The McGill Genome Centre provides Canadian and international researchers with high-throughput technologies and cutting-edge approaches to enable next-generation genomic studies. Our Centre addresses key challenges in harnessing the power of next-generation sequencing (NGS) into innovation in biomedicine and the life sciences by: 1) Empowering study designs, sequencing and analytical solutions for nucleotide variation, 2) Providing comprehensive tools to assess functional differences in human tissues and cells, 3) Furnishing scalable techniques and robust interrogation of functional genomic variation in populations. We work closely with the other two major national genome centres as a founding member of the CGEn consortium to coordinate these efforts across Canada. Initiatives led by our scientists have enabled us to secure state-of-the-art technologies and integrate innovative methodologies, setting the stage for contemporary and future genomics operations with scope and impact beyond simply producing raw data, and thereby allowing Quebec and Canadian scientists leadership in genome-based science.