Purpose: Infectious mononucleosis (IM) complicated with a neurological manifestation, including acute cerebellar ataxia, Guillain-Barre syndrome, meningitis, encephalitis, cranial nerve palsies, optic neuritis or transverse myelitis, has been rarely reported; however, IM complicated with acute cerebral infarction has never been reported in the literature.

Case Report: A 49-year-old man with diabetic mellitus suffered from IM with fever, pharyngitis, parotiditis with lymphadenopathies, thrombocytopenia and splenomegaly. After two weeks of conservative treatment, left upper limb paresis and left hemihypesthesia occurred. Neuroimaging demonstrated acute ischemic stroke involving the right frontal lobe. In view of the underlying infection, immediate intravenous rt-PA was not recommended; hence, oral aspirin 100 mg daily was prescribed and he received regular rehabilitation in the subsequent follow up.

Conclusion: Although IM is known to be self-limited, it could contribute to acute cerebral infarction, which is a rare IM neurological complication.

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