Professor Pshezhetsky obtained his Ph.D. in chemistry from Moscow State University. In 1989-1992 as a researcher at Moscow State University and Moscow Institute of Medical and Biological Chemistry, he studied genetic diseases of children caused by the inherited deficiencies of lysosomal enzymes. In 1993, he joined the Department of Pediatrics and Biochemistry, University of Montreal, Canada, where he was subsequently promoted to a tenured professor. Currently, he is a Director of the Elisa Linton Laboratory of lysosomal biology at CHU Ste-Justine Research Center, a scientific director of the Medical genetics diagnostic laboratory of CHU Ste-Justine, and an adjunct professor at the Department of Anatomy and Cell Biology at McGill University. Since November 2022, he has been an Elisa Linton Research Chair in Lysosomal Diseases.
Professor Pshezhetsky received many career awards, including a National Investigator Award, an Award of Excellence in Pediatric Research from the Foundation for the Research in Children's Disorders, and a Champion of Genetics award from the Genes for Cure Foundation. Current research interests include the molecular basis of lysosomal and other inherited metabolic disorders, sialoglycobiology, proteomics, and functional genomics of the cell.