An inducible system to study the regulatory functions of GSX2 in human lateral ganglionic eminence-like progenitors.

ABSTRACT: Animal models have demonstrated a critical role of the homeodomain transcription factor Genetic-Screened Homeobox 2 (‍‍‍‍‍‍‍‍‌‌‌‌‌‌Gsx‍‍2‍‍‍‍‍‍) in the developing basal ganglia. Moreover, recent clinical genetic studies have shown that GSX2 patient variants are associated with severe neurological symptoms and basal ganglia dysgenesis. Unfortunately, technical limitations with existing animal models, such as progenitor heterogeneity and limited temporal control, have impeded the investigation of direct regulatory targets. In this study, we engineered a Dox-inducible human embryonic stem cell (‍‍hESC) line to investigate the function of GSX2 in directed differentiation cultures that model developing lateral ganglionic eminence-like (LGE-like) progenitors. Transcriptomic, chromatin accessibil

SUBMITTER: Farrow E 

PROVIDER: S-EPMC12866604 | biostudies-literature | 2026 Apr

REPOSITORIES: biostudies-literature