Japanese encephalitis (JE) is a common infection caused by the Japanese encephalitis virus in Southeast Asia, which is transmitted to humans through Culex mosquitoes. Magnetic resonance imaging (MRI) is used to diagnose JE, which is often characterized by the presence of bilateral symmetric thalamic involvement. Here, we report a rare case of JE characterized by leptomeningeal enhancement without thalamic involvement. This leptomeningeal enhancement disappeared with the treatment; however, new non-specific multifocal and bilateral high signal intensities in the cerebral white matter were found on follow-up MRI.
