Leptospirosis is an acute infectious disease that manifests as a biphasic sickness with an initial leptospiraemic and a subsequent immunological phase. It is caused by a spirochete belonging to the genus Leptospira. Leptospirosis seldom manifests clinically as a primary neurological impairment. We report a case of a middle -aged man who was initially diagnosed with leptospirosis and subsequently complicated by development of cerebral venous sinus thrombosis. This intriguing case study emphasises the importance of identifying uncommon leptospirosis symptoms in endemic regions such as Malaysia.
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