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Purpose: To report a case of iridocorneal endothelial syndrome with pigmentary glaucoma. Methods: We encountered a 44-year-old unilateral glaucoma patient who complained of intermittently decreased visual acuity in the right eye. For differential diagnosis, we carried out ophthalmic and systemic examination. Results: Binocular corrected visual acuity was 1.0. His intraocular pressure, measured using a Goldmann applanation tonometer, was 50 mm Hg in the right eye and 18mm Hg in the left. Upon examination of the right eye, we found pigments with a Krukenberg`s spindle appearance on the corneal endothelium, peripheral anterior synechia extending beyond Schwalbe`s line, trabecular hyperpigmentation, endothelial pleomorphism, polymegathism, cell loss, dark area within the cells, a light central spot and light peripheral zone, retinal nerve fiber layer defects, and visual field defects, which together led to the diagnosis of iridocorneal endothelial syndrome with pigmentary glaucoma. We observed progressed glaucomatous injury upon examination of the disc stereo photograph, retinal nerve fiber layer photograph, and visual field test, even though we used topical IOP reducers. Eventually, we performed a trabeculectomy in the right eye. Conclusions: We experienced a case of iridocorneal endothelial syndrome with unilateral pigmentary glaucoma. The glaucoma was not well-controlled with topical IOP reducers. The glaucoma was then treated by a trabeculectomy. We report this case with a review of the literature.

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