Abstract
INTRODUCTION: Thrombotic thrombocytopenic purpura (TTP) is a rare, life-threatening thrombotic microangiopathy and a high mortality rate if untreated. While TTP can be primary or secondary to factors like drugs, reports linking it to anti-tuberculosis (anti-TB) therapy are scarce. This case highlights the diagnostic challenges and need for vigilance in TB patients receiving standard regimens. CASE PRESENTATION: A 76-year-old male on anti-TB therapy (isoniazid, rifampicin, pyrazinamide, ethambutol) presented with acute neurological symptoms, thrombocytopenia (platelets 9×10^9/L), microangiopathic hemolysis (schistocytes), and severely reduced ADAMTS13 activity (<5%). Imaging revealed multiple cerebral infarctions. Despite plasma exchange and steroids, the patient deteriorated and died after family-requested care withdrawal. CONCLUSION: This case highlights TTP as a rare but serious complication of anti-TB therapy. Clinical vigilance is essential, including platelet monitoring during initial treatment and a low threshold for ADAMTS13 testing in cases of unexplained thrombocytopenia. Future multicenter studies are needed to investigate immune mechanisms and assess therapies such as rituximab, with the aim of optimizing management strategies for rare adverse drug events and improving patient outcomes.