masticatory muscles. J Physiol 2003; 547 (Pt. 2: 613-620.
[14] F. Vial, P. Kassavetis, S. Merchant, D. Haubenberger, M. Hallett. How to do an electrophysiological study of tremor. Clin Neurophysiol Pract [Internet]. 2019; 4: 134–42. Available from: https://doi.org/10.1016/j.cnp.2019.06.002.
[15] Hess CW, Pullman SL. Tremor: Clinical Phenomenology and Assessment Techniques. Tremor and Other Hyperkinetic Movements. 2012; 2 (0): 02.
[16] Pal PK. Electrophysiologic Evaluation of Psychogenic Movement Disorders. J Mov Disord [Internet]. 2011 Apr 30; 4 (1): 21–32. Available from: http://e-jmd.org/journal/view.php?doi=10.14802/jmd.11004.
[17] D. M. M Mehndiratta, D. M Kumar, D. S Pandey. Tremor Differentiation on Surface Electromyography. IOSR J Dent Med Sci [Internet]. 2016 Jul; 15 (07): 23–9. Available from: http://iosrjournals.org/iosr-jdms/papers/Vol15-Issue 7/Version-12/E1507122329.pdf.
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    Rafaela Magalhães Britto Pacheco de Moraes, Fernando José de Souza, João Fellipe Santos Tatsch, Livia Souza Santos, José Furtado Alves Junior, et al. (2020). Orthostatic Jaw Tremor, a Diagnostic Challenge for Neurologists. Clinical Neurology and Neuroscience, 4(3), 61-65. https://doi.org/10.11648/j.cnn.20200403.14

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    ACS Style

    Rafaela Magalhães Britto Pacheco de Moraes; Fernando José de Souza; João Fellipe Santos Tatsch; Livia Souza Santos; José Furtado Alves Junior, et al. Orthostatic Jaw Tremor, a Diagnostic Challenge for Neurologists. Clin. Neurol. Neurosci. 2020, 4(3), 61-65. doi: 10.11648/j.cnn.20200403.14

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    AMA Style

    Rafaela Magalhães Britto Pacheco de Moraes, Fernando José de Souza, João Fellipe Santos Tatsch, Livia Souza Santos, José Furtado Alves Junior, et al. Orthostatic Jaw Tremor, a Diagnostic Challenge for Neurologists. Clin Neurol Neurosci. 2020;4(3):61-65. doi: 10.11648/j.cnn.20200403.14

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  • @article{10.11648/j.cnn.20200403.14,
      author = {Rafaela Magalhães Britto Pacheco de Moraes and Fernando José de Souza and João Fellipe Santos Tatsch and Livia Souza Santos and José Furtado Alves Junior and Maria Sheila Guimarães Rocha},
      title = {Orthostatic Jaw Tremor, a Diagnostic Challenge for Neurologists},
      journal = {Clinical Neurology and Neuroscience},
      volume = {4},
      number = {3},
      pages = {61-65},
      doi = {10.11648/j.cnn.20200403.14},
      url = {https://doi.org/10.11648/j.cnn.20200403.14},
      eprint = {https://article.sciencepublishinggroup.com/pdf/10.11648.j.cnn.20200403.14},
      abstract = {Jaw tremor is usually associated with tremor or other abnormal involuntary movements affecting additional body parts, but isolated jaw tremor is a rare clinical presentation. A 40-year-old man presented with history of paroxysmal jaw tremor for 3 years with no other neurological symptoms, psychiatric conditions, or a family history of neurological disorders. The tremor occurred only in the resting jaw position, with 10 episodes per day on an average, and lasted from 10 minutes to hours. It disappeared with any voluntary activation of the masseter muscle and did not change (in amplitude or frequency) with distraction maneuvers. The remainder of neurologic examination was unremarkable. The patient underwent extensive testing, including contrast-enhanced brain magnetic resonance imaging, blood tests and neuropsychiatric evaluation by two different neuropsychiatrists which failed to reveal the underlying cause. The needle electromyography examination revealed involuntary, intermittent, and synchronized muscular contractions in both masseter muscles, at a frequency of 13 to 14 Hz. No changes were discovered in the other muscles. He was treated with propranolol, primidone, valproic acid, diazepam, and amitriptyline, but the response was not significant. Injection of 25 units of botulinum toxin type A in the masseter muscle on both sides of the fac was effective five days after the injection. No adverse effects or masseter weakness were observed.},
     year = {2020}
    }
    

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    AU  - Rafaela Magalhães Britto Pacheco de Moraes
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    AB  - Jaw tremor is usually associated with tremor or other abnormal involuntary movements affecting additional body parts, but isolated jaw tremor is a rare clinical presentation. A 40-year-old man presented with history of paroxysmal jaw tremor for 3 years with no other neurological symptoms, psychiatric conditions, or a family history of neurological disorders. The tremor occurred only in the resting jaw position, with 10 episodes per day on an average, and lasted from 10 minutes to hours. It disappeared with any voluntary activation of the masseter muscle and did not change (in amplitude or frequency) with distraction maneuvers. The remainder of neurologic examination was unremarkable. The patient underwent extensive testing, including contrast-enhanced brain magnetic resonance imaging, blood tests and neuropsychiatric evaluation by two different neuropsychiatrists which failed to reveal the underlying cause. The needle electromyography examination revealed involuntary, intermittent, and synchronized muscular contractions in both masseter muscles, at a frequency of 13 to 14 Hz. No changes were discovered in the other muscles. He was treated with propranolol, primidone, valproic acid, diazepam, and amitriptyline, but the response was not significant. Injection of 25 units of botulinum toxin type A in the masseter muscle on both sides of the fac was effective five days after the injection. No adverse effects or masseter weakness were observed.
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Author Information
  • Department of Neurology, Hospital Santa Marcelina, S?o Paulo, Brazil

  • Department of Neuropsychiatry, Hospital Santa Marcelina, S?o Paulo, Brazil

  • Department of Neurophysiology, Hospital Santa Marcelina, S?o Paulo, Brazil

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