Relapsing-remitting painful ophthalmoplegia due to orbital myositis

We describe a woman with a long history of relapsing-remitting painful ophthalmoplegia in a seasonal pattern, due to an isolated orbital myositis that was - At our observation - Classified in the context of the so-called SAPHO syndrome. She had been previously treated with corticosteroid therapy, but the association with immunosuppressive drugs produced a more prompt resolution of both the headache and ophthalmoplegia, and of the magnetic resonance imaging pictures of isolated myositis as well.