Complexin I knockout rats exhibit a complex neurobehavioral phenotype including profound ataxia and marked deficits in lifespan

Xu, Yang; Zhao, Xiaoming; Liu, Jia; Wang, Yangyang; Xiong, Liu‐Lin; He, Xiuying; Wang, Ting‐Hua

doi:10.1007/s00424-019-02337-5

Cited by 11 publications

(5 citation statements)

References 53 publications

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“…Complexin‐1 KO mice and rats die 2–4 months after birth, are profoundly ataxic, exhibit dystonia and resting tremor, have motor impairments, and, in line with epilepsy observed in patients, some mice have sporadic seizures (Glynn et al., 2005; Reim et al., 2001; Xu et al., 2020). However, contrary to the clinical phenotype, complexin‐1 KO rodents have no evidence of atrophy or severe cognitive deficits, with only mild changes in sociability, impulsivity, exploratory behaviour and agitation (Drew et al., 2007; Glynn et al., 2005; Reim et al., 2001; Xu et al., 2020). This suggests that there may be species differences in the importance of complexin‐1 for cognition.…”

Section: Complexin‐1mentioning

confidence: 92%

“…Despite loss of complexin-1 having no overt effects on neurotransmission (likely resultant from compensation by complexin-2), comparatively more severe effects are seen at the systems level in rodent models lacking complexin-1. Complexin-1 KO mice and rats die 2-4 months after birth, are profoundly ataxic, exhibit dystonia and resting tremor, have motor impairments, and, in line with epilepsy observed in patients, some mice have sporadic seizures (Glynn et al, 2005;Reim et al, 2001;Xu et al, 2020). However, contrary to the clinical phenotype, complexin-1 KO rodents have no evidence of atrophy or severe cognitive deficits, with only mild changes in sociability, impulsivity, exploratory behaviour and agitation (Drew et al, 2007;Glynn et al, 2005;Reim et al, 2001;Xu et al, 2020).…”

Section: Cplx1-early Infantile Epileptic Encephalopathymentioning

confidence: 94%

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Disorders of synaptic vesicle fusion machinery

Melland

Carr

Gordon

2020

Journal of Neurochemistry

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Neuronal communication relies on the tightly controlled fusion of synaptic vesicles at nerve terminals, which results in the release of neurotransmitters with strict temporal and quantal precision. At rest, synaptic vesicle fusion is inhibited (Brunger et al., 2018). Action potential-mediated opening of voltage-gated Ca 2+ channels results in Ca 2+ influx into the nerve terminal, creating a Ca 2+ nanodomain that triggers the fusion of vesicles that are docked and primed at the plasma membrane, thereby evoking fast synchronous neurotransmitter release (Chanaday & Kavalali, 2018;Sudhof, 2013).Vesicles can additionally fuse asynchronously following evoked synchronous release and some spontaneous fusion of vesicles also occurs, though this is clamped (Chanaday & Kavalali, 2018).These processes are under exquisite regulatory control, preventing excessive neurotransmitter release and ensuring high-fidelity neuronal communication (Brunger et al., 2019;Rizo, 2018;; perturbation of this regulation can lead to a breakdown in neurotransmission.A range of presynaptic proteins are required to orchestrate these precision events. The SNARE (soluble N-ethylmaleimide-sensitive factor attachment protein receptor) complex, composed of the vesicular SNARE protein (v-SNARE) synaptobrevin and the target membrane SNARE proteins (t-SNAREs) syntaxin-1 and SNAP-25 (synaptosomal-associated protein-25), is the core fusion machinery that provides the energy required for fusion of synaptic vesicles with the plasma membrane (Weber et al., 1998). Several other key components regulate synaptic vesicle exocytosis in physiological environments (Brunger et al., 2019;Sudhof, 2014). SNARE complex assembly is orchestrated by Munc13 and Munc18, and further

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Section: Complexin‐1mentioning

confidence: 92%

Section: Cplx1-early Infantile Epileptic Encephalopathymentioning

confidence: 94%

Disorders of synaptic vesicle fusion machinery

Melland

Carr

Gordon

2020

Journal of Neurochemistry

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“…The deeper the degree of spinal cord injury, the less CPLX1 expression. Previous studies have found that CPLX1-/-rats exhibit severe ataxia, dystonia, movement and exploratory defects, and increase anxiety and sensory deficits, but normal cognitive function [17]. ReimK et al knocked out CPLX 1/2 and found that the mice died a few hours after birth.…”

