Optic neuritis (ON) is one of the most common manifestations of central nervous system involvement caused by various etiologies.\nLyme ONis an exceedingly rare ocular manifestation of Lyme disease (LD) and only a few cases have been published in the literature.\nLyme ON is very rare but should be included in the differential diagnosis in unexplained cases, particularly in Lyme endemic areas.\nCareful and detailed examination and investigation are warranted to make the diagnosis.We report this case to increase awareness\nof clinicians to include Lyme disease in differential diagnosis of ON for unexplained cases of ON. Herein we present a unique case\nwith a unilateral ON caused by LD along with pre- and posttreatment findings and literature review.
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