Infectious mononucleosis is a benign, self-limiting disorder caused by primary Epstein-Barr virus (EBV) infection. Various neurological diseases, such as encephalitis, meningitis, acute inflammatory polyneuropathy, Bell's palsy, and peripheral neuropathy, may be associated with EBV infection; however, involvement of the central nervous system (CNS) is relatively uncommon [1][2][3][4][5]. Rare cases of hypoglossal nerve palsy associated with acute EBV infection have been reported in children [6][7][8][9]. Herein, we report a case of unilateral and reversible hypoglossal nerve palsy with acute EBV infection. The study was reviewed and approved by the Ewha Womans University Seoul Hospital Institutional Review Board (IRB File No: SEUMC 2021-07-028). The requirement for informed consent was waived by the board.A previously healthy 17-year-old man presented with a 7-day history of severe left sublingual pain, paralysis, and lingual dysarthria. He had not received any dental treatment in recent years. However, he had recently experienced upper respiratory tract infection symptoms, which included cough and rhinorrhea, for 5 days before he was left with sublingual paralysis. He was barely able to move his tongue to the left side, with severe pain in the tongue that disturbed his sleep and made it difficult to swallow food