Tick-borne encephalitis (TBE) is an uncommon and potentially severe illness. TBE virus is transmitted to the humans by an infected tick and it spreads to the central nervous system determining various clinical pictures. A case in which TBE virus caused an encephalomyelitis with quadriplegia and respiratory insufficiency that persist one year after the diagnosis, is reported. Such a clinical manifestation of TBE has never been described in Italy till now.