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Patients diagnosed with thrombotic thrombocytopenic purpura (TTP) typically present with microangiopathic hemolytic anemia (MAHA) and thrombocytopenia; these two clinical manifestations were often believed to be essential indicators of TTP. However, such indicators are not always present in every case. Here, we present a patient affected by TTP but showing no distinctive schistocytes on blood smear review. TTP was diagnosed through a critically low level of a disintegrin and metalloproteinase with thrombospondin type 1 motif, member 13 (ADAMTS13) activity. Awareness of such an atypical presentation of TTP is essential for timely treatment to prevent serious and even fatal outcomes for patients.