Background: This report discusses a case of bilateral optic disc edema associated with celiac disease which resolved after many months of a gluten-free diet. Case report: A 3-year-old white male, recently diagnosed with celiac disease, presented to our eye clinic for comprehensive exam after referral from plastic surgery due to craniosynostosis. Ophthalmic exam detected optic disc edema. Additional testing ruled out pseudo-papilledema, cranio- cephalic isproportion, and venous sinus thrombosis as the cause of the optic disc edema. After many months on a gluten free diet the optic disc edema resolved. 

Conclusion: This case highlights one of the extra-intestinal manifestations of celiac disease and showcases the importance of screening eye exams for all patients with systemic diseases that have ophthalmic implications such as celiac disease and craniosynostosis.