Brain imaging scanning has shown that atrophy can appear even before clinical onset of the disease,16 and a recent study on FTD patients using transcranial magnetic stimulation, showed circuit alterations in presymptomatic patients 15 years before the expected onset of the disease.17 For these reasons and to complement newly generated iPSC models of CHMP2B-FTD, in this work, we generated a human embryonic stem cell (hESC)-based co-culture model of excitatory neurons and astrocytes to investigate functional alterations at the cellular and network level in FTD caused by CHMP2B mutations. Here, CHMP2B is linked to Atrophy.