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Uncovers how OTUD5 mutations affect neural progenitor cell stability, suggesting potential therapies for neurodevelopmental disorders.
This study establishes a novel disease mechanism whereby OTUD5 mutations disrupt NPC homeostasis through GSK3β destabilization, highlighting the critical role of ubiquitination regulation in neurodevelopment. Our iPSC model provides a platform for testing GSK3β-targeted therapies in OTUD5-related NDDs.
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Xu et al. (2026) studied this question.
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