Paradoxical Anatomic Response to Topical Carbonic Anhydrase Inhibitor in X-linked Retinoschisis

An 11-year-old boy presented for central vision blurring in each eye. Visual acuity was 20/80 and examination revealed spoke-wheel foveal schisis and peripheral elevated diaphanous inner retina in each eye. Spectral-domain optical coherence tomography showed inner-retinal, flat-topped cysts in each eye. Electrophysiologic testing was refused, but a clinical diagnosis of X-linked retinoschisis was made. Three months after topical dorzolamide (Trusopt; Santen Pharmaceutical, Osaka, Japan) was started, the macular cysts worsened significantly. The medication was stopped and 3 months later, the macular anatomy returned to baseline. Physicians should be aware of this potential paradoxical anatomic response to topical carbonic anhydrase inhibitor therapy in X-linked retinoschisis. [ Ophthalmic Surg Lasers Imaging Retina . 2018;49:142–144.]