The human silencing hub (HUSH) complex may be involved in complex disorders affecting the brain and neurons, but to date its mechanism of action hasn’t been understood. A study by researchers at the Institute of Molecular Biotechnology of the Austrian Academy of Sciences (IMBA) has now uncovered the in vivo targets and physiological functions of a component of the HUSH gene silencing complex and one of its associated proteins. The work, led by Astrid Hagelkruys, PhD, senior research associate in the Penninger group at IMBA, and conducted in laboratory mouse models and human brain organoids, links the HUSH complex to normal brain development, neuronal individuality, and connectivity, as well as mouse behavior.