Introduction: Thrombotic Thrombocytopenic Purpura (TTP) is a rare and potentially life-threatening condition, particularly in the postpartum period. While TTP affects approximately 3.7 per million individuals, postpartum TTP is even less common. This disorder is characterized by microvascular thrombi formation, resulting in endothelial damage and organ dysfunction, including the kidneys, although significant acute kidney injury (AKI) is more characteristic of Hemolytic Uremic Syndrome (HUS). Case presentation: A 31-year-old, P1L2 woman presented postpartum with hypertension, shortness of breath, and seizures. Initial investigations revealed anemia, thrombocytopenia, and renal dysfunction. The patient underwent emergency cesarean section in outside hospital and was referred here for further management. Clinical findings and investigations: Initial investigations revealed anemia, thrombocytopenia, and renal dysfunction. She subsequently developed TTP, confirmed by clinical and laboratory findings, including a negative ADAMTS13. Posterior Reversible Encephalopathy Syndrome (PRES) was also diagnosed. Schistocytosis was observed on peripheral smear. Interventions and outcome: Her treatment consisted of plasma exchange, hemodialysis, and corticosteroids, Anti epileptics, Anti hypertensives and diuretics leading to a complete recovery. Conclusion: Early diagnosis and treatment with Plasma Exchange could be the vital factors in reducing maternal mortality due to postpartum HUS. This case highlights the challenges in diagnosing postpartum TTP, especially with atypical presentations and negative ADAMTS13, and the importance of prompt diagnosis and intervention.