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Abstract Background To report a first case of late-onset diffuse lamellar keratitis (DLK) occurring 4 years after femtosecond laser-assisted small incision lenticule extraction (SMILE). Case presentation A 41-year-old man who underwent SMILE 4 years prior developed DLK in the right eye 1 day after he was struck in the eye by a finger while playing with his son. Slim-lamp microscopy and anterior segment optical coherence tomography (AS-OCT) were used to evaluate the cornea of the right eye. Slit-lamp examination of the right eye revealed epithelial exfoliation and stage 3 DLK with diffuse, dot-like, granular haze in the interface between the cap and stromal bed. After intensive treatment with topical corticosteroids, the DLK resolved and corneal transparency was achieved. Conclusions This case indicates that DLK can occur several years after SMILE. Ocular trauma may be a risk factor for the development of DLK. The prognosis is usually favorable with early diagnosis and treatment with topical corticosteroids.