Aim and background: Thrombotic microangiopathies (TMAs) are a range of conditions characterized by several key features, including microangiopathic hemolytic anemia (MAHA), thrombocytopenia, and the formation of microthrombi. This case report specifically examines one of the uncommon primary causes of TMA, namely atypical hemolytic uremic syndrome (aHUS). Case description: We present the scenario of a female in her late 20s, P1L1, who presented three days postdelivery with complaints of decreased urine output and fatigue. Her medical history comprised pregnancy-induced hypertension, primary postpartum hemorrhage (PPH), and two episodes of paroxysmal events accompanied by loss of consciousness, possibly indicative of tonic-clonic seizures. Blood investigations demonstrated features of intravascular hemolysis, thrombocytopenia, and significantly elevated levels of urea and creatinine. Further studies revealed low C3, normal C4, mildly reduced ADAMTS13, and slightly elevated ADAMTS inhibitor levels, which were insufficient to make a diagnosis of thrombotic thrombocytopenic purpura (TTP). Based on the above findings, the patient was diagnosed with aHUS and was started on repeated transfusions of packed red blood cells (PRBC) and fresh frozen plasma (FFP) along with intermediate hemodialysis and symptomatic measures. With an initial deterioration in condition requiring intubation on the second day, over the course of the week, she gradually improved and was discharged on day 21 of hospitalization. Conclusion: Early identification of aHUS is vital for cost-effective treatment. Advancements in complement studies can lead to quicker diagnosis, informed treatment decisions, and resource optimization. Clinical significance: This case report helps us derive, for example, one of the scenarios when an eye must be kept out for an uncommon diagnosis like aHUS.