BACKGROUND The aim of this study was to compare the morphological changes in cerebral and cerebellar gray matter in patients with essential tremor under 60-years-of-age, with age-matched and gender-matched normal healthy volunteer control subjects, using functional magnetic resonance imaging (fMRI) and voxel-based morphometry (VBM) analysis. MATERIAL AND METHODS A retrospective, controlled, comparative clinical study included 17 patients with essential tremor, <60 years-of-age, and 17 age-matched and gender-matched healthy volunteer control subjects, recruited between June 2010-July 2012. MRI and VBM analysis were used to compare cerebral and cerebellar gray matter density between groups. The Washington Heights-Inwood Genetic Study of Essential Tremor (WHIGET) rating scale was used to assess tremor severity in the patient group. Clinical and demographic characteristics were recorded for all study participants. RESULTS MRI and VBM analysis showed significant bilateral expansion of the cerebellum, occipital fusiform cortices, right inferior temporal gyrus, and precentral lobes in patients with essential tremor (P<0.005); reduction in gray matter was found in the left parietal lobe. The region of interest (ROI) analysis showed volume enlargement in the thalamus, midbrain, and the precuneus (P<0.005). No significant correlation between changes in gray matter and changes in clinical variables, including age, gender, tremor duration, the activity of daily living (ADL) scale, the mini-mental state examination (MMSE) scale, family history, and tremor severity were found. CONCLUSIONS Predominantly cerebellar gray matter expansion in patients less than 60 years-of-age with essential tremor might be the result of compensation for the decline in cerebellar function.