Scrub typhus is an infective acute febrile disorder caused by the intracellular parasite Orientia tsutsugamushi. Neurological manifestations of scrub typhus are meningoencephalitis, cerebellitis, transverse myelitis, papilledema, and cranial nerve palsy. However, opsoclonus-myoclonus syndrome associated with scrub typhus has been rarely reported. A 59-year-old man developed vertigo, nausea, vomiting, and imbalance following scrub typhus infection for eight days. Examination showed eschar at the axilla, decreased mentality, and opsoclonus-myoclonus syndrome. Video-oculography disclosed opsoclonus with an amplitude of 15°–20° and a frequency of 6–8 Hz. The serum antibody titers to Orientia tsutsugamushi were 1:5,120, and cerebrospinal fluid analysis revealed pleocytosis. Brain magnetic resonance imaging was normal. Neurological symptoms and signs completely improved by systemic steroid and antibiotics treatment. Various mechanisms including direct disseminating inflammation or indirect immune modulation may give rise to neurological complications following scrub typhus.