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To report a case of limbal stem cell deficiency (LSCD) secondary to diffuse non-necrotizing anterior scleritis (DNNAS).Interventional case report with clinicopathologic correlation. A 69-year-old white woman with known Crohn disease presented with DNNAS. The acute inflammatory phase was treated with topical and systemic steroids. After DNNAS, she developed secondary LSCD with loss of limbal palisades of Vogt and conjunctivalization of the corneal surface and corneal haze. She underwent superficial keratectomy combined with autologous limbal stem cell grafting from the fellow eye. The keratectomy specimen was sent for pathological examination.There were no intraoperative or post-operative complications. Histopathology and immunohistochemistry showed a cytokeratin 19–positive and cytokeratin 3– and cytokeratin 12 negative epithelium in keeping with a conjunctival phenotype on the corneal surface.LSCD can be a rare complication of DNNAS. After control of ocular surface inflammation, autologous limbal stem cell grafting and amniotic membrane transplantation can be effective in normalizing the ocular surface.