Rationale:Parasitic eosinophilic meningitis is rarely observed in infants. The diagnosis of this disease is complicated by its atypical and severe clinical manifestations.Patient concerns:An infant presented to our hospital with high fever and irritability, as well as refusal to walk. Cerebrospinal fluid collected through lumbar puncture showed increased eosinophil count and third-stage Angiostrongylus cantonensis larvae.Diagnoses:Eosinophilic meningitis was suspected.Interventions:We started empiric treatment with levamisole (14 mg bid, 2.5 mg/kg·day) and prednisone (17.5 mg qd, 1.5 mg/kg·day).Outcomes:All of the infant's symptoms were resolved approximately 72 hours after treatment. The patient fully recovered from her illness after completing 4 weeks of levamisole and prednisolone treatment.Lessons:A. cantonensis is the most common cause of parasitic eosinophilic meningitis cases in Southeast Asia. Physicians treating infants who live in areas where A. cantonensis is endemic and who present with irritability, abnormal motor function, and elevated eosinophil count should be aware of the disease to provide timely and rational therapy to the patients.