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
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.
Appendix Authors
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
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