Recent human functional neuroimaging studies have demonstrated intrinsic functional connectivity among the anterior insula (AI), anterior cingulate cortex (ACC), amygdala, thalamus, hypothalamus, striatum, and brainstem. These regions have been collectively referred to as the ''salience network'' due to their consistent activation by diverse, homeostatically relevant stimuli that carry emotional weight [8]. Evidence supporting a role for the salience network in social-emotional function comes from patients with behavioral variant frontotemporal dementia (bvFTD), an early age-of-onset neurodegenerative disorder that produces dramatic changes in social behavior [6,7]. The right ventral anterior insula, a focus of degeneration in bvFTD, has been proposed as a key hub within this network [9], and functional imaging studies reveal consistent activation of this region during social-emotional processing [3]. The consequences of isolated, destructive anterior insula lesions, however, have not been reported. Here, we describe a 66-year-old woman who developed an abrupt change in social behavior after isolated right ventral anterior insular infarction.A 66-year-old right-handed woman presented with sudden onset of slurred speech. She had suffered from hypertension for 3 years and had a history of ischemic stroke involving the left parietal cortex, manifested by a subjective dysarthria one-and-a-half years earlier, from which she completely recovered during the follow-up period. She had been taking an antiplatelet agent and had no history of alcohol use or smoking. She had no previous history of depression or other psychiatric illness. Neurological examination revealed mild dysarthria and left central facial weakness. Diffusion-weighted magnetic resonance imaging (MRI) on the same day of admission revealed focal hyperintensity in the ventral anterior portion of the right insular cortex (Fig. 1a, b), with a corresponding hypointensity on the apparent diffusion coefficient map. T2-weighted imaging showed no abnormalities. Single photon emission computed tomography (SPECT) performed 2 days post-stroke showed hypoperfusion in the same region (Fig. 1c), but also in distributed right anterior brain structures (temporal pole, striatum, lateral frontal cortex) with known connectivity to the AI. Magnetic Electronic supplementary material The online version of this article (