Multiple sclerosis (MS) is a chronic demyelinating disorder of central nervous system (CNS) leading to neurological disability. A wide variety of ophthalmic and neuro-ophthalmic manifestations have been associated with MS. Although this link is well recognized, controversial is still the risk of developing MS in patients with ophthalmic manifestations. Intermediate uveitis followed by panuveitis are the most common manifestations among the ophthalmic involvement in MS. Timely management of uveitis, is a crucial step to prevent irreversible visual impairment. A multidisciplinary team including ophthalmologists together with other specialists is required in the majority of cases for an adequate diagnostic and therapeutic approach of uveitis. Corticosteroids represent the mainstay of therapy in the acute phase, whereas conventional immunosuppressive drugs such as azathioprine may allow a steroid sparing effect. Increasing evidence from anti-CD 20 monoclonal antibodies such as rituximab have proven the efficacy on clinical and radiological outcomes in MS, on the contrary anti-Tumor Necrosis Factor (TNF) monoclonal antibodies such as infliximab and adalimumab are contraindicated in patients with demyelinating disorders, as they seem to worsen disease and neurological clinical symptoms. On this basis, MS-associated uveitis may deeply influence the therapeutic choices suggesting targeting inflammatory cytokines different from TNF. Recently, interleukin (IL)-1 blockade has been investigated as possible therapeutic tool in several inflammatory eye conditions such as Behcet's disease related uveitis, strengthening that IL-1 is also critical for the development of uveitis. Herein, we report our experience in a HLA-B27 positive patient with MS-associated uveitis successfully treated with the short-acting IL-1 receptor antagonist anakinra.