OBJECTIVE To identify blinking-induced functional magnetic resonance imaging (fMRI) activation patterns in five benign essential blepharospasm (BEB) patients and five age-matched control subjects. METHODS fMRI brain activation maps were obtained during repeated conditions of spontaneous and voluntary blinking in BEB and control groups. Blood oxygen level-dependent intensity images were collected from two separate runs as 16 axial and 16 coronal, 8 mm thick slices using a T2-star weighted gradient echo EPI sequence, coregistered with anatomic images. Spatially normalized and isotropically blurred activation maps for each subject were combined within groups of BEB patients and control subjects to generate maps of the intersubject mean fractional signal change.RESULTS Substantially greater activation during spontaneous and voluntary blinking was seen in BEB patients compared with control subjects in the anterior visual cortex, anterior cingulate cortex, primary motor cortex, central region of the thalamus, and superior cerebellum. In both groups, activations were generally greater for voluntary than for spontaneous blinking. CONCLUSIONS The activations observed might represent a hyperactive cortical circuit linking visual cortex, limbic system, supplementary motor cortex, cerebellum, and supranuclear motor pathways innervating the periorbital muscles.