In his serially published atlas of pathology, <i>Anatomie Pathologique du Corps Humain</i> (1829–1842), French anatomist and pathologist Jean Cruveilhier (1791–1874) provided an early clinical-pathologic description of Dyke-Davidoff-Masson syndrome. Cruveilhier’s case was initially published around 1830, more than a century before the clinical and radiologic report of Dyke and colleagues in 1933 based on a series of patients studied with pneumoencephalography. Although Dyke and colleagues were apparently unaware of Cruveilhier’s prior description, Cruveilhier’s case manifested all of the key osseous and neuropathological features of Dyke-Davidoff-Masson syndrome as later elaborated by Dyke and colleagues: (1) cerebral hemiatrophy with <i>ex vacuo</i> dilation of the lateral ventricle, (2) ipsilateral thickening of the diploe of the skull, and (3) ipsilateral hyper-pneumatization of the frontal sinuses. In addition, Cruveilhier noted crossed cerebral-cerebellar atrophy in his case and correctly inferred a “crossed effect” between the involved cerebral hemisphere and the contralateral cerebellum. Cruveilhier’s pathological case from 1830 clearly anticipated both the cases reported more than a century later by Dyke and colleagues based on pneumoencephalography and the more recent case reports recognized with computed tomography or magnetic resonance imaging.