Dissecting the Links Between Cerebellum and Dystonia

Malone, Ailish; Manto, Mario; Hass, Chris J.

doi:10.1007/s12311-014-0601-4

Cited by 31 publications

(16 citation statements)

References 25 publications

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“…These findings were reminiscent of our results in which we found that mouse cerebellum and motor cortex anatomy was abnormal. These results lead us to suspect that the motor defect of ZIKV-infected mice was at least partly, if not totally, a result of abnormal cerebella and motor cortices, considering the strong correlation between motor disorders and cerebellar defects (Malone et al, 2014). In Brazil, it is estimated that > 1800 babies were born with microcephaly.…”

Section: Discussionmentioning

confidence: 99%

Visual and Motor Deficits in Grown-up Mice with Congenital Zika Virus Infection

Cui

Zou

et al. 2017

EBioMedicine

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Human infants with congenital Zika virus (ZIKV) infection exhibit a range of symptoms including microcephaly, intracranial calcifications, macular atrophy and arthrogryposis. More importantly, prognosis data have lagged far behind the recent outbreak of ZIKV in 2015. In this work, we allow congenitally ZIKV-infected mice to grow into puberty. These mice exhibited motor incoordination and visual dysfunctions, which can be accounted by anatomical defects in the retina and cerebellar cortex. In contrary, anxiety level of the ZIKV-infected mice is normal. The spectrum of anatomical and behavioral deficits is consistent across different mice. Our data provided evidence that may help predict the public health burden in terms of prognosis of ZIKV-related congenital brain malformations in an animal model. Our study provided behavioral evaluation for the prognosis of congenital ZIKV infection and provides a platform for screening and evaluation of drugs candidates and treatment aiming at improving regeneration of infected neurons to prevent sequelae caused by ZIKV infection of fetus.

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Section: Discussionmentioning

confidence: 99%

Visual and Motor Deficits in Grown-up Mice with Congenital Zika Virus Infection

Cui

Zou

et al. 2017

EBioMedicine

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“…16 While studies with animal models clearly provide additional evidence that disturbances to the basal ganglia can induce dystonia, 15 they also provide evidence that disturbances to the cerebellum can cause dystonia. 15,18 In a network model, dystonia could result from dysfunction affecting one or more of the brain regions within the network or from abnormal communication between brain regions within the network. As a role for the cerebellum and associated thalamocortical circuits is increasingly recognized, one possibility is that a dysfunctional interaction between basal ganglia and cerebellar networks causes dystonia (figure).…”

Section: Dystonia As a Network Disordermentioning

confidence: 99%

Dystonia

Berman

Jinnah

2015

Neur Clin Pract

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SummaryThere has been considerable progress in our understanding of dystonia over the last century. Growing recognition of dystonia has enhanced awareness of its diverse motor phenomenology and brought attention to the importance that nonmotor features may play in this disorder, once considered to be purely motor. Using the latest technologies in human genetics, new genetic links are being discovered at an ever-quickening pace and expanding our knowledge of the disorder's complex pathogenesis. Furthermore, as we gain clearer insight into the pathophysiology of dystonia and an appreciation of the involvement of dysfunction outside the basal ganglia, dystonia has been increasingly viewed as a network disorder. Here we briefly discuss some of the recent noteworthy advances.

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“…The pathophysiology of focal dystonia is still not entirely clear. Although dystonia is considered a basal ganglia disorder (Bhatia and Marsden 1994; DeLong and Wichmann 2007), recent studies indicate that the cerebellum may also be involved in this condition (Sadnicka et al, 2012; Prudente et al, 2014; Malone et al, 2014). …”

Section: Introductionmentioning

confidence: 99%

Effects of cerebellar theta-burst stimulation on arm and neck movement kinematics in patients with focal dystonia

Bologna

Paparella

Fabbrini

et al. 2016

Clinical Neurophysiology

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Objective To investigate the cerebellar inhibitory influence on the primary motor cortex in patients with focal dystonia using a cerebellar continuous theta-burst stimulation protocol (cTBS) and to evaluate any relationship with movement abnormalities. Methods Thirteen patients with focal hand dystonia, 13 patients with cervical dystonia and 13 healthy subjects underwent two sessions: (i) cTBS over the cerebellar hemisphere (real cTBS) and (ii) cTBS over the neck muscles (sham cTBS). The effects of cerebellar cTBS were quantified as excitability changes in the contralateral primary motor cortex, as well as possible changes in arm and neck movements in patients. Results Real cerebellar cTBS reduced the excitability in the contralateral primary motor cortex in healthy subjects and in patients with cervical dystonia, though not in patients with focal hand dystonia. There was no correlation between changes in primary motor cortex excitability and arm and neck movement kinematics in patients. There were no changes in clinical scores or in kinematic measures, after either real or sham cerebellar cTBS in patients. Conclusions The reduced cerebellar inhibitory modulation of primary motor cortex excitability in focal dystonia may be related to the body areas affected by dystonia as opposed to being a widespread pathophysiological abnormality. Significance The present study yields information on the differential role played by the cerebellum in the pathophysiology of different focal dystonias.

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Dissecting the Links Between Cerebellum and Dystonia

Cited by 31 publications

References 25 publications

Visual and Motor Deficits in Grown-up Mice with Congenital Zika Virus Infection

Visual and Motor Deficits in Grown-up Mice with Congenital Zika Virus Infection

Dystonia

Effects of cerebellar theta-burst stimulation on arm and neck movement kinematics in patients with focal dystonia

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