Primary cilia are evolutionarily conserved organelles that regulate various aspects of cell development, differentiation, and function. Defects in primary cilia lead to diseases known as ciliopathies, with vision loss as one of the most frequent manifestations. Increasing evidence suggests that in addition to the connecting cilium of photoreceptors in the retina, ciliary defects in other ocular tissues contribute toward the vision loss phenotype seen in ciliopathy patients. This review explores the current literature on the role of primary cilia in the anterior chamber, including the cornea, trabecular meshwork, iris, and ciliary body, and in retinal non-photoreceptor cells, and retinal pigment epithelium.