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Purpose: To report a patient with chronic recurrent multifocal osteomyelitis (CRMO) complicated by optic neuropathy and central retinal artery occlusion (CRAO)., Observations: CRMO is a noninfectious, inflammatory bone disorder. It is thought to be an autoimmune condition related to an imbalance of pro- and anti-inflammatory cytokines. Retinal vasculitis has been reported in a patient with CRMO but not CRAO or optic neuropathy., Conclusions: We expanded the list of ophthalmic involvement of CRMO to include CRAO and optic neuropathy., Competing Interests: None., (© 2021 The Authors. Published by Elsevier Inc.)