51-year-old African American male with history of alcohol use and recent diagnosis of seizures presented with language difficulties and increased seizure frequency. On exam patient was encephalopathic with impaired comprehension. MRI brain showed numerous rim-enhancing, T1 and T2 centrally hypointense lesions with surrounding vasogenic edema throughout the brain. He was started on broad spectrum antibiotics. Initial CSF showed 330 WBCs with 60% lymphocytes,protein 295 but normal serum leucocyte count. Further CSF studies for bacterial culture, fungal culture, AFB stain, HSV, VZV, VDRL and cryptococcus was negative. Serum blastomyces, coccidioides, and toxoplasma antibody was negative .Transthoracic echocardiogram and CT chest, abdomen and pelvis was unremarkable. During the next few days he developed non-obstructive hydrocephalus leading to extraventricular drain placement. Serum next generation sequencing test reported positive for Balamuthia Mandrillaris. Brain biopsy was consistent with Balamuthia mandrillaris. Based on previous case reports antimicrobial therapy was initiated. Despite all this, he continued to decline and family transitioned him to comfort care.