Papilledema has previously been reported as the initial presentation of chronic myeloid leukemia (CML). Reported cases of CML have demonstrated bilateral, symmetrically edematous optic nerves attributed to poor CSF absorption given the hyperviscosity induced by an elevated white blood cell (WBC) count. We report an interesting case of unilateral optic disc edema as the first presentation of CML approximately one year prior to substantial elevation of WBC count