Data published by researchers at the Large Scale Biology laboratory show that the inactivation of a deubiquitinase (enzyme eliminating ubiquitin residues) is sufficient to cause the accumulation of ubiquinylated protein aggregates and open new avenues for understanding the origin of proteinopathies (Huntington disease, Alzheimer's ...). That works describe for the first time the involvement of deubiquitinase in the selective control of ubiquinylated autophagy substrates in Drosophila and also in models of human cells in culture.