which has been shown to regulate clathrin-mediated endocytosis, leads to the enlargement of early endosomes (Cossec et al., 2012). Transgenic mice harboring 4–5 copies of the human SOD1 gene showed an increase in GABAergic inhibition and diminished LTP in the CA1 region (Gahtan et al., 1998; Levkovitz et al., 1999). Increased Dyrk1A dose influenced the expression of proteins involved in GABAergic and glutamatergic neurotransmission (Souchet et al., 2014) as well as other defects characteristic of DS (Toiber et al., 2010). Genetic normalization of Dyrk1A by stereotaxic injection of adeno-associated virus containing a short hairpin RNA against Dyrk1A improved LTP in the CA1 region (Altafaj et al., 2013). Recently, our studies to genetically dissect the mouse DSCR examined behavioral and synaptic phenotypes in the Dp(16)1/+ model of DS, as well as in mice with smaller segmental trisomies based on this model (Jiang et al., 2015). A clear role for increased Dyrk1A was demonstrated. In the current study, we used a direct genetic approach to assess the role of Kcnj6, a gene adjacent to Dyrk1A.
