Ocular involvement is common in patients with leukae- mia principally with choroidal and retinal infiltration. However, leukemic vitreous infiltration is extremely rare and very few spo- radic cases have been reported in the literature. We present a case of chronic myelogenous leukaemia associated with an extensive vitreous infiltration. History and Signs A 35-year-old man recently diagnosed with chronic myelogenous leukemia in myeloid blast crisis, complained of decrease of vision and visual field defects in his right eye. Visual acuity (VA) was 0.6 in the right and 1.0 in the left eye. Fundus examination revealed bilateral choroidal and reti- nal infiltrates; additionally, the right eye showed a large, whitish vitreous lesion. Therapy and Outcome Pars-plana vitrectomy (PPV) of the right eye was performed. Cytopathologic and immunohis- tochemical examination confirmed leukemic vitreous invasion. Postoperative follow-up was uneventful, and the vitreous remained clear. The patient underwent systemic chemotherapy. Conclusions Chronic myelogenous leukaemia can exceptionally be complicated by vitreous involvement. Intensive systemic chemotherapy may be sufficient treatment while PPV is indicated in atypical cases.