PURPOSE: We studied a case of Fuchs' superficial marginal keratitis in which cystic corneal hydrops developed in one eye, and spontaneous corneal perforation occurred in the fellow eye. METHODS: The patient underwent bilateral annular corneoscleral lamellar patch grafts for tectonic support. RESULTS: The patient had an uncomplicated postoperative course and visual acuity of 20/25 bilaterally. CONCLUSION: Stromal loss in Fuchs' superficial marginal keratitis may be severe enough to result in cystic corneal hydrops. Patients with this disorder should be counseled regarding the possibility of corneal perforation.