Lack of TSK disrupts brain development by affecting the morphology of the anterior commissure, dentate gyrus, corpus callosum, and lateral ventricle (LV) (Ito et al., 2010; Hossain et al., 2013; Ahmad et al., 2020). Previously, we reported that TSK dysfunction was associated with hydrocephalus in mice and humans (Ito et al., 2021). Here, TSKU is linked to Hydrocephalus.