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Abstract Background Hyperammonemia is a medical condition described as increased or elevated serum ammonia levels. High serum levels of ammonia can cause neurotoxicity. Sudden onset severe hyperammonemia may cause severe encephalopathy with brain damage. It can result in cerebral edema, emesis, seizures, hypotonia, and death. We report a young postpartum woman who had a sudden rise in serum ammonia levels after vaginal delivery. Case presentation A 24-year-old, married, postpartum Pakistani woman was admitted to the intensive care unit through the emergency department, with complaints of fever, severe abdominal pain with distension, and altered levels of consciousness. The patient had a medical history of spontaneous vaginal delivery 2 weeks before this hospital admission, after which she gradually developed the above symptoms. However, the patient’s past medical history was unremarkable with no hepatic disease, but her investigations revealed a progressive rise in serum ammonia levels. In the intensive care unit, she developed generalized tonic–clonic seizures. This was followed by a coma, tonsillar herniation, and death. Conclusion Postpartum hyperammonemia is a rare entity. It is a critical illness and must be evaluated for underlying metabolic disorders. Early diagnosis and treatment may result in better outcomes and reduced mortality among postpartum women with hyperammonemia.