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Isolated alacrimia is a rare condition often reported with salivary gland agenesis in the literature. Children with the isolated absence of the lacrimal gland can have normal tear film but not during emotional stimuli. Microblepharon is a congenital anomaly with abnormally small eyelids due to vertical shortening, which leads to incomplete blink, exposure keratopathy, and corneal ulceration. The combined occurrence of both the conditions in our case led to ocular surface keratinization and visual impairment. Here, we report a case of isolated lacrimal gland agenesis in association with microblepharon in a 2-year-old child.