We describe the evolution of neuropathology in Canada, beginning with William Osler who began working in Montréal in 1874 and finishing with the major period of expansion in the 1970s. Organized services began in the 1930s, in Montréal with the neurosurgeons Wilder Penfield and William Cone, and in Toronto with Eric Linell and Mary Tom, who both began their careers as neuroanatomists. Jerzy Olszewski and Gordon Mathieson, who trained in Montréal and Toronto, drove the creation of the CanadianAssociation of Neuropathologists in 1960. Training guided by the Royal College of Physicians and Surgeons of Canada was formalized in 1965, with the first certifying examination in 1968 and the subsequent creation of formal structured training programs. The number of neuropathologists in Canada increased rapidly through the 1960s and 1970s, with individuals coming from both clinical neuroscience and anatomic pathology backgrounds, a pattern that persists to the present day.