To demonstrate a rare case of acute hemorrhagic leukoencephalitis(AHL) presumably secondary to COVID-19 infection and an excellent prognosis due to timely diagnosis and intervention.

A 21-year-old male with history of mild COVID-19 infection 6 weeks prior presented with a prodrome of fatigue and generalized malaise followed by progressive altered mental status and new onset seizure. Brain MRI showed extensive bilateral vasogenic edema of the temporal and parietal lobes with underlying macroscopic hemorrhages and extensive perivascular post-contrast enhancement. CSF protein(107) and WBC of 47(lymphocytic predominant). Extensive evaluation for infectious etiologies in the CSF was unrevealing. CSF AQP4, MOG, oligoclonal bands and IgG Index, and ACE were negative, and serum and CSF autoimmune encephalitis panels were also negative. Serum autoimmune labs including ANA and vasculitis panel were negative. There were no spine lesions. CTA and digital subtraction angiography were without vasculitis changes. Pan-CT to evaluate for malignancy was negative. He was treated with high-dose methylprednisolone and IVIG, with clinical and radiographic improvement. Seizures were controlled with levetiracetam. Prednisone taper was continued at discharge. MRI at one month demonstrated continued radiographic improvement and clinically returned to his prior baseline.

This unique case of AHL presumed secondary to COVID-19 infection (independently diagnosed at two different centers) showed excellent recovery due to early treatment with IVIG and steroids, which likely played a pivotal role in preventing a grave outcome.