Gray matter structural alterations of cortico-striato-thalamo-cortical loop in familial Paroxysmal Kinesigenic Dyskinesia

Wang, Dongcui; Jin, Hong; Xie, Fangfang; Wang, Ziyun; Xing, Wu

doi:10.1016/j.heliyon.2024.e36739

Heliyon

2024

DOI: 10.1016/j.heliyon.2024.e36739

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Gray matter structural alterations of cortico-striato-thalamo-cortical loop in familial Paroxysmal Kinesigenic Dyskinesia

Dongcui Wang,

Hong Jin,

Fangfang Xie

et al.

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2024

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Assembloid model to study loop circuits of the human nervous system

Miura,

Kim,

Jurjuț

et al. 2024

Preprint

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Neural circuits connecting the cerebral cortex, the basal ganglia and the thalamus are fundamental networks for sensorimotor processing and their dysfunction has been consistently implicated in neuropsychiatric disorders. These recursive, loop circuits have been investigated in animal models and by clinical neuroimaging, however, direct functional access to developing human neurons forming these networks has been limited. Here, we use human pluripotent stem cells to reconstruct an in vitro cortico-striatal-thalamic-cortical circuit by creating a four-part loop assembloid. More specifically, we generate regionalized neural organoids that resemble the key elements of the cortico-striatal-thalamic-cortical circuit, and functionally integrate them into loop assembloids using custom 3D-printed biocompatible wells. Volumetric and mesoscale calcium imaging, as well as extracellular recordings from individual parts of these assembloids reveal the emergence of synchronized patterns of neuronal activity. In addition, a multi-step rabies retrograde tracing approach demonstrate the formation of neuronal connectivity across the network in loop assembloids. Lastly, we apply this system to study heterozygous loss of ASH1L gene associated with autism spectrum disorder and Tourette syndrome and discover aberrant synchronized activity in disease model assembloids. Taken together, this human multi-cellular platform will facilitate functional investigations of the cortico-striatal-thalamic-cortical circuit in the context of early human development and in disease conditions.

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Assembloid model to study loop circuits of the human nervous system

Miura,

Kim,

Jurjuț

et al. 2024

Preprint

View full text Add to dashboard Cite

show abstract

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Gray matter structural alterations of cortico-striato-thalamo-cortical loop in familial Paroxysmal Kinesigenic Dyskinesia

Cited by 1 publication

References 30 publications

Assembloid model to study loop circuits of the human nervous system

Assembloid model to study loop circuits of the human nervous system

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