Introduction: This report is about a young lady displaying orolingual dyskinesia (OD) caused by Hashithyrotoxicosis (HTx) which was reversed back to normal by short course of both oral carbimazole and parental steroid. Case Report: A 23 year old female presented with throat discomfort followed by emotional lability, excessive talk, associated with protruding movements of tongue and occasionally facial grimacing like movements suggestive of OD. Further, she was tachycardic and had diffuse tender goiter. Her TSH was less than 0.015 mIu/mL and T4 was 4.62 nmoles/L while the anti-TPO antibodies were elevated to 1903 IU/mL. Her CSF protein was disproportionately elevated (150 mg/dL) with no cells. These features suggested Hashitoxic encephalopathy. This patient was started with oral carbimazole 15 mg thrice a day for the management of thyrotoxicosis. Two weeks later she was started with intravenous methyl-prednisone to achieve complete recovery of Hashimoto's encephalopathy. At eighth week she achieved 90% clinical improvement and her anti-TPO level reduced to 1020 IU/mL. Conclusion: OD along with elevated anti-TPO antibodies and thyrotoxicosis could be an indicator of Hashimoto's toxic encephalopathy; that is not yet well studied.