Progressive supranuclear palsy (PSP) is the most common frontotemporal lobar degeneration associated with tau pathology. The majority of patients with PSP can be accurately diagnosed antemortem due to a characteristic clinical syndrome; however, there are atypical clinicopathologic forms of PSP that can mimic Parkinson’s disease, frontotemporal dementia, progressive nonfluent aphasia and corticobasal syndrome. While rare pathogenic variants, common risk factors, and – more recently – rare risk-associated variants have been identified in PSP, a significant proportion of the heritability for neurodegenerative tauopathies and other frontotemporal lobar degenerations remains unexplained, strongly suggesting that additional genetic risk factors await discovery. We assembled 900 PSP samples for whole-genome sequencing (WGS) of neuropathologically characterized PSP. About half of the samples come from the Mayo Clinic in Jacksonville, and the others were compiled from investigators across the US and Europe. Macrogen performed HiSeq whole genome sequencing. The Genome Center for Alzheimers Disease (GCAD) process the files with their standardized pipeline.