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Lymphomatoid granulomatosis (LYG) is a rare B-cell lymphoproliferative disorder, strongly associated with Epstein-Barr virus (EBV) infection.It commonly affects patients in their 4th-6th decades of life.Lung is the most common site involved.Although CNS involvement is present in ~25% of cases, it is rarely the primary or exclusive site.We report two cases of LYG with isolated CNS involvement.Case 1 was a 62-year-old woman presented with headaches and diplopia.MRI showed 3-4 small areas of punctate enhancement in the left anterior temporal lobe as well as T2 signal and FLAIR abnormalities in the surrounding temporal lobe, midbrain and right brachium pontis.Over the next 2 months she became withdrawn, less communicative and blunted in affect.Repeat MRI showed progression of the lesions.Case 2 was a 55-year-old woman presented with symptoms and signs associated with a brain mass.Biopsies of the two cases revealed geographic necrosis surrounded by a cellular infiltrate with scattered, markedly atypical lymphoid cells in an angiocentric pattern which were strongly positive for the B-cell marker CD20 and EBV.