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Coronavirus Disease 2019 (COVID-19) is well documented as a cause of respiratory tract infection. Increasingly, multi-systemic effects, including COVID-19-related neurologic features, are being reported. Here we report, what we believe to be, the first reported case of acute haemorrhagic leukoencephalitis (AHLE) with presence of oligoclonal bands in the cerebrospinal fluid. AHLE is a rare fulminant demyelinating disease, associated with severe COVID-19 infection., Highlights • Acute Haemorrhagic Leukoencephalitis (AHLE) could be a neurological complication of severe COVID-19 infection. • The presence of oligoclonal bands in the CSF could highlight the central inflammatory response. • Here a rare case of AHLE in a severe COVID-19 infection with positive oligoclonal bands in CSF is described.