Strongyloidiasis is an infestation caused by the intestinal nematode Strongyloides stercoralis. It is potentially fatal in immunocompromised hosts due to its capacity to cause an overwhelming hyperinfestation however infested healthy individuals are usually asymptomatic. Hyperinfestation is extremely rare in the Central Nervous System (CNS) and usually limited to the gastrointestinal tract or lungs. CNS involvement in strongyloidiasis has only been seen in patients with hyperinfestation syndrome and may be fatal when misdiagnosed. In this report, we describe an unusual case of a 13-year-old girl presented with epileptic fits, multiple brain lesions and diagnosed as Strongyloides stercoralis infestation. Suitable frontal lesion biopsied and diagnosed as Strongyloides infestation and vasculitis. MRI features were totally regressed after the treatment with oral Albendazole for 3 months.