Microdysgenesis (MD) is a term used to refer to subtle brain dysplasia based on structural tissue characteristics seen exclusively under the microscope. Although MD is often referred to within the field of modern epileptology, the term and its implications have actually evolved over a long period in neurology, starting in the late 19th century. This article undertakes a careful evaluation of original publications in the epilepsy literature and demonstrates that the concept of MD is anchored within a set of papers written from 1890 to 1930 and their contemporaneous reception in classic neuropsychiatric handbooks. Both the text of these early publications and the development of the MD concept are examined and illustrated. This perspective provides insight into historical scientific views of epilepsy as a mental disease that was thought to be dependent largely on hereditary or developmental factors.