We report a case of unilateral acute-onset ptosis and mydriasis in a girl 2 years and 7 months of age from an area endemic for Lyme disease. She lacked other signs of oculomotor nerve palsy. She tested positive for Borrelia burgdorferi and negative for Bartonella henselae on serology and was diagnosed with Lyme neuroborreliosis. The ptosis and mydriasis resolved after 1 week of oral doxycycline.
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