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| Open AccessHuman iPSC-derived motoneurons harbouring TARDBP or C9ORF72 ALS mutations are dysfunctional despite maintaining viability
Amyotrophic lateral sclerosis (ALS) is a neurodegenerative disease that affects spinal cord motor neurons. Here the authors use induced pluripotent stem cell-derived motor neurons obtained from patients with ALS-linked mutations, and find functional deficits resulting from a progressive decrease in voltage-activated Na+ and K+currents that occur in the absence of cell death.
- Anna-Claire Devlin
- , Karen Burr
- & Gareth B. Miles
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Cerebellar plasticity and motor learning deficits in a copy-number variation mouse model of autism
Impairments of cerebellar-dependent motor control and learning are implicated in some forms of autism spectrum disorder (ASD). In this study, the authors provide a characterization of the motor deficits and cerebellar function abnormalities in a transgenic mouse model of ASD.
- Claire Piochon
- , Alexander D. Kloth
- & Christian Hansel
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| Open AccessVAV-1 acts in a single interneuron to inhibit motor circuit activity in Caenorhabditis elegans
VAV proteins are guanine nucleotide exchange factors that have various neuromodulatory roles in the nervous system. Here, Fry et al. show that in Caenorhabditis elegans, VAV-1 negatively regulates locomotor speed and only needs to be expressed in a specific interneuron to do so.
- Amanda L. Fry
- , Jocelyn T. Laboy
- & Kenneth R. Norman
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| Open AccessReal-time estimation and biofeedback of single-neuron firing rates using local field potentials
The use of local field potential (LFP) brain signals may allow development of more efficient and robust neural prosthetic devices. Here, Hall et al. develop a method for estimation and biofeedback control of single-neuron firing rates using signals extracted from multiple low-frequency LFPs.
- Thomas M. Hall
- , Kianoush Nazarpour
- & Andrew Jackson
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| Open AccessImmediate tool incorporation processes determine human motor planning with tools
Previous studies have shown that repeated tool use results in extension of body representation. Ganesh et al. now show that in addition to extension of body representation, tool use is also accompanied by an immediate shortening of perceived arm length on a trial-by-trial basis.
- G. Ganesh
- , T. Yoshioka
- & T. Ikegami
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| Open AccessBalanced activity in basal ganglia projection pathways is critical for contraversive movements
The striatum is required for evoking contraversive movements from each brain hemisphere, but it is unclear how. Here, Tecuapetla et al.use optogenetics to inhibit direct and indirect downstream striatal projection pathways, and show that activity in both pathways is necessary for contraversive movements.
- Fatuel Tecuapetla
- , Sara Matias
- & Rui M. Costa
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Activity-dependent facilitation of Synaptojanin and synaptic vesicle recycling by the Minibrain kinase
Dysregulation of the kinase Mnb is implicated in autism spectrum disorder and Down’s syndrome. Here the authors demonstrate that Mnb is required for correct synaptic morphology and synaptic vesicle endocytosis, which it achieves via phosphorylation of the synaptic endocytosis protein Synaptojanin.
- Chun-Kan Chen
- , Catherine Bregere
- & Karen T. Chang
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| Open AccessSnf2h-mediated chromatin organization and histone H1 dynamics govern cerebellar morphogenesis and neural maturation
The chromatin remodelling proteins Snf2h and Snf2l regulate nucleosome spacing. Here, the authors show that Snf2hablation impairs chromatin organization of neuronal lineages during mouse embryonic and post-natal cerebellar development.
- Matías Alvarez-Saavedra
- , Yves De Repentigny
- & David J. Picketts
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Morphological and functional remodelling of the neuromuscular junction by skeletal muscle PGC-1α
The peroxisome proliferator activated receptor γ co-activator 1α (PGC-1α) is implicated in regulating the morphology and function of the neuromuscular junction. Here, Arnold et al.show that PGC-1α promotes the remodeling of pre- and postsynaptic neuromuscular junction sites, even in the absence of physical activity.
- Anne-Sophie Arnold
- , Jonathan Gill
- & Christoph Handschin
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High-efficiency motor neuron differentiation from human pluripotent stem cells and the function of Islet-1
Motor neurons generated from human pluripotent stem cells are used for disease modelling and drug screening. Here the authors present a protocol for efficient generation of mature spinal motor neurons from human pluripotent stem cells by precisely timing the application of the neural patterning molecule retinoic acid.
