Language

English

Publication Date

3-1-2025

Journal

Neurotherapeutics

DOI

10.1016/j.neurot.2025.e00543

PMID

39948022

PMCID

PMC12014419

PubMedCentral® Posted Date

2-12-2025

PubMedCentral® Full Text Version

Post-print

Abstract

Dystonia is the third most common movement disorder and an incapacitating co-morbidity in a variety of neurologic conditions. Dystonia can be caused by genetic, degenerative, idiopathic, and acquired etiologies, which are hypothesized to converge on a "dystonia network" consisting of the basal ganglia, thalamus, cerebellum, and cerebral cortex. In acquired dystonia, focal lesions to subcortical network regions lead to dystonia that can be difficult to manage with canonical treatments, including deep brain stimulation (DBS). While studies in animal models have begun to parse the contribution of individual nodes in the dystonia network, how acquired injury to the cerebellar outflow tracts instigates dystonia; and how network modulation interacts with symptom latency remain unexplored questions. Here, we present an electrolytic lesioning paradigm that bilaterally targets the cerebellar outflow tracts. We found that lesioning these tracts, at the junction of the superior cerebellar peduncles and the medial and intermediate cerebellar nuclei, resulted in transient, acute, and severe dystonia with immobility and fixed posturing similar to status dystonicus. We observed a rapid reduction in dystonia with 1 h of DBS of the centrolateral thalamic nucleus, a first order node in the network downstream of the cerebellar nuclei. In contrast, 1 h of stimulation at a second order node in the short latency, disynaptic projection from the cerebellar nuclei, the striatum, did not show similar rapid modulation of dystonia. Our study introduces a robust paradigm for inducing acute, severe dystonia, and demonstrates that targeted modulation based on network principles powerfully rescues motor behavior. These data inspire the identification of a short latency therapeutic target for acquired dystonia and status dystonicus.

Keywords

Deep Brain Stimulation, Animals, Thalamus, Cerebellum, Male, Dystonic Disorders, Movement, Rats, Disease Models, Animal, Dystonia

Published Open-Access

yes

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