Background: With the availability of the COVID-19 vaccine, post-vaccination neurological complications have occasionally been reported. Case presentation: We present a case of neuromyelitis optica spectrum disorder (NMOSD) that developed 1 month after the second dose of BIBP COVID-19 vaccine (SARS-CoV-2-Vaccine [Vero Cell] Inactived). The patient presented itching, numbness of the hand and right side of the face, associated with nausea, vomiting and hiccups. Brain MRI showed lesions in the area postrema, medulla, and bilateral hypothalamus, typical of NMOSD. Serum antibodies to anti-AQP4 and anti-MOG were negative. Conclusions: The pathogenesis of NMOSD development and the vaccine is still unknown. The presentation of NMOSD is generally aggressive and disabling, it is important for the neurologist to be attentive to the highly variable clinical presentation after vaccination against COVID-9 for early diagnosis and effective treatment.

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