We report the first adult case of Influenza A virus infection with acute unilateral oculomotor nerve palsy. Unlike previous reports, our patient showed isolated unilateral oculomotor nerve palsy as soon as she developed general symptoms with Influenza A infection, and demonstrated no significant increases of anti-ganglioside antibodies including anti-GQ1b IgG antibody. She recovered immediately after treatment of oseltamivir phosphate. As for the mechanism by which Influenza A infection caused ophthalmoparesis, small vessel vasculitis due to direct invasion of the virus was speculated. Although influenza encephalitis/encephalopathy including acute necrotizing encephalopathy are most frequently reported in children, it is noteworthy that influenza virus can also cause focal neurological signs such as ophthalmoparesis in adult cases.