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April 30, 2019; 92 (18) Video NeuroImages

Tongue base retraction and airway obstruction in drug-induced oromandibular dystonia

Gohei Yamada, Yoshino Ueki, Kenji Okita, Noriyuki Matsukawa
First published April 29, 2019, DOI: https://doi.org/10.1212/WNL.0000000000007409
Gohei Yamada
From the Departments of Neurology (G.Y., K.O., N.M.) and Rehabilitation Medicine (Y.U.), Nagoya City University Graduate School of Medical Science, Japan.
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Yoshino Ueki
From the Departments of Neurology (G.Y., K.O., N.M.) and Rehabilitation Medicine (Y.U.), Nagoya City University Graduate School of Medical Science, Japan.
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Kenji Okita
From the Departments of Neurology (G.Y., K.O., N.M.) and Rehabilitation Medicine (Y.U.), Nagoya City University Graduate School of Medical Science, Japan.
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Noriyuki Matsukawa
From the Departments of Neurology (G.Y., K.O., N.M.) and Rehabilitation Medicine (Y.U.), Nagoya City University Graduate School of Medical Science, Japan.
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Citation
Tongue base retraction and airway obstruction in drug-induced oromandibular dystonia
Gohei Yamada, Yoshino Ueki, Kenji Okita, Noriyuki Matsukawa
Neurology Apr 2019, 92 (18) 859; DOI: 10.1212/WNL.0000000000007409

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A 29-year-old man began haloperidol therapy (1.5 mg/d) for severe agitation due to mental stress. He developed jaw opening dystonia at 1 month, and paroxysmal stridor and dyspnea at rest occurred at 5 months. A fiberscope revealed both involuntary tongue base retraction and airway obstruction (video), suggesting diagnosis of drug-induced oromandibular dystonia. Haloperidol was discontinued. At follow-up 1 year following cessation of haloperidol, he reported gradual disappearance of tongue base retraction within 1 month and the jaw opening dystonia within 4 month. Although tongue base retraction is an uncommon form of oromadibular dystonia,1,2 it is a serious complication of therapy.

Video 1

A fiberscope showed the larynx at rest. Subsequently, the base of the tongue suddenly retracted and the airway became obstructed for 7 seconds, which was assumed to be a result of the contraction of both the hypoglossus and styloglossus muscles. Specifically, the contraction of the vocalis muscle preceded.Download Supplementary Video 1 via http://dx.doi.org/10.1212/007409_Video_1

Author contributions

G. Yamada: drafting/revising the manuscript, data acquisition, study concept or design, analysis or interpretation of data, accepts responsibility for conduct of research and final approval, study supervision. Y. Ueki: drafting/revising the manuscript, accepts responsibility for conduct of research and final approval, acquisition of data. K. Okita: drafting/revising the manuscript, data acquisition, accepts responsibility for conduct of research and final approval, acquisition of data. N. Matsukawa: drafting/revising the manuscript, accepts responsibility for conduct of research and final approval, study supervision.

Study funding

No targeted funding reported.

Disclosure

The authors report no disclosures relevant to the manuscript. Go to Neurology.org/N for full disclosures.

Footnotes

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  • © 2019 American Academy of Neurology

References

  1. 1.↵
    1. Mehanna R,
    2. Jankovic J
    . Respiratory disorders associated with dystonia. Mov Disord 2012;27:1816–1819.
    OpenUrlCrossRefPubMed
  2. 2.↵
    1. Sankhla C,
    2. Lai EC,
    3. Jankovic J
    . Peripherally induced oromandibular dystonia. J Neurol Neurosurg Psychiatry 1998;65:722–728.
    OpenUrlAbstract/FREE Full Text

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