Open science involves making scientific knowledge (in the form of papers, data, research tools, and other outputs) openly and practically available in order to accelerate discovery, innovation and clinical translation (Gold, 2016; Global Alliance for Genomics and Health, 2016; Fecher and Friesike, 2014). In a world first for an academic research institution, in 2016 the Montreal Neurological Institute and Hospital (the MNI) adopted a broad open science policy across all of its research activities. The MNI committed to make publicly available all positive and negative data by the date of first publication, to open its biobank to registered researchers and, perhaps most significantly, to withdraw its support of patenting on any direct research outputs (Figure 1; Poupon et al., 2017; Owens, 2016a; Rouleau, 2017). The span of this openness raises unique hopes and concerns for stakeholders, and also offers an important opportunity to examine and understand the potential benefits and costs of open science.