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Background Miller Fisher syndrome (MFS) is considered a rare variant of Guillain-Barré syndrome (GBS), an acute-onset immune-mediated neuropathies characterized by the classic triad of ataxia, areflexia, and ophthalmoparesis. 1 We report a case of atypical MFS presentation.Results A 42-year-old female presented to emergency department with 10 hours of right arm weakness, difficulty swallowing, visual blurring and slurred speech. Neurological examination revealed dysphagia, dysarthria, dysphonia and bilateral ptosis. Extraocular eye movements were full. Right arm strength was grade 3/5 Acute stroke imaging was unremarkable. MRI brain and whole spine showed no acute pathology and no evidence of demyelination. CT chest, abdomen and pelvis showed no evidence of malignancy. Empiric treatment for GBS and myasthenia gravis was initiated with intravenous immunoglobulin (IVIG), pyridostigmine, and IV methylprednisolone 1g for five days. Initial comprehensive serum and CSF work-up was unremarkable. CSF showed normal biochemistry and cells. Nerve conduction studies were within normal limits. Patient started to clinically improve and was discharged home on four weekly IVIG. GQ1b antibody in the serum returned positive confirming the diagnosis of MFS. On three monthly follow-up patient made complete clinical recovery and IVIG was ceased.Conclusion This case demonstrates atypical MFS presentation with stroke like symptoms.Reference Noioso CM, Bevilacqua L, Acerra GM, Della Valle P, Serio M, Vinciguerra C, Piscosquito G, Toriello A, Barone P, Iovino A. Miller Fisher syndrome: an updated narrative review. Front Neurol. 2023 Aug 24;14:1250774. doi: 10.3389/fneur.2023.1250774. PMID: 37693761; PMCID: PMC10484709.