BACKGROUND: Opsoclonus myoclonus syndrome (OMS) is a rare neuro-ophthalmic disorder characterized by chaotic, multidirectional saccadic eye movements. It is associated with various etiologies, including infectious and immune-mediated encephalitis. Scrub typhus, caused by OBJECTIVE: To highlight scrub meningoencephalitis as a potential cause of OMS and emphasize the importance of prompt recognition and early treatment to enhance patient outcomes. MATERIALS AND METHODS: We describe the case of a 32-year-old female presenting with fever, altered sensorium, and saccadomania, along with a characteristic eschar, diagnosed with scrub meningoencephalitis. Clinical, laboratory, and imaging findings, as well as treatment modalities, are presented. RESULTS: The patient demonstrated generalized jerky movements and rapid eye movements in all directions, consistent with saccadomania. Diagnosis was confirmed through positive IgM enzyme-linked immunosorbent assay (ELISA) for scrub typhus and a characteristic eschar. Treatment with doxycycline and dexamethasone resulted in significant symptom improvement within days, with complete resolution by day 10. CONCLUSION: This case underscores the importance of considering scrub meningoencephalitis as a potential cause of OMS, particularly in endemic regions. Prompt recognition and initiation of appropriate antibiotic therapy, along with adjunctive corticosteroids, are crucial for favorable patient outcomes. Increased awareness among clinicians can lead to timely diagnosis and management, preventing potential neurological sequelae associated with this treatable condition.