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Objective: There is no report on Bickerstaffs brainstem encephalitis (BBE) patients in China. We here report the first case of BBE in China. Methods: Clinical features, results of electromyography, electroencephalography (EEG), magnetic resonance imaging (MRI) and cerebrospinal fluid (CSF) examination were studied to clarify the characteristics of this syndrome.Results: A 44-year-old man presented himself at our inpatient department with somnolence and dizziness as his initial symptoms.He developed multiple cranial nerves paralysis especially internal and external ophthalmoplegia, ataxia and tetraparesis within 1 week. His condition rapidly deteriorated, and he experienced coma. Electromyography showed indications of peripheral nerve dysfunction, electroencephalography revealed loss of basic rhythm, MRI demonstrated high-intensity abnormalities on T2-weighted images of medulla oblongata, and CSF albuminocytological dissociation was defined abnormally as high protein. Ten similar clinically; BBE and FS were proposed to be the variant of GBS.