Rationale:Posterior reversible encephalopathy syndrome (PRES), which diagnosis is based on clinical symptoms and radiological features, is a neurotoxic disease characterized by a set of clinical manifestations, such as seizure, headache, visual, and/or consciousness disturbance. It is the first case of PRES followed by postpartum hemorrhage (PPH) without underlying disease.Patient concerns:A 37-year-old healthy woman had PPH after caesarean section. Six days after delivery, headache occurred suddenly, followed by episodes of clonus seizure.Diagnoses:Brain computed tomography showed ischemic stroke. However, magnetic resonance imaging revealed characteristics consistent with PRES.Interventions:The patient received phenytoin for seizure control.Outcomes:Seizure was under good control over the following days. Three months later, repeated magnetic resonance imaging showed complete remission.Lessons:PRES may be triggered by PPH and is not necessarily secondary to typical predisposing factors such as hypertension or pre/eclampsia. Hormone fluctuation, increased blood pressure variation, and massive blood transfusion may be contributed to the development of PRES in our case. Also, it is necessary to rule out those life-threatening diseases, such as cavernoma hemorrhage, cerebral venous thrombosis, and ischemic stroke before the diagnosis of PRES.