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Background Miller Fisher Syndrome (MFS), a rare variant of Guillain-Barre syndrome (GBS), usually presents with the classic triad of ophthalmoplegia, areflexia and ataxia. Ophthalmoplegia is commonly the earliest feature.Method We describe an atypical presentation of MFS.Results A 57-year-old male presented to the emergency department with a 2-day history of unsteadiness while mobilising and non-specific visual complaints. There was a 1-week history of viral upper respiratory tract infection associated with right ear pain. Initial examination revealed mild gait ataxia without truncal or limb ataxia or abnormal sensory findings. Reflexes were intact and eye movements were normal. MRI brain was normal.By day 4 of his admission, he had developed a complex ophthalmoplegia and asymmetric bilateral ptosis; areflexia had developed by day 6. Enhanced skullbase MRI confirmed subtle enhancement of the oculomotor and ophthalmic nerves bilaterally. Cerebrospinal fluid revealed very mildly elevated protein (0.65g/L) and no white cells. Nerve conduction studies, including late responses, were within normal limits. Treatment with intravenous immunoglobulin was commenced. Symptoms stabilised and then gradually improved. The serum Anti-GQ1b antibody returned positive and myasthenic antibodies were not detected.Conclusion The diagnosis of MFS is usually based on clinical presentation and supported by laboratory investigations. This case highlights the importance of (i) revisiting the diagnosis when new clinical features develop, and, (ii) having clinical suspicion for this syndrome even in the absence of some of the hallmark features initially.