Objective and Clinically Feasible Analysis of Diffusion MRI Data can Help Predict Dystonia After Neonatal Brain Injury

Chintalapati, Keerthana; Miao, Hanyang; Mathur, Amit M.; Neil, Jeff; Aravamuthan, Bhooma R.

doi:10.1016/j.pediatrneurol.2020.11.011

Cited by 5 publications

(2 citation statements)

References 20 publications

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“…Dystonia is typically associated with striatal injury, particularly striatal cholinergic interneuron (ChIN) excitation which has been viewed as a unifying mechanism of non-CP dystonia etiologies like DYT1 and DYT6. (Bonsi et al, 2011; Chintalapati et al, 2020; Eskow Jaunarajs et al, 2019, 2015; Gemperli et al, 2023; Pappas et al, 2015) By measuring leg adduction variability and amplitude, we have also shown that chronic striatal ChIN excitation can cause dystonia. (Gemperli et al, 2023) Finally, we have shown that a rat model of hypoxic-ischemic injury at human-equivalent term gestation demonstrates an increased number of striatal ChINs.…”

Section: Introductionmentioning

confidence: 79%

Mice born preterm develop gait dystonia and reduced cortical parvalbumin immunoreactivity

Gemperli,

Folorunso,

Norin

et al. 2024

Preprint

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Preterm birth leading to cerebral palsy (CP) is the most common cause of childhood dystonia, a movement disorder that is debilitating and often treatment refractory. Dystonia has been typically associated with dysfunction of striatal cholinergic interneurons, but clinical imaging data suggests that cortical injury may best predict dystonia following preterm birth. Furthermore, abnormal sensorimotor cortex inhibition has been found in many studies of non-CP dystonias. To assess the potential for a cortical etiology of dystonia following preterm birth, we developed a new model of preterm birth in mice. Noting that term delivery in mice on a C57BL/6J background is embryonic day 19.1 (E19.1), we induced preterm birth at the limits of pup viability at embryonic day (E) 18.3, equivalent to human 22 weeks gestation. Mice born preterm demonstrate display clinically validated metrics of dystonia during gait (leg adduction amplitude and variability) and also demonstrate reduced parvalbumin immunoreactivity in the sensorimotor cortex, suggesting dysfunction of cortical parvalbumin-positive inhibitory interneurons. Notably, reduced parvalbumin immunoreactivity or changes in parvalbumin-positive neuronal number were not observed in the striatum. These data support the association between cortical dysfunction and dystonia following preterm birth. We propose that our mouse model of preterm birth can be used to study this association and potentially also study other sequelae of extreme prematurity.

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Section: Introductionmentioning

confidence: 79%

Mice born preterm develop gait dystonia and reduced cortical parvalbumin immunoreactivity

Gemperli,

Folorunso,

Norin

et al. 2024

Preprint

Self Cite

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“…While the majority of patients (87%) had cerebral abnormalities on imaging (including PVL, cortical gray matter injury, evidence of stroke, and cerebral malformations), dystonic/dyskinetic CP patients had either non-specific changes (7/15; delayed myelination or volume loss) or normal imaging (4/15) findings [ 43 ]. Along these lines, a more recent study looked at imaging in patients with acute HIE at day 4–5 of life; they found that evidence of cytotoxic edema (ADC map on magnetic resonance imaging (MRI)) to the thalamus and striatum was highly predictive of subsequent dystonia [ 44 ]. Notably, diffuse low grade cytotoxic edema, in contrast to the dense cytotoxic edema associated with ischemic stroke, may lead to more subtle volume loss and perhaps explains the findings noted above associating mild imaging abnormalities with dystonic CP.…”

Section: Acquired Lesion-associated and Isolated Idiopathic Focal/seg...mentioning

confidence: 99%

Function and dysfunction of the dystonia network: an exploration of neural circuits that underlie the acquired and isolated dystonias

Gill,

Nguyen,

Hull

et al. 2023

Dystonia

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Dystonia is a highly prevalent movement disorder that can manifest at any time across the lifespan. An increasing number of investigations have tied this disorder to dysfunction of a broad “dystonia network” encompassing the cerebellum, thalamus, basal ganglia, and cortex. However, pinpointing how dysfunction of the various anatomic components of the network produces the wide variety of dystonia presentations across etiologies remains a difficult problem. In this review, a discussion of functional network findings in non-mendelian etiologies of dystonia is undertaken. Initially acquired etiologies of dystonia and how lesion location leads to alterations in network function are explored, first through an examination of cerebral palsy, in which early brain injury may lead to dystonic/dyskinetic forms of the movement disorder. The discussion of acquired etiologies then continues with an evaluation of the literature covering dystonia resulting from focal lesions followed by the isolated focal dystonias, both idiopathic and task dependent. Next, how the dystonia network responds to therapeutic interventions, from the “geste antagoniste” or “sensory trick” to botulinum toxin and deep brain stimulation, is covered with an eye towards finding similarities in network responses with effective treatment. Finally, an examination of how focal network disruptions in mouse models has informed our understanding of the circuits involved in dystonia is provided. Together, this article aims to offer a synthesis of the literature examining dystonia from the perspective of brain networks and it provides grounding for the perspective of dystonia as disorder of network function.

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Evolving understanding of CP phenotypes: the importance of dystonia

Lewis,

Aravamuthan,

Fehlings

et al. 2024

Pediatr Res

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Objective and Clinically Feasible Analysis of Diffusion MRI Data can Help Predict Dystonia After Neonatal Brain Injury

Cited by 5 publications

References 20 publications

Mice born preterm develop gait dystonia and reduced cortical parvalbumin immunoreactivity

Mice born preterm develop gait dystonia and reduced cortical parvalbumin immunoreactivity

Function and dysfunction of the dystonia network: an exploration of neural circuits that underlie the acquired and isolated dystonias

Evolving understanding of CP phenotypes: the importance of dystonia

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