- Qiuhao Qu
- , Dong Li
- & Fei Wang
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Temporal identity transition from Purkinje cell progenitors to GABAergic interneuron progenitors in the cerebellum
GABAergic neuron progenitors in the cerebellum give rise to Purkinje cells and GABAergic interneurons. Here the authors show that the transcription factors Olig2 and Gsx1 regulate an identity transition from Purkinje cell progenitors to interneuron progenitors during mouse development.
- Yusuke Seto
- , Tomoya Nakatani
- & Mikio Hoshino
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| Open AccessSpike timing-dependent selective strengthening of single climbing fibre inputs to Purkinje cells during cerebellar development
Cerebellar development involves activity-dependent strengthening of synaptic contacts between climbing fibres and Purkinje cells. Kawamura et al.show that temporally clustered multiple climbing fibre inputs contribute to characteristic burst spiking in immature Purkinje cells before specific contacts are strengthened.
- Yoshinobu Kawamura
- , Hisako Nakayama
- & Masanobu Kano
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| Open AccessRole of motor cortex NMDA receptors in learning-dependent synaptic plasticity of behaving mice
Motor cortex NMDA receptors have a key role in the acquisition of associative memories. Hasan et al. generate mice lacking NMDA receptor activity in the motor cortex and find that this impairs LTP, strengthening of synapses between somatosensory and motor cortices, and associative learning.
- Mazahir T. Hasan
- , Samuel Hernández-González
- & José M. Delgado-García
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Homeostatic regulation of dendritic dynamics in a motor map in vivo
Various homeostatic mechanisms have been implicated in regulating neuronal excitability. Kishore and Fetcho study homeostatic mechanisms in zebrafish and find they regulate topographic patterns of dendritic dynamics within pools of motoneurons, that map onto ordered patterns of recruitment during behaviour.
- Sandeep Kishore
- & Joseph R. Fetcho
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Behavioural and neurobiological implications of linear and non-linear features in larynx phonations of horseshoe bats
Echolocating bats produce a wide variety of calls at different frequencies. In this study, the physiological basis of these frequencies are investigated, revealing that small changes in laryngeal air flow result in jumps between frequencies used for either echolocation or communication.
- Kohta I. Kobayasi
- , Steffen R. Hage
- & Walter Metzner
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Opposing regulation of dopaminergic activity and exploratory motor behavior by forebrain and brainstem cholinergic circuits
Dopaminergic circuits are implicated in exploratory motor behaviour and are modulated by acetylcholine. Using transgenic mouse models, Patelet al. find that loss of forebrain acetylcholine results in exaggerated dopamine efflux and hyperactivity, whereas loss of brainstem acetylcholine leads to hypoactivity.
- Jyoti C. Patel
- , Elsa Rossignol
- & Robert P. Machold
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Structured neuronal encoding and decoding of human speech features
Speech is encoded by the firing patterns of speech-controlling neurons in different regions of the brain, which Tankus and colleagues analyse in this study. They find highly specific encoding of vowels in medial–frontal neurons and nonspecific tuning in superior temporal gyrus neurons.
- Ariel Tankus
- , Itzhak Fried
- & Shy Shoham
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Unravelling cerebellar pathways with high temporal precision targeting motor and extensive sensory and parietal networks
The cerebellum has important roles in motor control and sensory perception. Sultan and colleagues use electrical stimulation and functional magnetic resonance imaging to reveal that the cerebellum provides inputs to specialized cortical regions of the brain that modulate cerebro-cortical processing.
- Fahad Sultan
- , Mark Augath
- & Peter Thier
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Vocalization frequency and duration are coded in separate hindbrain nuclei
Vocal communication is relatively common among fish: the midshipman being an example with a particularly wide dynamic range. In this paper, the authors demonstrate that different populations of hindbrain neurons are responsible for the frequency and duration of these calls.
- Boris P. Chagnaud
- , Robert Baker
- & Andrew H. Bass
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| Open AccessRetinoid-independent motor neurogenesis from human embryonic stem cells reveals a medial columnar ground state
There is much interest in generating motor neurons from embryonic stem cells because they may be useful for the study of neurodegenerative disease. Pataniet al. show that in the absence of retinoic acid, neurons with features of medial motor columnar neurons can be generated from human embryonic stem cells.
- R. Patani
- , A. J. Hollins
- & S. Chandran
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| Open AccessAncestry of motor innervation to pectoral fin and forelimb
It was previously thought that the nerves in the pectoral fin of fish came solely from the spinal cord. Here, motoneurons in ray-finned fish are shown to also originate from the hindbrain, demonstrating that innervation was from both the hindbrain and the spinal cord in ancesteral vertebrates.
- Leung-Hang Ma
- , Edwin Gilland
- & Robert Baker