20.500.12556/DiRROS-24300 An evolutionarily conserved role for CTNNB1/ β-CATENIN in regulating the development of the corpus callosum The corpus callosum (CC) is a major nerve bundle that connects the two hemispheres of the brain. Dysgenesis of the CC is associated with neurodevelopmental disorders such as the CTNNB1 syndrome. We identified that five individuals carrying CTNNB1 mutations displayed CC deficits. To explore CTNNB1/β-CATENIN-dependent mechanisms that regulate CC midline crossing, we examined mice with Ctnnb1 gain-of-function (GOF) or loss-of-function (LOF) selectively targeted to the early embryonic central nervous system midline using an Lmx1aCre driver. We identify that the Lmx1a lineage contributes to midline cell populations known to regulate CC pathfinding: the glial wedge, the indusium griseum glia, and a population of midline glutamatergic neurons. We find that each of these structures are affected in both GOF and LOF embryos, resulting in a profound disruption of CC crossing and formation of Probst bundles. Thus, regulated β-CATENIN function in midline cell populations is critical for CC development and its dysregulation may underlie the CC deficits associated with CTNNB1 syndrome. neurodevelopment molecular neuroscience CTNNB1 syndrome corpus callosum development true false true Angleški jezik Ni določen Neznano 2025-11-24 15:21:40 2025-11-24 15:21:40 2025-11-26 03:39:01 0000-00-00 00:00:00 2025 0 0 Nasl. z nasl. zaslona; Opis vira z dne 19. 8. 2025; str. 1-10, e1-e4 issue 9, [article no.] 113335 Vol. 28 2025 0000-00-00 Zaloznikova Objavljeno apc 0000-00-00 0000-00-00 0000-00-00 616.8 2589-0042 10.1016/j.isci.2025.113335 245893635 RAZ_Parichha_Arpan_2025.pdf RAZ_Parichha_Arpan_2025.pdf 1 E068430A045E406AE4E4D7392B472F80 f8dd119b44ca1be5fee89fc6707464f7c55929b90e8987ecfb0ca09d42460cb6 05c25ccc-c941-11f0-94a7-001a4af901a5 https://dirros.openscience.si/Dokument.php?lang=slv&id=36193 https://doi.org/10.1016/j.isci.2025.113335 0 1d0d1aaa-c941-11f0-94a7-001a4af901a5 https://dirros.openscience.si/Dokument.php?lang=slv&id=36194 Univerzitetni klinični center Ljubljana 0 0 0