Section: Discussionmentioning

confidence: 99%

Expression of CPLX1 in Different Degrees of Spinal Cord Injury in Subacute Stage

Xi¹,

Ji²,

Bao³

et al. 2020

IJN

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This study is to explore the expression of CPLX1 (Complexin 1) in spinal cord tissues with different degrees of injury in the subacute phase of spinal cord injury. Twenty-four SD rats were randomly divided into three experimental groups (light, medium and severe injury) and a sham-operated control group. The experimental group made acute spinal cord injury models according to the lisa method, and the spinal cord tissue were collected 72 hours after injury. The tissues of rats were taken for transcriptome high-throughput sequencing and proteomic analysis experiments to observe the expression of CPLX1 in the different degrees during the subacute phase of spinal cord injury. The mRNA and protein of CPLX1 were detect in rat spinal cord tissue, and they were expressed high in the sham operation group. The protein expression in the light injury group began to decrease, and with the increase of the degree of injury, the transcription level of CPLX1 and the amount of protein expression also gradually decreased. The change trend of mRNA transcription level and protein expression level was basically the same. When the spinal cord injury heavier, the less CPLX1 was expressed, the two showed a significant negative correlation. In the subacute stage of spinal cord injury, the translation and protein levels of CPLX1 decreased significantly. The more severe the injury, the lower the transcription level and protein expression level. CPLX1 may be used as a marker gene for the severity of spinal injury.

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“…For seqFISH+，the single-cell ST data of the mouse somatosensory cortex dataset was retrieved from the “Github repository [ https://rubd.github.io/Giotto_site/articles/mouse_seqFISH_cortex_200914 ]” [ 25 ].For Seq-Scope, the single-cell ST data of the human colon dataset was downloaded from “Deep Blue Data[Data Set | Seq-Scope processed datasets for liver and colon results (RDS) and H&E images | ID: 9c67wn05f | Deep Blue Data (umich.edu)]” [ 51 ]. Source codes for the CPPLS_MLP R, python packages and the related scripts are available at “CPPLS_MLP Github [ https://github.com/wuzhenao/CPPLS-MLP ]”.…”

Section: Data Availabilitymentioning

confidence: 99%

CPPLS-MLP: a method for constructing cell–cell communication networks and identifying related highly variable genes based on single-cell sequencing and spatial transcriptomics data

Zhang,

Wu,

et al. 2024

Briefings in Bioinformatics

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In the growth and development of multicellular organisms, the immune processes of the immune system and the maintenance of the organism’s internal environment, cell communication plays a crucial role. It exerts a significant influence on regulating internal cellular states such as gene expression and cell functionality. Currently, the mainstream methods for studying intercellular communication are focused on exploring the ligand–receptor–transcription factor and ligand–receptor–subunit scales. However, there is relatively limited research on the association between intercellular communication and highly variable genes (HVGs). As some HVGs are closely related to cell communication, accurately identifying these HVGs can enhance the accuracy of constructing cell communication networks. The rapid development of single-cell sequencing (scRNA-seq) and spatial transcriptomics technologies provides a data foundation for exploring the relationship between intercellular communication and HVGs. Therefore, we propose CPPLS-MLP, which can identify HVGs closely related to intercellular communication and further analyze the impact of Multiple Input Multiple Output cellular communication on the differential expression of these HVGs. By comparing with the commonly used method CCPLS for constructing intercellular communication networks, we validated the superior performance of our method in identifying cell-type-specific HVGs and effectively analyzing the influence of neighboring cell types on HVG expression regulation. Source codes for the CPPLS_MLP R, python packages and the related scripts are available at ‘CPPLS_MLP Github [https://github.com/wuzhenao/CPPLS-MLP]’.

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Complexin I knockout rats exhibit a complex neurobehavioral phenotype including profound ataxia and marked deficits in lifespan

Cited by 11 publications

References 53 publications

Disorders of synaptic vesicle fusion machinery

Disorders of synaptic vesicle fusion machinery

Expression of CPLX1 in Different Degrees of Spinal Cord Injury in Subacute Stage

CPPLS-MLP: a method for constructing cell–cell communication networks and identifying related highly variable genes based on single-cell sequencing and spatial transcriptomics data

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