Like BRWD1, DYRK1A overexpression can impair cognition in mice48, and restoring Dyrk1a to euploid copy number (or pharmacologically inhibiting it) can rescue DS-related cognitive impairments49,50, skeletal abnormalities51,52 and Alzheimer’s disease related phenotypes in trisomic mice53. Here, BRWD1 is linked to early-onset autosomal dominant Alzheimer disease.