A 15-year-old boy had acute-onset encephalopathy following a viral prodrome, associated with frequent, dramatic yawning without EEG change (video 1). There were no focal deficits on examination. CSF protein elevation (1.72 g/L) without pleocytosis, brain MRI (figure), and significant improvement following methylprednisolone were consistent with acute disseminated encephalomyelitis (ADEM). Pathologic yawning is described in other demyelinating diseases, neuromyelitis optica spectrum disorder and multiple sclerosis, but is rarely reported with ADEM. Although prior reports postulated that brainstem and hypothalamic lesions are responsible,1 yawning is a complex reflex mediated by both supratentorial and infratentorial structures; its precise localization is not yet defined.2
Video 1
Pathologic yawning. During a video EEG, the patient was noted to have frequent, exaggerated yawning, a behavior previously reported by his parents.Download Supplementary Video 1 via http://dx.doi.org/10.1212/009595_Video_1
Axial views show normal T1 (A), asymmetric T2 (B), and fluid-attenuated inversion recovery (FLAIR) (C) hyperintensity of the dorsal brainstem involving the reticular formation (arrowheads) at the level of cranial nerves V, VI, and VII nuclei, and T2 and FLAIR hyperintensity of the bilateral, left more than right, insula (arrows).
Study funding
No targeted funding reported.
Disclosure
V. Birca and C. Saint-Martin report no relevant disclosures. K.A. Myers receives/has received research funding from the Research Institute of the McGill University Health Center, Citizens United for Research in Epilepsy, Savoy Foundation, and Dravet Canada. Go to Neurology.org/N for full disclosures.
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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.
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