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Here, we present a case of Bickerstaff’s brain stem encephalitis secondary to suspected campylobacter infection. Diagnosis was based on the classical presentation; ophthalmoplegia, ataxia, reduced consciousness and hyperreflexia, exclusion of other pathology and was supported by MRI, cerebrospinal fluid and serological testing. Despite the strong association of anti-GM1 antibodies in campylobacter infection and subsequent Guillain-Barré syndrome, this is the first reported case of brainstem encephalitis secondary to campylobacter infection demonstrating positive antiganglioside IgG GM1 serology.