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Blastomycosis is a fungal infection that mostly involves the skin and lung with only a 5-10% incidence of Central nervous system involvement . It occurs worldwide but it is endemic to areas like the Midwestern United States, Manitoba and Ontario provinces of Canada and equally affects both immunocompetent and immunocomprimised individuals with the likelihood of being more aggressive in the latter . We present a 51 year old hypertensive factory machine operator from a Blastomyces non- endemic area of Cape Town, South Africa with no immunosuppressive condition who presented with worsening protracted headaches and cerebellar signs. Brain Computed Tomography and Magnetic Resonance imaging appearances of a heterogeneously enhancing left cerebellar hemisphere lesion posed an initial diagnostic dilemma which led to worsening of the patient’s clinical condition from hydrocephalus as a result of increased perilesional oedema and compression of the fourth ventricle. Eventual biopsy and culture after craniectomy yielded a diagnosis of primary cerebellar blastomycosis which necessitated a 6 week intravenous treatment with amphotericin B followed by oral itraconazole for 12 months leading to complete radiological resolution of the cerebellar lesion and clinical resolution of the headaches and ataxia.