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March 31, 2020; 94 (13) Resident & Fellow Section

Mystery Case: Symptomatic isolated tongue tremor of cortical origin due to stroke

View ORCID ProfileSmathorn Thakolwiboon, Doungporn Ruthirago, Pavis Laengvejkal, Henrik Wilms
First published March 18, 2020, DOI: https://doi.org/10.1212/WNL.0000000000009174
Smathorn Thakolwiboon
From the Department of Neurology (S.T., H.W.), Texas Tech University Health Sciences Center, Lubbock; Department of Neurology (D.R.), Massachusetts General Hospital, Harvard Medical School, Boston; and Department of Neurology (P.L.), Bumrungrad International Hospital, Bangkok, Thailand.
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  • ORCID record for Smathorn Thakolwiboon
Doungporn Ruthirago
From the Department of Neurology (S.T., H.W.), Texas Tech University Health Sciences Center, Lubbock; Department of Neurology (D.R.), Massachusetts General Hospital, Harvard Medical School, Boston; and Department of Neurology (P.L.), Bumrungrad International Hospital, Bangkok, Thailand.
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Pavis Laengvejkal
From the Department of Neurology (S.T., H.W.), Texas Tech University Health Sciences Center, Lubbock; Department of Neurology (D.R.), Massachusetts General Hospital, Harvard Medical School, Boston; and Department of Neurology (P.L.), Bumrungrad International Hospital, Bangkok, Thailand.
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Henrik Wilms
From the Department of Neurology (S.T., H.W.), Texas Tech University Health Sciences Center, Lubbock; Department of Neurology (D.R.), Massachusetts General Hospital, Harvard Medical School, Boston; and Department of Neurology (P.L.), Bumrungrad International Hospital, Bangkok, Thailand.
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Mystery Case: Symptomatic isolated tongue tremor of cortical origin due to stroke
Smathorn Thakolwiboon, Doungporn Ruthirago, Pavis Laengvejkal, Henrik Wilms
Neurology Mar 2020, 94 (13) 591-592; DOI: 10.1212/WNL.0000000000009174

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An 82-year-old right-handed man with atrial fibrillation presented with abnormal tongue movement after awakening. Neurologic examination showed tremulous tongue movements (video 1) without other neurologic deficit. Brain MRI showed acute infarction in the left inferolateral precentral and inferior frontal gyri (figure) without abnormality in brainstem, cerebellum, or basal ganglia. Similar to the only previously reported case of tongue tremor with right hand weakness due to stroke, tongue tremor in our case also spontaneously improved without specific treatment.1

Video 1

Tongue tremor. Rhythmic alternating protrusion–retrogression movements worsen with tongue protrusion. Improvement was seen on day 2 of hospitalization. At 3-week follow-up, tongue tremor was almost completely resolved.Download Supplementary Video 1 via http://dx.doi.org/10.1212/009174_Video_1

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Figure Brain MRI

Brain MRI shows 2 hyperintensity foci on the diffusion-weighted image (A) with corresponding hypointensity area on the apparent diffusion coefficient sequences in the left inferolateral precentral and inferior frontal gyri (B). These findings are suggestive for acute infarction.

This case provides evidence that tongue tremor can develop after sudden unilateral cortical function loss in the precentral or inferior frontal gyri.

Mystery Case responses: Symptomatic isolated tongue tremor of cortical origin due to stroke

The Mystery Case series was initiated by the Neurology® Resident & Fellow Section to develop the clinical reasoning skills of trainees. Residency programs, medical student preceptors, and individuals were invited to use this Mystery Case as an educational tool. Responses were solicited through a group email sent to the American Academy of Neurology Consortium of Neurology Residents and Fellows and through social media.

A total of 259 participants responded to this mystery case. A large majority, 77%, correctly localized the lesion to the CNS.1 Similarly, 63% correctly identified the left inferolateral precentral gyrus stroke. However, only 27% noted the left inferior frontal gyrus stroke. This is likely due to confusion with the left angular gyrus, which was the most common incorrect answer. Twenty-nine percent of respondents correctly answered that spontaneous full recovery is expected. Seven respondents answered all 3 questions correctly.

This mystery case highlights isolated tongue tremor due to stroke, which is very rare. As discussed by the authors, one other example of poststroke isolated tongue tremor has been described.1 Both the previously described patient and this patient exhibited full recovery. Isolated tongue tremor from other etiologies is also rare. Previous cases have been reported due to tumors, following gamma-knife surgery, in Wilson disease, and from neoplastic Hu antibodies.2

Ariel M. Lyons-Warren, MD, PhD

Baylor College of Medicine, Texas Children's Hospital

Study funding

No targeted funding reported.

Disclosure

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

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Table
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Footnotes

  • Go to Neurology.org/N for full disclosures. Funding information and disclosures deemed relevant by the authors, if any, are provided at the end of the article.

  • Teaching slides links.lww.com/WNL/B62

  • © 2020 American Academy of Neurology

References

  1. 1.↵
    1. Pandey S,
    2. Sarma N,
    3. Jain S
    . Tongue tremor in acute cortical infarct. Mov Disord Clin Pract 2016;3:214–215.
    OpenUrl
  2. 2.↵
    1. Ure RJ,
    2. Dhanju S,
    3. Lang AE,
    4. Fasano A
    . Unusual tremor syndromes: know in order to recognise. J Neurol Neurosurg Psychiatry 2016;87:1191–1203.
    OpenUrlAbstract/FREE Full Text

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