Myositis has been reported as a rare manifestation of Lyme disease, and the Lyme disease spirochetes can be an important consideration in the differential diagnosis of unusual cases of myositis, especially in patients who live in or travel to endemic areas. We report the case of two patients who presented with focal orbital myositis which are rare localization for Lyme disease. Myositis were confirmed by magnetic resonance imaging. Diagnosis criteria for Borrelia burgdorferi (B. burgdorferi) infection was supported by (i) medical history (tick bite in an endemic area), (ii) systemic clinical findings (Erythema migrans, neurological manifestation or arthritis), (iii) positive Lyme serology and/or the detection of B. burgdorferi DNA by polymerase chain reaction, as well as (iv) exclusion of other infectious and inflammatory causes. The current cases are reviewed in the context of findings from previous myositis descriptions.