| First Author | Ma L | Year | 2022 |
| Journal | Neurosci Bull | Volume | 38 |
| Issue | 1 | Pages | 81-94 |
| PubMed ID | 34460072 | Mgi Jnum | J:337794 |
| Mgi Id | MGI:7506771 | Doi | 10.1007/s12264-021-00763-z |
| Citation | Ma L, et al. (2022) beta-Catenin Deletion in Regional Neural Progenitors Leads to Congenital Hydrocephalus in Mice. Neurosci Bull 38(1):81-94 |
| abstractText | Congenital hydrocephalus is a major neurolog-ical disorder with high rates of morbidity and mortality; however, the underlying cellular and molecular mecha-nisms remain largely unknown. Reproducible animal models mirroring both embryonic and postnatal hydro-cephalus are also limited. Here, we describe a new mouse model of congenital hydrocephalus through knockout of b-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 proï¬ling revealed severe dysfunctions in progenitor maintenance in the ventricular zone and therefore in cilium biogenesis after b-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 b-catenin in regional neural progenitors leads to congenital hydro-cephalus and provides a reproducible animal model for studying pathological changes and developing therapeutic interventions for this devastating disease. |
