Resting Beta Hypersynchrony in Secondary Dystonia and Its Suppression During Pallidal Deep Brain Stimulation in DYT3+ Lubag Dystonia

Whitmer, Diane; Solages, Camille de; Hill, Bruce C.; Yu, Hong; Brontë‐Stewart, Helen

doi:10.1111/j.1525-1403.2012.00519.x

Cited by 17 publications

(8 citation statements)

References 16 publications

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“…For example, deep brain stimulation of the GPi results in dystonic motor improvement in cerebral palsy patients (34,35). GPi deep brain stimulation may be effective in cerebral palsy patients by suppressing pathologic β oscillatory activity, as has been demonstrated in patients with primary dystonia (36) and Parkinson's disease (37). …”

Section: Discussionmentioning

confidence: 92%

Long-term increase in coherence between the basal ganglia and motor cortex after asphyxial cardiac arrest and resuscitation in developing rats

Aravamuthan

Shoykhet

2015

Pediatr Res

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BACKGROUND The basal ganglia are vulnerable to injury during cardiac arrest. Movement disorders are a common morbidity in survivors. Yet, neuronal motor network changes post-arrest remain poorly understood. METHODS We compared function of the motor network in adult rats that, during postnatal week 3, underwent 9.5 min of asphyxial cardiac arrest (n = 9) or sham intervention (n = 8). Six months after injury, we simultaneously recorded local field potentials (LFP) from the primary motor cortex (MCx) and single neuron firing and LFP from the rat entopeduncular nucleus (EPN), which corresponds to the primate globus pallidus pars interna. Data were analyzed for firing rates, power, and coherence between MCx and EPN spike and LFP activity. RESULTS Cardiac arrest survivors display chronic motor deficits. EPN firing rate is lower in cardiac arrest survivors (19.5 ± 2.4 Hz) compared with controls (27.4 ± 2.7 Hz; P < 0.05). Cardiac arrest survivors also demonstrate greater coherence between EPN single neurons and MCx LFP (3—100 Hz; P < 0.001). CONCLUSIONS This increased coherence indicates abnormal synchrony in the neuronal motor network after cardiac arrest. Increased motor network synchrony is thought to be antikinetic in primary movement disorders. Characterization of motor network synchrony after cardiac arrest may help guide management of post-hypoxic movement disorders.

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Section: Discussionmentioning

confidence: 92%

Long-term increase in coherence between the basal ganglia and motor cortex after asphyxial cardiac arrest and resuscitation in developing rats

Aravamuthan

Shoykhet

2015

Pediatr Res

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“…Deep brain stimulation of the globus pallidus internus (GPi-DBS) has been used in several patients with XDP and severe generalized dystonia [ 18 , 33 , 34 , 35 , 36 , 37 , 38 , 39 ]. The effects of bilateral GPi-DBS are always immediate and striking [ 38 ] and lead to a 34–95% improvement in dystonia symptoms, as determined using BFM-DRS [ 18 , 33 , 34 , 35 , 36 , 37 , 38 , 39 ]. In general, GPi-DBS may have beneficial effects on dystonia symptoms in patients with XDP, but not on parkinsonian symptoms [ 35 ].…”

Section: Treatmentsmentioning

confidence: 99%

“…Notably, a study using intraoperative pallidal microdialysis suggested that GPi-DBS might suppress dystonia symptoms by enhancing local gamma-aminobutyric acid (GABA) release in the GPi [ 39 ]. Another study of intraoperative local field potentials also provides evidence suggesting that GPi-DBS might improve dystonia symptoms by suppressing beta hypersynchrony in the GPi [ 37 ].…”

Section: Treatmentsmentioning

confidence: 99%

Clinicopathological Phenotype and Genetics of X-Linked Dystonia–Parkinsonism (XDP; DYT3; Lubag)

et al. 2017

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X-linked dystonia–parkinsonism (XDP; OMIM314250), also referred to as DYT3 dystonia or “Lubag” disease, was first described as an endemic disease in the Philippine island of Panay. XDP is an adult-onset movement disorder characterized by progressive and severe dystonia followed by overt parkinsonism in the later years of life. Among the primary monogenic dystonias, XDP has been identified as a transcriptional dysregulation syndrome with impaired expression of the TAF1 (TATA box-binding protein associated factor 1) gene, which is a critical component of the cellular transcription machinery. The major neuropathology of XDP is progressive neuronal loss in the neostriatum (i.e., the caudate nucleus and putamen). XDP may be used as a human disease model to elucidate the pathomechanisms by which striatal neurodegeneration leads to dystonia symptoms. In this article, we introduce recent advances in the understanding of the interplay between pathophysiology and genetics in XDP.

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“…Oscillatory activity in the beta band frequency range (13-30 Hz) has been detected in local field potential (LFP) recordings from various nodes within the dopamine-depleted cortico-basal gangliathalamo-cortical loop, and are widely believed to be pathological Engel and Fries, 2010;Weinberger et al, 2006). High-frequency stimulation at three nodes within this motor loop, the STN, internal globus pallidus, and substantia nigra pars reticulata, are each effective in treating motor symptoms, while concurrently suppressing local beta band rhythms (McConnell et al, 2012;Sutton et al, 2013;Whitmer et al, 2013;Wingeier et al, 2006). The establishment of a causal role for beta band rhythms in motor impairment will do much to strengthen this popular hypothesis of DBS action.…”

Section: Introductionmentioning

confidence: 99%

Neural Circuit Modulation During Deep Brain Stimulation at the Subthalamic Nucleus for Parkinson's Disease: What Have We Learned from Neuroimaging Studies?

Albaugh

Shih²

2013

Brain Connectivity

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Deep brain stimulation (DBS) targeting the subthalamic nucleus (STN) represents a powerful clinical tool for the alleviation of many motor symptoms that are associated with Parkinson's disease. Despite its extensive use, the underlying therapeutic mechanisms of STN-DBS remain poorly understood. In the present review, we integrate and discuss recent literature examining the network effects of STN-DBS for Parkinson's disease, placing emphasis on neuroimaging findings, including functional magnetic resonance imaging, positron emission tomography, and single-photon emission computed tomography. These techniques enable the noninvasive detection of brain regions that are modulated by DBS on a whole-brain scale, representing a key experimental strength given the diffuse and far-reaching effects of electrical field stimulation. By examining these data in the context of multiple hypotheses of DBS action, generally developed through clinical and physiological observations, we define a multitude of consistencies and inconsistencies in the developing literature of this rapidly moving field.

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Resting Beta Hypersynchrony in Secondary Dystonia and Its Suppression During Pallidal Deep Brain Stimulation in DYT3+ Lubag Dystonia

Cited by 17 publications

References 16 publications

Long-term increase in coherence between the basal ganglia and motor cortex after asphyxial cardiac arrest and resuscitation in developing rats

Long-term increase in coherence between the basal ganglia and motor cortex after asphyxial cardiac arrest and resuscitation in developing rats

Clinicopathological Phenotype and Genetics of X-Linked Dystonia–Parkinsonism (XDP; DYT3; Lubag)

Neural Circuit Modulation During Deep Brain Stimulation at the Subthalamic Nucleus for Parkinson's Disease: What Have We Learned from Neuroimaging Studies?

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