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Background: Leptospirosis is a zoonotic disease caused by spirochetes of the genus Leptospira. While the majority of leptospirosis cases occur in the tropics, some cases are also observed in temperate regions of developing countries. The disease has various clinical presentations ranging from a mild influenza-like form to a severe potentially fatal illness accompanied by multi-organ failure. However, atypical presentations of leptospirosis have occasionally been described. Here, a case of leptospirosis presenting as thrombotic thrombocytopenic purpura (TTP) is reported. Case Report: A 58-years-old male presented with fever, oliguria, darkening of urine, and visual hallucinations. Laboratory investigations revealed anaemia, severe thrombocytopaenia, elevated total bilirubin with indirect predominance, high lactate dehydrogenase, and increased urea (293 mg/dL) and creatinine (7.6 mg/dL) levels. He was diagnosed with TTP. Patient was thought leptospirosis due to atypical clinical manifestations. Leptospirosis was confirmed by strongly positive Microscopic Agglutination Test. Patient recovered completely with antibiotics and plasmapheresis. Conclusion: Leptospirosis may be accompanied by thrombotic thrombocytopenic purpura in particular subtropic regions. [ABSTRACT FROM AUTHOR]