Rationale:Human infection withAngiostrongylus cantonensisis uncommon and has only been sporadically reported in the literature. Patients infected withA cantonensisusually have a delayed diagnosis and sometimes a poor prognosis.Patient concerns:A 70-year-old woman presented to the respiratory department with complaints of headache, chest pain, myalgia, fatigue, and anorexia for 7 days.Diagnoses:Complete blood count showed eosinophilia. The serum was tested showing a positive finding ofA cantonensisantibody. Cerebrospinal fluid was tested using high-throughput metagenomics analysis and 16 reads forA cantonensiswere mapped. The patient was diagnosed withA cantonensisinfection.Interventions:The patient received a 7-day course of albendazole and 4-day course of prednisone.Outcomes:When discharged from the hospital, the patient still suffered from fatigue and poor memory. Aminotransferase levels were high due to albendazole’s liver toxicity. In a post-discharge follow-up about 1 month later she had recovered completely both physically and mentally, and peripheral eosinophil count and aminotransferase levels were both normal.Lessons:Because the direct identification of parasites is difficult, high-throughput metagenomics analysis may provide a reliable alternative tool for the diagnoses of infection withA cantonensis. When albendazole is prescribed, caution must be taken with respect to its liver toxicity.