ArticlePloS one2012
Tensor-based morphometry and stereology reveal brain pathology in the complexin1 knockout mouse.
Article in PloS one, 2012. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 15 papers.
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15 citing papers in PubMed, 24 citations in OpenAlex.
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- Transcriptome Analysis of the Chicken Follicular Theca Cells with miR-135a-5p Suppressed.G3 (Bethesda, Md.) · 2020Article
- Single-Cell RNA-Seq of Mouse Dopaminergic Neurons Informs Candidate Gene Selection for Sporadic Parkinson Disease.American journal of human genetics · 2018Article
- Variants in CPLX1 in two families with autosomal-recessive severe infantile myoclonic epilepsy and ID.European journal of human genetics : EJHG · 2017Article
- Therapeutic inhibition of soluble brain TNF promotes remyelination by increasing myelin phagocytosis by microglia.JCI insight · 2017Article
- In vivo microscopic voxel-based morphometry with a brain template to characterize strain-specific structures in the mouse brain.Scientific reports · 2017Article
- Acute Simian Varicella Virus Infection Causes Robust and Sustained Changes in Gene Expression in the Sensory Ganglia.Journal of virology · 2016Article
- MR fingerprinting with simultaneous B1 estimation.Magnetic resonance in medicine · 2016Article
- Regional Volume Decreases in the Brain of Pax6 Heterozygous Mutant Rats: MRI Deformation-Based Morphometry.PloS one · 2016Article
- Red nucleus and rubrospinal tract disorganization in the absence of Pou4f1.Frontiers in neuroanatomy · 2015Article
- Cerebellar transcriptional alterations with Purkinje cell dysfunction and loss in mice lacking PGC-1α.Frontiers in cellular neuroscience · 2014Article
- Automated determination of axonal orientation in the deep white matter of the human brain.Brain connectivity · 2012Article
- Huntington's disease mouse models online: high-resolution MRI images with stereotaxic templates for computational neuroanatomy.PloS one · 2012Article
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Authors and funding
5 authors at 2 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Complexins (Cplxs) are small, soluble, regulatory proteins that bind reversibly to the SNARE complex and modulate synaptic vesicle release. Cplx1 knockout mice (Cplx1(-/-)) have the earliest known onset of ataxia seen in a mouse model, although hitherto no histopathology has been described in these mice. Nevertheless, the profound neurological phenotype displayed by Cplx1(-/-) mutants suggests that significant functional abnormalities must be present in these animals. In this study, MRI was used to automatically detect regions where structural differences were not obvious when using a traditional histological approach. Tensor-based morphometry of Cplx1(-/-) mouse brains showed selective volume loss from the thalamus and cerebellum. Stereological analysis of Cplx1(-/-) and Cplx1(+/+) mice brain slices confirmed the volume loss in the thalamus as well as loss in some lobules of the cerebellum. Finally, stereology was used to show that there was loss of cerebellar granule cells in Cplx1(-/-) mice when compared to Cplx1(+/+) animals. Our study is the first to describe pathological changes in Cplx1(-/-) mouse brain. We suggest that the ataxia in Cplx1(-/-) mice is likely to be due to pathological changes in both cerebellum and thalamus. Reduced levels of Cplx proteins have been reported in brains of patients with neurodegenerative diseases. Therefore, understanding the effects of Cplx depletion in brains from Cplx1(-/-) mice may also shed light on the mechanisms underlying pathophysiology in disorders in which loss of Cplx1 occurs.
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