A 22-years old male patient was admitted to a tertiary care hospital with complaints of an alleged history of intentional poisoning (organophosphorus compound and nitrofurantoin) and developed hemolytic anemia after receiving methylene blue for 8 days. The patient presented with hematuria and hemoglobin level 3.1 which confirmed hemolytic anemia G6PD level was normal. Methylene blue was discontinued and PRBC transfusion (3 pints) was given. After 4 days of blood transfusion, the patient’s Hb level became 9.4 g/dl. Causality assessment was suggestive of a probable relationship between the drug and reaction.