Toxocariasis is a zoonosis disease with high sero-prevalence in Southeast Asian. Neurotoxocariasis has never been reported in Taiwan. Herein, we presented 2 cases of neurotoxocariasis. The first case is a 48-year-old man with febrile headache, rapid progressive cognitive problems and later thoracic myelitis. Meningeal enhancements on the corresponding sites were found on magnetic resonance imaging (MRI). Eosinophilic pleocytosis was present in peripheral blood and cerebrospinal fluid (CSF). A positive Toxocara canis larval excretory-secretory antigen (TcES)-based immunoblotting test for CSF confirmed the diagnosis. The second case is a 42-year-old woman of progressive headache with features of increased intracranial pressure. CSF analysis showed lymphocytic pleocytosis initially and eosinophilic pleocytosis later. Her brain MRI was normal. The diagnosis was confirmed by the presence of anti-TcES IgG in CSF. The two cases were soonly cured by mebendazole. Neurotoxocariasis presented a broad spectrum of neurological symptoms and the CSF profile can be non-eosinophilic pleocytosis. The prevalence of neurotoxocariasis may be seriously underestimated due to low awareness of physicians and lack of standard conventional diagnostic test in Taiwan. The two cases of neurotoxocariasis firstly reported in Taiwan will raise physician's awareness in recognizing this curable disease particularly in managing patients with aseptic meningitis or meningoencephalitis of undetermined pathogen.
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