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β-Catenin Deletion in Regional Neural Progenitors Leads to Congenital Hydrocephalus in Mice / 神经科学通报·英文版
Neuroscience Bulletin ; (6): 81-94, 2022.
Article in English | WPRIM | ID: wpr-929082
ABSTRACT
Congenital hydrocephalus is a major neurological disorder with high rates of morbidity and mortality; however, the underlying cellular and molecular mechanisms remain largely unknown. Reproducible animal models mirroring both embryonic and postnatal hydrocephalus are also limited. Here, we describe a new mouse model of congenital hydrocephalus through knockout of β-catenin in Nkx2.1-expressing regional neural progenitors. Progressive ventriculomegaly and an enlarged brain were consistently observed in knockout mice from embryonic day 12.5 through to adulthood. Transcriptome profiling revealed severe dysfunctions in progenitor maintenance in the ventricular zone and therefore in cilium biogenesis after β-catenin knockout. Histological analyses also revealed an aberrant neuronal layout in both the ventral and dorsal telencephalon in hydrocephalic mice at both embryonic and postnatal stages. Thus, knockout of β-catenin in regional neural progenitors leads to congenital hydrocephalus and provides a reproducible animal model for studying pathological changes and developing therapeutic interventions for this devastating disease.
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Full text: Available Index: WPRIM (Western Pacific) Main subject: Mice, Knockout / Disease Models, Animal / Beta Catenin / Hydrocephalus / Neurons Limits: Animals Language: English Journal: Neuroscience Bulletin Year: 2022 Type: Article

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Full text: Available Index: WPRIM (Western Pacific) Main subject: Mice, Knockout / Disease Models, Animal / Beta Catenin / Hydrocephalus / Neurons Limits: Animals Language: English Journal: Neuroscience Bulletin Year: 2022 Type: Article