Objective: The objective of this study is to investigate the occurrence and management of acute thrombotic microangiopathy, a severe complication associated with antiphospholipid syndrome (APS), particularly in the context of catastrophic APS (CAPS) during pregnancy. The study aims to enhance the understanding of CAPS in pregnancy, improve early diagnosis, and evaluate the effectiveness of different treatment modalities.Materials and methods: The study employed a case report methodology, focusing on a pregnant patient diagnosed with CAPS and experiencing acute thrombotic microangiopathy. The clinical presentation, laboratory findings, diagnostic procedures, and treatment interventions were documented and analysed. The treatment approach included the use of anticoagulants, glucocorticoids, and plasma exchange. The response to treatment, complications, and patient outcomes were assessed.Results: The study observed a rapid development of liver, central nervous system, and renal lesions within a week, indicating the severity of CAPS in the pregnant patient. Despite the pregnancy termination, the patient's clinical condition did not show improvement, suggesting a lack of response similar to observations in other cases. The patient received standard CAPS treatment, except for intravenous administration of immunoglobulins. Further details were reported regarding the treatment outcomes, laboratory parameters, and imaging findings.Conclusion: This study highlights the challenges associated with diagnosing and managing CAPS during pregnancy, specifically focusing on acute thrombotic microangiopathy. Early diagnosis, aggressive treatment with anticoagulants and glucocorticoids, and the use of plasma exchange or intravenous immunoglobulin are crucial in achieving positive outcomes. The study suggests the consideration of alternative interventions, such as immunosuppressive therapy or targeted biologic agents, in refractory cases of CAPS. Further research is needed to elucidate the optimal treatment strategies and long-term outcomes in pregnant patients with CAPS.