Neuroschistosomiasis is an infection of the central nervous system caused by Schistosoma species and constitute a severe manifestation of the disease. Schistosoma japonicum is well-known cause of cerebral schistosomiasis and Schistosoma mansoni for spinal schistosomiasis. Although neuroschistosomiasis is not rare, reports on cerebral vasculitis associated with Schistosoma mansoni infection is scarce. With regards to schistosomiasis in Asia, most of the published literature has been on Schistosoma japonicum and the prevalence of Schistosoma mansoni in Asia is not well established. We report here a 54-yearold Rakhine woman with history of diabetes mellitus for more than ten years presenting with three recurrent stroke occurring over 2 months, twelve months after returning to Yangon from Rakhine, an endemic area for Schistosoma mansoni infection. Cerebral MRA revealed beaded appearance along left ACA and segmental narrowing beyond terminal ICA both sides, and contrast enhancement was noted at left frontal lobe with restricted diffusion on MRI brain. Serum serology testing for Schistosoma mansoni infection was positive. With oral praziquantel and high dose corticosteroid, aphasia and swallowing improved. This is the first report of delayed onset cerebral vasculitis associated with chronic Schistosoma mansoni infection in Asia.