Hemorrhagic fever with renal syndrome (HFRS) and reversible splenial lesion syndrome are both considered uncommon conditions relatively rare. Fever, hemorrhage, and acute kidney injury are the prevailing symptoms frequently observed in cases of HFRS. We describe a case of a middle-aged man who had been hospitalized with fever and acute neurological symptoms. His main symptom was recurrent dizziness. Cranial computed tomography (CT) did not reveal any obvious lesions, such as encephalorrhagia or infarctions. The splenium of corpus callosum showed hyperintensity on brain magnetic resonance imaging (MRI), which is in line with the characteristic radiographic observations of reversible splenial lesion syndrome (RESLES). Further analyses revealed that the patient's platelet counts had decreased to 7×109/L while hemorrhagic fever antibodies were positive. Eventually, the patient was diagnosed with HFRS and exhibited clinical improvements after active treatment.