ABSTRACT Bothrops erythromelas are serpents that belong to the Viperidae family, which are the main species responsible for human snakebites in Ceara State, Northeast Brazil. Thrombotic microangiopathy (TMA) is an uncommon group of disorders characterized by microangiopathic hemolytic anemia (MAHA), thrombocytopenia and acute kidney injury (AKI), and occurrence after snakebites have been rarely reported. In this report, we described the case of a 57 year-old-man without comorbidities who was bitten by a Bothrops erythromelas on his right ankle. He presented with pain, edema and local bleeding. Symptomatology and laboratory tests were compatible with the diagnosis of TMA. He received specific antivenom and fluids replacement without any anaphylactic reaction. The conservative treatment was effective and there was no need for red blood cells transfusion or plasmapheresis. The aim of this report was to describe the first case of thrombotic microangiopathy following Bothrops erythromelas envenoming in the Northeast Brazil, providing insights about important mechanistic pathways of Bothrops snakebite-associated TMA and how to change the prognosis of the disease.