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Article Abstract
Objective: Sialidosis is an inborn error of metabolism. There is evidence that the myoclonic movements observed in this disorder have a cortical origin, but this mechanism does not fully explain the bilaterally synchronous myoclonus activity frequently observed in many patients. We present evidence of a subcortical basis for synchronous myoclonic phenomena.
Methods: Electromyographic investigations were undertaken in two molecularly and biochemically confirmed patients with sialidosis type-1.
Results: The EMG recordings showed clear episodes of bilaterally synchronous myoclonic activity in contralateral homologous muscles. We also observed a high muscular-muscular coherence with near-zero time-lag between these muscles.
Conclusion: The absence of coherence phase lag between the right-and-left homologous muscles during synchronous events indicates that a unilateral cortical source cannot fully explain the myoclonic activity. There must exist a subcortical mechanism for bilateral synchronization accounting for this phenomenon.
Significance: Understanding this mechanism may illuminate cortical-subcortical relationships in myoclonus.
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| http://www.ncbi.nlm.nih.gov/pmc/articles/PMC9253402 | PMC |
| http://dx.doi.org/10.1016/j.cnp.2022.05.004 | DOI Listing |
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