Jacques Jean Lhermitte (1877–1959) was among the most accomplished neurologists of the 20th century. In addition to working as a clinician and instructor, he authored more than 800 papers and 16 books on neurology, neuropathology, psychiatry, and mystical phenomena. In addition to the well-known “Lhermitte’s sign,” an electrical shock–like sensation caused by spinal cord irritation in demyelinating disease, Lhermitte was a pioneer in the study of the relationship between the physical substance of the brain and the experience of the mind. A fascinating example of this is the syndrome of peduncular hallucinosis, characterized by vivid visual hallucinations occurring in fully lucid patients. This syndrome, which was initially described as the result of a midbrain insult, also may occur with injury to the thalamus or pons. It has been reported as a presenting symptom of various tumors and as a complication of neurosurgical procedures. Here, the authors review the life of Lhermitte and provide a historical review of the syndrome of peduncular hallucinosis.