We report a patient diagnosed with Whipple's disease (WD) who presented only with neurological symptoms. Neuroimaging (MRI) showed lesions with marked mass effect similar to infiltrative tumors, which were hypersignal on long TR and hyposignal on short TR images, located in several areas of the brain. In serial controls, lesions resolved with gliosis and atrophic changes as well as migration of active infiltrative-like lesions to new areas. MR findings of the brain WD are discussed, which confirmed by stereotactic brain biopsy. Familiarity with the range of possible MR imaging appearances of WD enables the radiologist to place WD more effectively on the differential diagnosis which motivates the clinician to consider both the diagnosis and early initiation of treatment; so, this may significantly impact outcome. Moreover, repeated MR investigations may serve as a valuable method to evaluate efficacy of treatment and long term follow-up of WD involving the CNS.

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